Physical symptoms in Young children with Cancer: comparison of electronic versus paper-and-pencil diaries

Childhood cancer is the leading cause of death from disease in 1- to 14-year-olds with greater incidence among children 6 and younger. Young children are often excluded from studies because of their developmental abilities. Since these children primarily receive outpatient treatment, their parents are the ones managing symptoms. The purpose of this dissertation was to describe the physical symptoms of children 6 and younger and to compare two symptom diaries. Specific aims were to (a) describe the frequency and severity of physical symptoms as reported by parents; (b) compare parents' perceived usefulness of an electronic symptom diary to a paper-and-pencil symptom diary, and (c) explore the usability of an electronic diary to report symptoms. This study used a mixed-methods, crossover design. Participants were 20 parents of children with cancer and their children. Parents used two types of symptom diaries to report symptoms. Symptom data were collected over two, 7-day periods spanning three ambulatory visits. At visits 2 and 3, parents completed two questionnaires along with a short interview. Descriptive statistics were used to summarize symptoms and Wilcoxon signed-ranks tests were used to compare questionnaire responses. Qualitative data were analyzed using content analysis. Nineteen (95%) parents reported at least one daily symptom and nine (45%) reported at least 1 day of a moderate/severe symptom. Fatigue was the most commonly iv reported symptom (56.8%) followed by nausea (51.4%) and lack of appetite (37.8%). The symptom most frequently reported as moderate/severe was fatigue (17.5%). Parents most frequently used their child's activity level and verbal cues to determine symptom presence. Parents' satisfaction (z = -3.454, p = .001) and parents' perceived usefulness (z = -3.061, p = .002) of the electronic diary was greater than that of the paper. This study provides insight into how parents perceived symptoms and their associated characteristics in young children. Young children with cancer experience persistent symptoms, sometimes of moderate or greater severity even in the ambulatory setting. Parents preferred the electronic diary because of its features, tracking capabilities, and familiarity. This study fills a gap in the existing body of literature by exploring symptoms in this population.

PHYSICAL SYMPTOMS IN YOUNG CHILDREN WITH CANCER: COMPARISON OF ELECTRONIC VERSUS PAPER-AND-PENCIL DIARIES by Sharifa M. Al-Qaaydeh A dissertation submitted to the faculty of The University of Utah in partial fulfillment of the requirements for the degree of Doctor of Philosophy College of Nursing The University of Utah August 2019 Copyright © Sharifa M. Al-Qaaydeh 2019 All Rights Reserved The University of Utah Graduate School STATEMENT OF DISSERTATION APPROVAL Sharifa M. Al-Qaaydeh The dissertation of has been approved by the following supervisory committee members: Lauri Linder , Chair 04/29/19 Date Approved Bob Wong , Member 04/29/19 Date Approved Katherine Sward , Member 04/29/19 Date Approved Christina Baggott , Member Date Approved Susan Beck , Member Date Approved and by Barbara Wilson the Department/College/School of and by David B. Kieda, Dean of The Graduate School. , Chair/Dean of Nursing ABSTRACT Childhood cancer is the leading cause of death from disease in 1- to 14-year-olds with greater incidence among children 6 and younger. Young children are often excluded from studies because of their developmental abilities. Since these children primarily receive outpatient treatment, their parents are the ones managing symptoms. The purpose of this dissertation was to describe the physical symptoms of children 6 and younger and to compare two symptom diaries. Specific aims were to (a) describe the frequency and severity of physical symptoms as reported by parents; (b) compare parents’ perceived usefulness of an electronic symptom diary to a paper-and-pencil symptom diary, and (c) explore the usability of an electronic diary to report symptoms. This study used a mixed-methods, crossover design. Participants were 20 parents of children with cancer and their children. Parents used two types of symptom diaries to report symptoms. Symptom data were collected over two, 7-day periods spanning three ambulatory visits. At visits 2 and 3, parents completed two questionnaires along with a short interview. Descriptive statistics were used to summarize symptoms and Wilcoxon signed-ranks tests were used to compare questionnaire responses. Qualitative data were analyzed using content analysis. Nineteen (95%) parents reported at least one daily symptom and nine (45%) reported at least 1 day of a moderate/severe symptom. Fatigue was the most commonly reported symptom (56.8%) followed by nausea (51.4%) and lack of appetite (37.8%). The symptom most frequently reported as moderate/severe was fatigue (17.5%). Parents most frequently used their child’s activity level and verbal cues to determine symptom presence. Parents’ satisfaction (z = –3.454, p = .001) and parents’ perceived usefulness (z = –3.061, p = .002) of the electronic diary was greater than that of the paper. This study provides insight into how parents perceived symptoms and their associated characteristics in young children. Young children with cancer experience persistent symptoms, sometimes of moderate or greater severity even in the ambulatory setting. Parents preferred the electronic diary because of its features, tracking capabilities, and familiarity. This study fills a gap in the existing body of literature by exploring symptoms in this population. iv This dissertation is dedicated to my children, Sajed and Summer. They are the reason for all that I do. TABLE OF CONTENTS ABSTRACT ......................................................................................................................iii LIST OF TABLES ..........................................................................................................viii ACKNOWLEDGEMENTS .............................................................................................. ix Chapters 1 INTRODUCTION .......................................................................................................... 1 Statement of the Problem ....................................................................................... 1 Study Purpose and Specific Aims .......................................................................... 2 Theoretical Framework .......................................................................................... 3 Significance of the Study to the Field of Health Care ............................................ 4 2 REVIEW OF THE LITERATURE ................................................................................ 5 Introduction ............................................................................................................ 5 Cancer in Young Children ...................................................................................... 5 Early Child Development ....................................................................................... 7 Symptoms in Children With Cancer ....................................................................... 8 Assessment of the Literature ................................................................................ 11 Parent Proxy Reports of Symptoms ..................................................................... 13 Technology to Track Symptoms .......................................................................... 14 Theoretical Framework: Sociotechnical Framework ........................................... 17 Summary ............................................................................................................... 20 3 METHODS ................................................................................................................... 27 Design ................................................................................................................... 27 Sample .................................................................................................................. 28 Setting ................................................................................................................... 29 Procedures ............................................................................................................ 29 4. PARENTS’ REPORT OF PHYSICAL SYMPTOMS IN YOUNG CHILDREN WITH CANCER ...................................................................................................................... 41 Abstract .................................................................................................................. 41 Parents’ Report of Physical Symptoms in Young Children With Cancer ............. 42 Symptoms in Children With Cancer .....................................................................42 Parents as Proxy ................................................................................................... 45 Symptom Diaries .................................................................................................. 47 Study Purpose and Framework...............................................................................47 Methods ................................................................................................................ 48 Results .................................................................................................................. 52 Discussion ............................................................................................................. 56 5 PERCEIVED USEFULNESS AND USER SATISFACTION OF ELECTRONIC AND PAPER-AND-PENCIL DIARIES IN YOUNG CHILDREN WITH CANCER ...................................................................................................................... 69 Abstract ................................................................................................................. 69 Background ........................................................................................................... 70 Methods ................................................................................................................ 74 Results .................................................................................................................. 80 Discussion ............................................................................................................. 84 Conclusion ............................................................................................................ 88 6 DISCUSSION AND CONCLUSIONS ........................................................................ 96 Discussion ............................................................................................................. 96 Recommendations for Future Research .............................................................. 100 Recommendations for Practice ........................................................................... 102 Recommendations for Policy ............................................................................. 103 Limitations .......................................................................................................... 103 Summary ............................................................................................................. 104 Appendices A: PARENT INTERVIEW QUESTIONS ..................................................................... 106 B: PAPER-AND-PENCIL VERSION OF THE ICHEMO DIARY ............................. 107 C: QUIS 7.0 SECTION ................................................................................................. 108 D: PERCEIVED USEFULNESS QUESTIONS ADAPTED FROM THE TAM ......... 109 REFERENCES ................................................................................................................ 110 vii LIST OF TABLES Table 2.1 Publications Including Children With Cancer < 7 Years of Age .............................. 21 2.2 Application of the Sociotechnical Framework .......................................................... 26 3.1 Description of Symptoms in the Symptom Diary and Corresponding Measurements ............................................................................................................ 38 3.2 Summary of Data Collection and Measures .............................................................. 39 4.1 Parent Interview Questions ........................................................................................ 62 4.2 Participant Characteristics ......................................................................................... 63 4.3 Symptom Frequency and Prevalence by Diary ......................................................... 64 4.4 Frequency and Prevalence of Symptom Severity ...................................................... 65 4.5 Identifying Presence and Severity of Symptoms by Categories and Subcategories .. 66 4.6 Wilcoxon-Signed Ranks Comparing Symptom Identification Responses by Diary Type ................................................................................................................. 67 4.7 Symptom Relevance .................................................................................................. 68 5.1 Application of the Sociotechnical Framework .......................................................... 90 5.2 Parent Demographics ................................................................................................. 91 5.3 Wilcoxon Signed-Ranks Test Results Comparing TAM and QUIS 7.0 Responses Between Diaries ....................................................................................... 92 5.4 Categories and Subcategories of Parents’ Responses to Interview Questions .......... 93 5.5 Wilcoxon-Signed Ranks Comparing Diary Use Responses by Diary Type ............. 94 5.6 Diary Preference and Rationale ................................................................................. 95 ACKNOWLEDGEMENTS I started this dissertation journey many years ago, and throughout this time, many people have contributed, supported, and mentored me along the way. I would like to thank and express my gratitude to: • All of the members of my supervisory committee. They have been more supportive and instrumental than they even realize. • My colleagues at the University of Utah College of Nursing for all of their encouragement and help with navigating children, work, and school. • Primary Children’s Hospital for allowing me to conduct my research in such a supportive environment. • All of the amazing and resilient children and families I had the pleasure of meeting and who were willing to participate despite cancer. • Sigma Theta Tau International, Gamma Rho Chapter for the generous research grant that allowed me to purchase necessary supplies for this study. • My friends and family who always pushed and encouraged me even throughout difficult life circumstances. I will never forget their help and will be forever grateful. CHAPTER 1 INTRODUCTION Statement of the Problem Childhood cancer is the leading cause of death from disease among 1- to 14-yearolds in the United States, with over 10,000 children less than 15 years of age diagnosed each year (Noone et al., 2017). The most common types of pediatric cancer are leukemia and central nervous system (CNS) tumors. The incidence rates of cancer are greatest among children less than 6 years of age and in adolescents. Although the incidence of the most common types of childhood cancer is higher among children less than 6 years of age, these young children frequently are excluded from studies addressing symptoms and symptom management for children with cancer. As a result, an understanding of the symptom experience of infants, toddlers, and preschool children with childhood cancer is limited. Symptoms in young children with cancer can be difficult to assess because of these children’s’ limited ability to express themselves verbally, recall information, and specify exact symptoms due to their developmental stage (Linder, 2008; Riggins, Miller, Bauer, Georgieff, & Nelson, 2009). Thus, the primary caregiver for the child often is the one who observes, interprets, and presents symptoms to the health care provider on the child’s behalf. Because all cancer patients of every age experience symptoms, young children 2 need to have their symptoms assessed and reported. This symptom assessment should also address the occurrence and severity of symptoms. The treatment for many types of childhood cancers is delivered in outpatient settings with intervals between visits ranging from approximately 10 days to 6 weeks. This pattern of follow-up and the fact that parents must manage symptoms at home makes it difficult for parents of young children to keep their healthcare provider apprised of what symptoms their child is experiencing daily, or even to remember exact timings for particular symptoms. Communication between provider and parent can be delayed, and children may suffer as a result of symptoms not conveyed to healthcare providers. Providing a symptom tracking tool for parents to report their child’s symptoms could improve communication as well as provide evidence and insight into what symptoms young children with cancer are experiencing as well as when they are occurring. Study Purpose and Specific Aims The purpose of this study was twofold: (a) to describe the physical symptoms of children less than six years old with cancer and (b) to compare traditional paper-andpencil symptom tracking diaries and an electronic symptom tracking diary as tools to support parents in reporting their children’s symptom experience. Having a tool that parents could download and use to record their child's symptoms daily could help to (a) provide insight into what symptoms young children with cancer are experiencing and (b) alleviate some of the communication barriers between parents and providers by providing a report of symptoms experiences. The findings could also provide evidence related to aspects of the usability of a smartphone application for parents to report and track symptoms of young children with cancer. For pediatric patients being cared for in an 3 outpatient environment, this tool could be used to improve communication and allow for early intervention in the future. The primary aims of the study were as follows: 1. To describe the occurrence, frequency, and severity of collective physical symptoms of children less than 6 years of age with cancer as reported by their parents; 2. To compare parents’ perceived usefulness of a smartphone application symptom diary to a paper and pencil symptom diary in reporting symptoms to their child’s provider; and 3. To explore the usability of using a smartphone application to report symptoms experienced by children with cancer as reported by their parents. Theoretical framework The overarching framework for this study was the sociotechnical framework, adapted from the approach described by Barber, Cornford, and Klecun (2007). The sociotechnical framework describes how humans and technology interact and how this interaction can affect outcomes. For this study, the description by Barber and colleagues (2007) has been modified to exclude the organizational component of the model because this study will not involve the implementation of the technology on an organizational level. Because the sociotechnical framework is considered a high-level theory, the descriptions within the framework are broad. Thus, the physical component, symptom experience, from the University of California, San Francisco (UCSF) symptom management theory has been added to augment the social component of the sociotechnical framework. The concept of “Perceived Usefulness” from the Technology 4 Acceptance Model has also been included to gain parent perspectives on the use of the two types of diaries which influences usability (Davis, 1989). Usability is “the quality of a user’s experience when interacting with products or systems” (usability.gov, n.d.). Although many aspects influence usability, perceived usefulness of a given technology can help determine whether or not a person will use that technology (Holden & Rada, 2011). The use of these frameworks, in conjunction with the sociotechnical framework, is described in greater detail in the Review of Literature section (Chapter 2). Significance of the Study to the Field of Health Care The main strength of this study is that it addresses a significant gap in the science of symptom management for children with cancer. Little is understood about symptoms in young children with cancer and implementing current technology as a resource to support parents in monitoring and responding to their child’s symptoms. This study also evaluates the perceived usefulness and usability of a symptom-tracking tool to provide insight into how parents used the information they tracked and if a tool for tracking would be useful in the future. CHAPTER 2 REVIEW OF THE LITERATURE Introduction This chapter presents the current literature regarding the symptom experience in children less than 6 years of age with cancer. The databases, Cumulative Index to Nursing and Allied Health Literature (CINAHL), PubMed, and PsycINFO were utilized to search for relevant literature. Searches included the terms: “childhood cancer” and “symptoms” and included the years 2000 through December 2018. The search was limited to those studies that included children less than 6 years of age, those receiving curative treatment and articles published in English. Because literature addressing symptoms in young children was limited, children with cancer of all ages were also examined for this review. Literature using the search terms “proxy-reporting” and “symptom diaries” were also included in this search. Cancer in Young Children Prevalence and Incidence Childhood cancer is the leading cause of death from disease among children one to fourteen years of age (National Cancer Institute, 2018). Acute lymphoblastic leukemia (ALL) is the most prevalent type of childhood cancer representing approximately 25% of all childhood cancer cases (PDQ Pediatric Treatment Editorial Board, 2018). Rates provided by 6 the National Cancer Institute have also noted a four-fold greater incidence of ALL among children aged 2-3 years compared to infants and older children (Howlader et al., 2015; PDQ Pediatric Treatment Editorial Board, 2018). CNS tumors are the second most common cancer in children with approximately 2,500 children diagnosed each year, with over half diagnosed in children less than 6 years of age (National Cancer Institute, 2014). Additionally, over the past 20 years, the incidence of invasive forms of childhood cancer has increased (National Cancer Institute, 2014). Among the most common five cancer sites, the rates of cancer in children less than 6 are higher than other age groups with a peak incidence of 24.3/100,000 at age 2 (National Cancer Institute, 2014). The rates begin to decline after age 6 and then increase again in midadolescence (National Cancer Institute, 2014). Definitions A symptom is defined as a “physical or mental problem that a person experiences” which “cannot be seen and do not show up on medical tests” (National Cancer Institute, n.d.). Terms often used to describe symptoms are symptom prevalence/occurrence, symptom characteristics, and symptom patterns and trajectory. Symptom prevalence/occurrence refers to the presence or absence of a symptom while symptom characteristics which refer to the severity or intensity, frequency, and distress of symptoms. Symptom patterns and trajectory refer to how the prevalence of a symptom and its characteristics change over time. This study will report the occurrence and severity of symptoms daily as reported by parents. The severity will be based on the greatest perceived severity of the symptom during the day and will be reported as mild, moderate, or severe. 7 Childhood Cancer Treatment Treatment regimens for childhood cancer vary considerably due to many factors which including the child’s age at diagnosis, gender, primary cancer site, cytogenetic properties of the cancer cells, white blood cell counts at the time of diagnosis, the presence or absence of tumor markers, and stage of cancer at diagnosis (Brodeur & Maris, 2010). However, the mainstay of childhood cancer treatment is chemotherapy. More and more childhood cancers are treated with chemotherapies in the ambulatory setting (Hendershot et al., 2005; National Center on Caregiving, 2015). Thus, much of the child’s treatment-related supportive care is provided by parents in the home setting. The majority of children diagnosed with cancer in the United States receive treatment based on clinical trials offered through the Children’s Oncology Group (COG). The COG is the consortium for childhood cancers for over 200 hospitals, universities and cancer centers and is an NCI clinical trials group. Since the creation of the COG, childhood cancer cure rates have increased dramatically, and children treated at COG institutions have significantly higher survival rates. In the United States, nearly 90% of children and adolescents with cancer receive care at COG-affiliated institutions (“Children’s Oncology Group: About Us,” n.d.). Early Child Development From birth through 6 years of age, children undergo many key physical, emotional, social, and intellectual changes. Language development is increasing from infancy; however, many young children cannot fully convey feelings, emotions, or pain through the use of language alone (Magnusson & Cairns, 2009). Young children with cancer can have difficulty participating in conversations about symptoms because of their 8 limited verbal ability as well as their fear of making their parents worried (C M Ruland, Hamilton, & Schjodt-Osmo, 2009). In previous studies examining symptoms in children with cancer, children were noted to describe symptoms as “feelings” and did not often refer to symptoms as individual experiences but rather a collective experience of how they felt at the time (Woodgate, Degner, & Yanofsky, 2003). During this stage of early development, young children also tend to be presentoriented and have trouble recalling information beyond the past few days. They also have limited attention spans and typically have an exaggerated view of timing (Hagan, Duncan, & Shaw, 2008). Young children begin developing memory ability during the first year of life; however, their ability to recall information like that of adults is typically not present until early adolescence (Magnusson & Cairns, 2009; Riggins et al., 2009). These developmental characteristics make young children’s ability to respond to questions with a recall period of days to weeks challenging. Symptoms in Children With Cancer The experience of symptoms in young children with cancer remains largely unexplored. To date, adolescents and school-age children have been the focus of the majority of the research (Pinheiro et al., 2018; C M Ruland et al., 2009; D. Tomlinson et al., 2017; Vatne, Slaugther, & Ruland, 2010; Walker, Gedaly-Duff, Miaskowski, & Nail, 2010). Current research involving school-age children and adolescents indicates that they experience as many as 10 co-occurring symptoms that can be intermittent or constant and can vary in severity (Christina Baggott et al., 2010; Kestler & Lobiondo-Wood, 2012; Yeh, Chang, & Chang, 2005). However, many children and adolescents reported a high number of moderate and severe symptoms (Baggott et al., 2010; Collins et al., 2000; 9 Ruland et al., 2009; Yeh, Chang, & Chang, 2005). The most common symptoms were: nausea, vomiting, fatigue, hair loss, fever and pain (Collins et al., 2000; Hinds, Quargnenti, & Wentz, 1992; Pöder, Ljungman, & Von Essen, 2010; Williams, Schmideskamp, Ridder, & Williams, 2006). Young children were often excluded from research due to their age-related developmental abilities (Linder, 2008). Across all study samples that included children 6 years of age and younger, the most commonly reported symptoms were fatigue, nausea, anorexia, drowsiness, and pain (Abu-Saad Huijer, Sagherian, & Tamim, 2013; Dupuis, Ethier, Tomlinson, Hesser, & Sung, 2012; Kestler & Lobiondo-Wood, 2012; Linder, Bratton, Nguyen, Parker, & Wawrzynski, 2018; Miller, Jacob, & Hockenberry, 2011; Pöder et al., 2010; Rodgers, Krance, Street, & Hockenberry, 2014; Williams et al., 2006). When children less than 7 years were compared separately from older children, the most distressing symptoms reported were pain, confinement, and a feeling of alienation. However, the younger children generally had their parents proxy-reporting on their behalf compared with the older children who provided self-reports (Hedström, Haglund, Skolin, & Von Essen, 2003). Table 2.1 provides a summary of studies that included children 6 and younger with cancer. The most bothersome symptoms did vary among children. When parents were proxy-reporting, missing activities, worry over the disease and aching bones were reported as most severe. Parents of older children (8-18 years) also perceived them as having more severe and distressing symptoms than parents of younger children (4-7 years) (Dupuis et al., 2010). Children of all ages often reported nausea and pain as the most severe and distressing symptoms (Hedström et al., 2003; Miller et al., 2011; 10 Rodgers et al., 2014). Limited quantitative research has been conducted with children as young as 3 (Hedström et al., 2003; Dupuis, Lau, & Greenberg, 2001; Tomlinson et al., 2017). A visual analogue scale for nausea was used as a self-report measure for children between ages 3-4.9 years. In this study, children were able to self-report and had similar reported frequencies of nausea as older age groups (>5 years) (Dupuis et al., 2001). Other studies included children between the ages of 4-7 as one age group to compare with older children (7-18 years). For these studies, parent report was utilized for the younger children and self-report for the older children. 4- to 7-year-olds had similar reports of pain and rates of disrupted sleep as older participants in these studies (Adlard et al., 2004; Hedström et al., 2003; Zupanec, Jones, & Stremler, 2010). Several qualitative studies examining symptoms and the symptom experience have included younger participants. The most notable of these is the series of studies conducted by Woodgate and colleagues in which children as young as four were interviewed and observed (Woodgate, 2008; Woodgate et al., 2003; Woodgate & McClement, 1998). In these interviews, the family was also included in the interviews and observations. However, like most studies, only 13% of those interviewed were less than six years of age. From this qualitative work, important glimpses into the symptom experience of young children were discovered. First, children often define their symptoms in terms of how they feel and not specifically what symptom they are experiencing. Woodgate and colleagues (2003) describe this finding, “….children’s cancer symptoms were experienced as overall feeling states with assigned meaning” (p. 812). The children would often use terms such as “I feel sick” or “I feel yucky” which 11 can make it hard to determine specifically what symptoms were experienced (Woodgate et al., 2003, p. 807). Thus, it can be difficult to extrapolate specific symptoms from children as they may be experiencing multiple symptoms at one time but assign just one feeling to describe how they feel (Hedström et al., 2003; Woodgate et al., 2003). Among children less than three years of age, a “feeling of confinement” was the most distressing for them and their parents while the “feeling of alienation” was most distressing for children aged 4 to 7 as reported by their parents (Hedström et al., 2003). These feelings most likely relate to frequent appointments and separation from the rest of the world when undergoing treatment. Table 2.1 summarizes the articles found in the review of literature, age of the sample, population, symptoms studied, and type of data collected. Assessment of the Literature The Gap in Age Group Few studies of the cancer symptom experience include children younger than 6 years of age (Docherty, 2003; Hedström et al., 2003; Kestler & Lobiondo-Wood, 2012; Linder, 2008; Tomlinson et al., 2017; Woodgate et al., 2003). Most studies addressing symptoms in children with cancer are conducted with children ages 10-18, and those studies that include younger children often combine age groups of participants (Kestler & Lobiondo-Wood, 2012; Linder, 2008; Ruland et al., 2009; Williams et al., 2012). Although older children can respond to traditional self-report symptom measures, the highest rates of childhood cancer occur among children less than six years of age. Additionally, most of the instruments developed to measure symptoms also excluded children less than 6 years of age. Lack of research in younger children with cancer has created a gap related to knowledge about symptoms in children younger than 12 7. The consequence of this gap is that the symptom experience in young children is relatively unknown or data from children of older ages are extrapolated to younger children, which may not be accurate. A gap in knowledge exists, which could mean the potential for inadequate symptom management and decreased health-related quality of life. Identifying symptoms and their associated severity in children with cancer poses many challenges, especially in young children. Young children can have difficulty understanding symptom meanings, explaining symptoms to parents or providers or sometimes refuse to participate in studies (Scolnik, Atkinson, Hadi, Caulfeild, & Young, 2003; Tomlinson et al., 2017; Tomlinson et al., 2016; Vatne et al., 2010). The Gap in Measurement and Design Many studies of symptoms in children focus on one symptom or individual symptoms. Only within the past 20 years, has the use of instruments to examine multiple symptoms been addressed (Baggott, Dodd, Kennedy, Marina, & Miaskowski, 2009). However, these instruments have only been created for and utilized in children greater than 7 years of age (Collins et al., 2000; Collins et al., 2002; Docherty, 2003; Linder, 2005). Most of the studies used a cross-sectional approach to the symptom experience rather than longitudinal or repeated measures to examine symptoms over time. Few examined symptoms at multiple time points (Docherty, 2003; Woodgate et al., 2003; Woodgate & McClement, 1998). Much of the research has focused on the individual symptom experience rather than the family experience. For young children, involving the parents is important because they are the ones who interpret, respond, and treat symptoms for their child. As 13 some findings suggest, self-report may not always be the best method to collect data in children as children often tire easily when having to fill out surveys or checklists (Woodgate et al., 2003). Multiple methods could be useful in capturing the symptom experience in children. Sample sizes for most of the studies addressing childhood cancer symptoms were relatively small, primarily due to the relative rarity of childhood cancer, limited multi-site studies, and the vulnerable nature of children. Additionally, most of the knowledge known about symptoms in children with cancer was collected on White, non-Hispanic children. Research is needed in children of other races and ethnicities (Docherty, 2003; Kestler & Lobiondo-Wood, 2012; Tomlinson et al., 2017). Parent Proxy Reports of Symptoms Although self-report is seen as the gold standard for identifying symptoms, not all individuals have the ability to identify their symptoms using traditional self-report instruments. Young children have limited ability to express their symptoms to others because of their developmental abilities, including verbal and cognitive abilities. Young children also have limited ability to recall beyond the present, limiting their ability to respond to traditional self-report instruments with varied recall periods such as the past week or past month. Additionally, many parents of young children with cancer feel that their child may be too ill or sick to report (Ruland et al., 2009; Woodgate & McClement, 1998). For these reasons, most research in symptoms and children with cancer using selfreport has been conducted with children aged 7 and older which is consistent with developmental aspects of children younger than 7 years (Linder, 2005; Sherifali & Pinelli, 2007). 14 In school-age children and adolescents with cancer, agreement in self-reports between parent and child in the reporting of symptoms such as nausea, vomiting, and quality of life have been demonstrated. Concordance among parents’ and children’s reports have been observed for many symptoms, primarily physical symptoms with more observable manifestations, among 10-18 year-olds (Baggott, Cooper, Marina, Matthay, & Miaskowski, 2014b; Hyslop et al., 2018; Lo & Hayman, 1999; Speyer, Herbinet, Vuillemin, Chastagner, & Briançon, 2009). Concordance between parent and child reports of psychosocial symptoms, however, is lower (Baggott, Cooper, Marina, Matthay, & Miaskowski, 2014a; Hyslop et al., 2018; Lo & Hayman, 1999). In children younger than 6 years of age, self-report can be extremely hard to obtain; thus, despite the limitations, proxy-reporting may serve as the most reliable source of information on more objectively observed symptoms. Technology to Track Symptoms Initial research with adolescents and adults with cancer supports the feasibility of mobile technology to support symptom tracking. Among adolescents, the work of Gibson and colleagues (2010) utilized a personal digital assistant (PDA) to deliver questions derived from the Memorial Symptom Assessment Scale (MSAS) before, during and after chemotherapy (Gibson et al., 2010). Mobile symptom diaries have also been used in adult cancer patients and adolescents and young adults with cancer (Blondin, Abu-Hasaballah, Tennen, & Lalla, 2010; Rodgers et al., 2014). A mobile phone-based symptom diary created for adolescents and young adults showed the feasibility and had a high adherence of >90% (Baggott et al., 2012). Studies with adults have included various forms of technology to report 15 symptoms. Smartphones and telephone-based computerized systems have been used in adults with various types of cancer to report chemotherapy-related symptoms/toxicity to their healthcare provider directly (Beck, Eaton, Echeverria, & Mooney, 2017; McCann, Maguire, Miller, & Kearney, 2009; Mooney, Beck, Friedman, & Farzanfar, 2002; Mooney, Beck, Friedman, Farzanfar, & Wong, 2014). In these studies, participants reported that technology-based symptom reporting improved communication with providers, improved symptom management, and provided a feeling of security in knowing someone was aware of their symptoms. Both studies also reported high patient satisfaction with each technology-based resource. Studies evaluating the usability and perceived usefulness of this technology to support symptom tracking by families of children with cancer are sparse. Smartphones have been used in adults with various types of cancer to support reporting chemotherapyrelated symptoms/toxicity to their healthcare provider directly (McCann et al., 2009). Apps are being developed and utilized for cancer patients to not only track symptoms but also to receive answers to questions and to learn about various cancers and treatments (Carroll et al., 2017; Pandey, Hasan, Dubey, & Sarangi, 2013). Many people in the United States currently use other tracking apps. Many smartphone owners use fitness tracking apps such as Fitbit and MyFitnessPal with 23.6 and 11.7 million monthly users, respectively (“Verto Index Health and Fitness Apps,” 2017). With the number of smartphone users increasing, this number is only expected to increase. Among adolescents, the work of Gibson and colleagues focused on utilizing items/symptoms from the Memorial Symptom Assessment Scale and utilizing a personal digital assistant (PDA) to deliver questions before, during and after chemotherapy 16 (Gibson et al., 2010). Other studies, including adolescents and young adults (AYAs), have reported high adherence rates with electronic symptom diaries (>90%; Gibson et al., 2013). Additionally, AYAs also reported few technical issues relating to the diary itself and reported it to be useful for themselves. Electronic Versus Paper-and-Pencil Diaries Several studies have examined the differences between electronic and paper-andpencil diaries in a variety of conditions, including adult cancers, asthma, and sleeping problems. Paper-and-pencil and electronic diaries have also been used in different patient populations, including children and even ill infants for whom parents provided proxy reports (Kelly, Young, Lane, & Shames, 2004). Paper-and-pencil and electronic symptom diaries have been utilized by children and their parents to report and monitor symptoms in different illnesses such as cystic fibrosis and ill infants (Bennett, Patrick, Lymp, Edwards, & Goss, 2010; Müller, Hemmi, Wilhelm, Barr, & Schneider, 2011). Although few studies have examined the differences between paper-and-pencil diaries and mobile diaries in children, when compared to paper-and-pencil diaries, mobile symptom diaries measuring pain in children have shown greater compliance and accuracy. Those using the mobile diaries reported an average of 6.6 days compared to 3.8 days using the paper-and-pencil (Palermo, Valenzuela, & Stork, 2004). Additionally, children preferred the mobile diary and had fewer errors inputting information than with the paper-and-pencil diary (Palermo et al., 2004). Potential advantages of electronic diaries include: a reduction in missing data, decreased burden for participants, convenience, and the ability to time-stamp diary entries (Gwaltney, Shields, & Shiffman, 2008; Müller, Hemmi, Wilhelm, Barr, & 17 Schneider, 2011; Stone, Shiffman, Schwartz, Broderick, & Hufford, 2003). However, the most important advantage reported is the increase in daily adherence rates with electronic diaries with reports ranging from 83-94% compliance versus 11-20% for paper-andpencil with participants reporting symptoms 1-3 times per day over a period of weeks to months (Gwaltney et al., 2008; Morren, Dulmen, Ouwerkerk, & Bensing, 2009; Stone et al., 2003). Although adherence was greater for electronic diaries, a high-level of agreement overall between electronic and paper-and-pencil diaries was noted (Gwaltney et al., 2008; Müller et al., 2011). Theoretical Framework: Sociotechnical Framework This study is guided by the sociotechnical framework as described by Barber, et al. (2007), who mapped the sociotechnical framework to the 1988 Donabedian Model for assessing the quality of care in the health care system (Barber, Cornford, & Klecun, 2007; Donabedian, 1988). The sociotechnical framework is a set of models and approaches that suggest both human (social) and technical aspects must be accounted when examining information management and technology use in a business (Barber et al., 2007). Additionally, both of these social and technical aspects interact with each other and do not occur in isolation. The broad nature of the sociotechnical framework connects the human and technical aspects of this study as related to the symptom experience and tracking symptoms. Although the framework also encompasses the system and organizational context, for this study, the focus will be on the individual human-technical interaction. The human aspects (social component) are the people involved in the symptom experience. For this study, the social component includes parents and their children with 18 a focus on the child’s symptoms as evaluated and monitored by the parent. Technology is “the practical application of knowledge, especially in a particular area” (Merriam Webster, 2016). The technical components examined in this study are the symptom diaries, both electronic and paper-and-pencil. The sociotechnical framework provides a basic perspective for approaching the study. The framework will be adapted with additional detail to address the symptom experience using concepts from the UCSF symptom management theory and to address the technical facets of this study using the Technology Acceptance Model (Davis, 1989; Bender, M.S., 2018, Venkatesh & Davis, 2000). Table 2.2 describes the sociotechnical framework as it will be applied in the proposed study. UCSF Symptom Management Theory The UCSF symptom management theory will augment the sociotechnical framework to address the “symptom experience” aims of this study (aims 1 & 2). The UCSF symptom management theory was first proposed as a conceptual model in 1994 and was further refined in 2001. It was set forth as a theory in 2008 to describe the complex, multi-faceted symptom management process (Bender, M.S., 2018). This study will emphasize the “symptom experience” portion of the model, which will be symptoms as reported by parents. Because this study emphasizes the description of the symptom experience and not on a symptom management intervention, only this component of the theory will be used. The ellipses on the outside of the model will also inform the symptom experience aims of this study. The symptom experience in children is influenced by a person, environment, and health and illness. For this study, the personal dimension is important 19 in influencing how symptoms are expressed and how others (i.e., parents) respond to them. Because most of the children will be too young to recall and report symptoms using the study instruments, parents will be the ones to interpret their child’s symptoms, evaluate the child’s response to symptoms and respond to those symptoms. To examine the symptom experience, symptom diaries, both electronic and paper-and-pencil, will be utilized. Technology Acceptance Model (TAM) The Technology Acceptance Model (TAM) was developed in 1985 from behavioral theories including the Theory of Reasoned Action and the Theory of Planned Behavior, to address the multiple components that influence a person’s acceptance and use of a particular technology system (Davis, 1989). Perceived usefulness and perceives ease of use can predict a person’s intent to use a given technology which then influences actual use of the technology. These are both components of whether or not someone will use a given technology which impacts usability. Usability is “…the quality of a user’s experience when interacting with products or systems…”(usability.gov, n.d.). User satisfaction, another component of usability, will also be evaluated in this study (Holden & Rada, 2011). Perceived usefulness is the focus of this study. Davis (1989) described perceived usefulness as “the degree to which a person believed that using a particular system would enhance his or her job performance” (p. 320). For this study, perceived usefulness will be defined as the degree to which the parent of a child with cancer feels that each type of symptom diary would provide a beneficial representation of their child’s symptoms, be useful in reporting to providers, and be useful in enhancing communication between the 20 parent and provider. Summary Young children with cancer experience symptoms, whether self-reported or reported by their parents. However, because many studies combine age groups, it is hard to establish if these symptoms vary or if there are unique symptoms exclusive to this population. Additionally, because of age, very few self-report measures exist for young children, although visual measures could be a possibility (Dupuis et al., 2001; Pinheiro et al., 2018). To fully understand the symptom experience of children with cancer, studies, including young children, are warranted. To provide developmentally appropriate symptom relief and management in young children with cancer, symptoms must be recognized and then reported to support the initiation of interventions. This proposed study aims to address what types of symptoms are reported as well as how symptom diaries could be utilized in the report of symptoms in young children with cancer. Evaluation of this approach to monitor symptoms and its potential to improve patient/provider communication and facilitate interventions to alleviate symptoms is lacking. The findings from this study could lead to future research into symptom interventions for young children with cancer. Table 2.1 Publications Including Children With Cancer < 7 Years of Age Sample Characteristics Type of Report Symptoms Studied Type of Study 124 children ages 1– 17 undergoing chemotherapy Child report Nausea and vomiting Quan Among younger children (3–4.9), the happy face nausea scale was used for self-report. Reported nausea severity & prevalence was the same as older age groups, but younger children had more vomiting episodes than other age groups (+1/day). Symptom Assessment in Children Receiving Cancer Therapy: The Parents' Perspective (Dupuis et al. 2010) Parents of children 4–18 with cancer Parent report Multisymptom Quan Most prevalent symptoms were: mood swings, fatigue, missing activities. Symptoms causing the most bother were: missing activities, worry, and aching. More severity and bother reported with older age of the child by parents. A Systematic Review of Symptom Assessment Scales in Children With Cancer (Dupuis et al., 2012) Studies utilizing symptom assessment scales in children 2–19 Six parents; eight children Multisymptom Quan Scales most often used to assess the presence of symptoms and symptom burden. No scales were used to manage symptoms. Most frequent symptoms were: fatigue, pain, drowsiness, and anorexia. Distressing Events for Children and Adolescents With Cancer: Child, Parent, and Nurse Perceptions (Hedstrom et al., 2003) 121 children ages 0– 19 and their parents Parent & child report Multisymptom Qual Ages 0–12 most distressing physical symptom: pain from procedures/treatment. Most distressing emotional: 0–3 yrs: confinement; 4–7 years: the feeling of alienation. For ages 0–7, physical symptoms expressed most frequently Title Delayed Nausea and Vomiting in Children Receiving Antineoplastics (Dupuis et al., 2001) Study Findings 21 Table 2.1 (Continued) Sample Characteristics Type of Report Symptoms Studies Type of Study The Influence of Oxidative Stress on Symptom Occurrence, Severity, and Distress During Childhood Leukemia Treatment. (Hockenberry et al., 2014) 36 children ages 3– 14 newly diagnosed with ALL Child or parent Multisymptom Quan Fatigue and Carnitine Levels Over Multiple Cycles of Chemotherapy in Children and Adolescents (Hooke et al., 2015) 58 children ages 3– 18 undergoing cancer treatment Parent or child report Fatigue and carnitine Quan Carnitine level changes were not significant and were not associated with reports of fatigue. Fatigue was not as bothersome in school-age children which may be indicative of increased resilience in this population. Children that received highest amounts of doxorubicin had the highest fatigue reports. Review of Symptom Experiences in Children and Adolescents With Cancer (Kestler et al., 2012) Fifty studies & 2 dissertations on cancer symptoms in children 0–21 Parent or child report Multisymptom Quan Fifteen studies included children 6 and younger. More preschool and non-White children should be assessed. Instrumentation for the prereading group should be developed. Further studies examining differences in age groups and gender should be investigated. Measuring VincristineInduced Peripheral Neuropathy in Children With Acute Lymphoblastic Leukemia (Lavoie-smith et al., 2013) Children receiving Vincristine ages 1– 18 Parent or child report Neuropathy Quan Cronbach’s a for Total Neuropathy Score-Pediatric Vincristine (TNS-PV) was 0.84 and correlated with vincristine dosage, pharmacokinetic parameters, and grading scale scores. Pain scores from the FACES scale correlated with the TNS-PV neuropathic pain items and 95% of children six and older could report. Younger children had a much harder time reporting. Title Study Findings Lack of energy, pain, irritability, nausea, nervousness, lack of appetite, and difficulty concentrating occurred in > 30% of children and the majority of the time periods (5/6). Symptoms experienced during treatment are associated with increased oxidative stress. 22 Table 2.1 (Continued) Sample Characteristics Type of Report Symptoms Studied Type of Study Nighttime Sleep Disruptions, the Hospital Care Environment, and Symptoms in Elementary School-Age Children With Cancer (Linder et al., 2012) Children ages 5–12 receiving inpatient chemotherapy Child report Sleep, environment, pain, N/V Quan Sleep was often delayed until 10 pm or later. 57% of the variance in the amount of sleep was due to sound, lights, medications, and symptoms such as nausea and pain. Excessive sound levels were seen throughout the night. Symptoms and SelfManagement Strategies Identified by Children With Cancer Using Drawand-Tell Interviews (Linder et al., 2018) Children ages 6–12 undergoing chemotherapy Child report Multisymptom Mixed Symptoms most frequently reported were: nausea, fatigue, pain, and sadness. Child and Adolescent Self-Report Symptom Measurement in Pediatric Oncology Research: A Systematic Literature Review (Pinheiro et al., 2018) ROL examining studies with children <8 Child or parent report Multisymptom Mixed Most instruments are made for children eight years and older. For children less than eight years, more visuals were used. Studies varied in symptoms, language, and time points. Parents' Perceptions of Their Children's CancerRelated Symptoms During Treatment: A Prospective, Longitudinal Study (Poder et al., 2010) 214 parents of children ages 0–18 receiving treatment Parent report Multisymptom Quan Most troubling symptoms were: emotional distress, fatigue, nutrition, and pain. Most of the symptoms and symptom burden decreased over time. Parents with more emotional distress had higher reports of symptom burden than those with lower distress. Parents of adolescents also had higher symptom burden reports than those of parents with younger children. Title Study Findings 23 Table 2.1 (Continued) Title Sample Characteristics Type of Report Symptoms Studied Type of Study Study Findings Self-Report of Symptoms in Children With Cancer Younger Than Eight Years of Age: A Systematic Review (Tomlinson et al., 2017) A systematic review of studies with children < 8 Child or parent report Multisymptom Mixed There is a gap in the literature of instruments in children eight and younger. Trouble noted from previous studies in self-report among 5- to 7-year-olds having trouble understanding questions or refusing to participate. Youngest age able to self-report was three years with nausea/vomiting scale. The Pain Experience of Children With Leukemia During the First Year After Diagnosis (Adlard et al., 2004) 95 Spanish- and English-speaking children ages 4–17 with ALL A parent or child report Pain Quan In children aged 4–7, reported pain intensity scores were similar to older age groups. Pain management seemed to be less effective in Latinos even though reports of pain were similar to White, non-Latino children. The PedsQL in Pediatric Cancer: Reliability and Validity of the Pediatric Quality of Life Inventory Generic Core Scales, Multidimensional Fatigue Scale, and Cancer Module (Varni et al., 2002) 339 children ages 2– 18 in treatment Parent or child report Multisymptom Quan Reliability and validity of the PedsQL Generic Core Total Scales, Multi-dimensional Fatigue Scale, and Cancer Module were demonstrated. PedsQL can be used in studies with children focusing on Health-Related Quality of Life. Symptom Monitoring and Dependent Care During Cancer Treatment in Children: Pilot Study (Williams et al., 2006) 11 parents of children 2–18 years receiving treatment Parent report Multisymptom Quan Nausea was experienced by all children. The most frequently reported symptoms were: fatigue, nausea, fever, mouth/throat pain, pain, and alopecia. 24 Table 2.1 (Continued) Sample Characteristics Type of Report Symptoms Studied Type of Study A Symptom Checklist for Children With Cancer: The Therapy-Related Symptom ChecklistChildren (Williams et al., 2012) 385 children ages 5– 11 receiving treatment Child report Multisymptom Quan Fourteen of 30 symptoms reported by 40% of patients. Top five symptoms: feeling sluggish, nausea, lack of appetite, irritability, and vomiting. Not separated by age groups. A Different Perspective to Approaching Cancer Symptoms in Children (Woodgate et al., 2003) 39 children ages 4.5–18 and their families Parent & child report Multisymptom Qual Symptoms in children are better understood multidimensionally rather than as single symptoms. Children experienced symptoms as feelings. Sleep Habits and Fatigue of Children Receiving Maintenance Chemotherapy for all and Their Parents (Zupanec et al., 2010) 64 children ages 4– 18 & 58 parents Parent and child report Sleep & fatigue Mixed Title Study Findings Majority of children had disturbed sleep. Common causes were nightmares, bathroom use, child movement, and fear. Note. Quan = quantitative; Qual = qualitative. 25 26 Table 2.2 Application of the Sociotechnical Framework Concept Technical System Function Social Human Perspective Structure Symptom diaries Children Parents Electronic diary Paper-and-pencil diary Interaction Concepts from Technology Acceptance Model (TAM) UCSF symptom management theory Outcome (aims) Aim 3 User Satisfaction (Usability) Diary Compliance Aims 1 and 2 Symptoms Perceived Usefulness Process CHAPTER 3 METHODS Design This study used an exploratory, descriptive, crossover design using both quantitative and qualitative data. Data were collected prospectively, over two 14 to 28 day periods, concurrent with the child’s outpatient treatment schedule. This design supported the achievement of the study aims: 1) To describe the occurrence, frequency, severity, and change over time of individual and collective physical symptoms of children less than 6 years of age with cancer as reported by their parents; 2) To compare parents’ perceived usefulness of a smartphone application symptom-tracking device to a paper and pencil symptom diary in reporting symptoms to their child’s provider; and 3) To explore the usability of a smartphone application to report symptoms experienced by children with cancer as reported by their parents. A crossover design strengthened this study by controlling for variations between participants by pairing participants with themselves to serve as their own controls. Similar designs without using a crossover approach require 4 to 10 times more participants to achieve the same power (Garcia, Benet, Arnau, & Cobo, 2004). Another advantage of a crossover design was that participants could be randomly assigned to each group. Figure 3.1 provides a visual representation of the design of this study. 28 Sample Twenty child and twenty parent participants were recruited from the outpatient hematology/oncology clinic at Primary Children’s Hospital (PCH). Inclusion criteria for child participants were: 1) primary diagnosis of childhood cancer; 2) between the ages of birth to age 6; 3) treated at PCH outpatient clinic with chemotherapy for cancer with at least three scheduled visits with the same degree of treatment intensity, and 4) the child was at least 30 days post-diagnosis. The exclusion criterion for the child was that the child was receiving end-of-life chemotherapy and not curative treatment. The inclusion criteria for parents were: 1) the parent or legal guardian of the child who meets inclusion criteria; 2) able to speak, read, and understand English. Parents with a physical and cognitive impairment which inhibited their ability to complete the questionnaires and tools were excluded. The proposed sample size met or exceeded most recommendations for usability/feasibility studies. Although recommendations from the literature vary on the number of participants needed for usability studies, most recommend sample sizes range between three to twenty participants (Faulkner, 2003; Lewis, 1994; Nielsen, 2000; Virzi, 1992). Childhood cancer is also much more infrequent than adult cancers; thus, large sample sizes are hard to acquire. A crossover design addressed the issue of sample size and matched the children and parents with themselves – a powerful design control. The desired sample size was also feasible based on the number of potentially eligible patients seen at PCH during the past 3 years (~123 patients/3 years). 29 Setting The study setting was the outpatient hematology/oncology clinic at PCH in Salt Lake City, Utah. PCH is a 289-bed pediatric hospital that serves the largest geographic region of children in the United States. PCH serves more than 300 inpatient and outpatient pediatric patients for cancer treatment each year. PCH is a Children’s Oncology Group (COG) affiliated institution serving children with cancer throughout the Intermountain West. The Hematology/Oncology and Bone Marrow Transplant Outpatient Clinic provides chemotherapy and biotherapy administration, blood product infusions, and hydration/supportive cares for children with cancer and their families. This clinic operates Monday-Fridays and consists of 12 exam rooms, two procedure rooms, and eight infusion bays. Procedures Recruitment IRB approval was granted from 3 institutions which included: the University of Utah, Primary Children’s Hospital, and Huntsman Cancer Institute. The principal investigator met with an oncology team member at the outpatient clinic every week to identify potentially eligible participants who were scheduled for chemotherapy treatment the following week. When eligible participants arrived at the clinic for their appointment, an oncology team member would then briefly explain the study purpose and ask the parent for permission to be contacted by the principal investigator. If the parent agreed, the principal investigator then met with the parent to explain the study and obtain informed consent for parent participants and parental permission for child participants. Because child participants were less than 7 years of age, assent was not obtained. 30 For parents who declined to participate (n = 1), a reason for not participating was requested. A password-protected file was maintained by the investigator to identify the names of parents who declined to participate to ensure they were not contacted again. This file was destroyed at the end of the data collection period. Participants were enrolled during an outpatient clinic visit for chemotherapy administration and enrolled for a total of three chemotherapy treatment visits to the outpatient clinic. Because of the variation in treatment protocols, the duration of study enrollment varied based on the child’s diagnosis. Participants were randomly assigned to begin using either the paper-and-pencil diary or the electronic diary on Clinic Visit #1 and were instructed to track their child’s symptoms daily until their next outpatient clinic at which point the parent was given the other diary. Ranges between clinic visits varied based on the child’s diagnosis but ranged between 7-28 days. A blocked randomization process was used so that similar samples were assigned to each group. Blocks of four were used to avoid anticipatory bias. The technique of sequentially numbered, opaque sealed envelopes ensured true randomization of participants (Doig & Simpson, 2005). A washout period was not implemented because participants were using different versions of the same diary with no treatment carryover and no intervention to impact parents’ use of the subsequent diary. Parent Responsibilities Parents who already had a smartphone or other device that was compatible with the symptom tracking application were assisted in downloading the iChemo Diary application and instructed on its use. Those without a smartphone (n = 10) were provided with an iPod Touch to use for the study period with the application already downloaded. 31 Graphical information about the iChemo diary was provided to these families. Parents were instructed to report symptoms once daily and were sent a reminder of their choice by the principal investigator each day to remind them to enter symptoms. Parents chose what type of daily reminder was most convenient for them at the start of the study. Data Collection and Investigator Responsibilities Data were obtained from parents at the next clinic visit for chemotherapy per protocol schedule by the principal investigator either from the electronic diary or the paper-and-pencil-dairy. An ID number was assigned to participants to support the confidentiality of the data. All data were transferred to an encrypted partition on the hard drive of a password-protected computer. Participants were only identified by the ID number, and documentation linking the ID number to protected health information was stored in a separate password-protected file. Parents were informed and asked to complete the TAM, QUIS, and parent interview questions at the end of each data collection period. A $50 gift card was provided for compensation after the entire data collection period. Study Measures Demographic and Clinical Data A demographic information data collection sheet created by the principal investigator was administered electronically to gather demographic data from child and parent participants at the time of enrollment. Clinical, including the child’s current medications, diagnosis, and treatment plan, were retrieved from the medical record. Child participant data collected included: age, diagnosis, date of diagnosis, outpatient clinic 32 schedule, race/ethnicity, gender, and treatment protocol. This was collected from the parent and the medical record. Parent/guardian participant data included: age, race/ethnicity, gender, marital status, education level, number of children, individuals with whom the child lives, and where the child and parent reside. Parent Interview A parent interview about the use of each of the symptom diaries was administered at the end of each period to describe the use of the symptom diaries qualitatively. Interviews were audio-recorded and transcribed for analysis. Data obtained from these interviews were analyzed qualitatively using content analysis. Questions for this semistructured interview are presented in Appendix A. Symptom Diaries Two symptom diaries were used in this study to measure symptoms. These were the iChemo Diary version 1.1 (Merck & Co., 2011) and a paper-and-pencil symptom diary. iChemo Diary The iChemo Diary application is a free smartphone/tablet application developed by Merck (Merck & Co, 2011). This touch screen application supports tracking commonly occurring physical symptoms related to treatment for cancer. Symptoms include nausea, pain, fatigue, vomiting, fever, lack of appetite, tingling in extremities, diarrhea, constipation, and rash. A free-text portion was also provided for parents to list other symptoms. The symptoms, along with their corresponding measurements, are described in Table 3.1. 33 The iChemo diary was designed to take only a few minutes to complete. Thus, the minimal burden was expected of parent participants to complete the diary each day. The expected time to complete the diary was between 5 to 10 minutes per day. There was no burden to the children in this study. If a parent did not have a smartphone/tablet or a compatible device, one was loaned to them to use during the study duration. Data from the iChemo diary were saved automatically in the iChemo program as the parent entered the information. Parents were asked to bring the personal smartphone or iPod Touch used to enter data with them to their next clinic visit. Data were downloaded by the principal investigator. Additionally, the application provided a graphic representation of symptoms that the parent could use to refer to if needed. The iChemo diary was completed during each study phase in between outpatient visits for chemotherapy. Parents were reminded via text, telephone, or built-in calendar reminder each day to complete the diary. Diary data were erased from devices between participants to ensure privacy. Paper-and-Pencil Symptom Diary The paper-and-pencil diary was developed by the investigator and included the same items from the iChemo electronic version in a similar format. Parents were asked to complete a diary form each day (Appendix B). Parents were given a 30-page booklet with a diary-page to fill out each day and were asked to return the booklet at their next clinic visit. Parents were reminded via text, telephone, or phone calendar reminder each day to fill out the diary. Again, the expected time of completion was less than 10 minutes. A summary of measurements and collection is provided in Table 3.2. 34 Measures and Reliability/Validity of Tools Questionnaire for User Interface Satisfaction The Questionnaire for User Interaction Satisfaction (QUIS) 7.0 is a questionnaire developed at the University of Maryland to measure user satisfaction with computers and electronic tools. This questionnaire was used to evaluate the overall user reaction of both diaries. The QUIS is a modular tool composed of several semantic differential scales measuring user satisfaction. Because of its hierarchical nature, certain sections may be included or omitted with no major effects on the reliability of the tool (“Questionnaire for User Interface Satisfaction,” n.d.). This questionnaire has demonstrated acceptable internal consistency (Cronbach’s alpha = .88 -.96) and high overall user satisfaction with the questionnaire (Chin, Diehl, & Norman, 1988; Harper, Slaughter, & Norman, 1997; Slaughter & Norman, K. L. Schniederman, 1995). Construct validity has been established for each of the subscales within the questionnaire. The QUIS has also been used to evaluate other computer tools in other areas of nursing research (Hortman & Thompson, 2005; Johnson, Zhang, Tang, Johnson, & Turley, 2004; Staggers, Kobus, & Brown, 2007). Part 3 of the QUIS, Overall User Reaction, was used for this study. The QUIS questionnaire included five items and was expected to take less than 5 minutes to complete. Parent participants completed the QUIS electronically, in-person after each diary period (Appendix C). Technology Acceptance Model: Perceived Usefulness Questions adapted from the perceived usefulness component of the Technology Acceptance Model Measurement (TAM) scales were used to gauge parents’ perceptions 35 of both the electronic diary and the paper-and-pencil symptom diary (Davis, 1989). This tool is a Likert-type scale consisting of five items with seven selections ordered from extremely likely to extremely unlikely. Acceptability reliability (α = .87 to .98) of the Perceived Usefulness scale within the TAM has been demonstrated in previous studies (Venkatesh & Davis, 2000). These items were administered at the end of the two collection periods. The scores were summed into one score for the perceived usefulness of each diary (Appendix D). Study data were collected and managed using REDCap electronic data capture tools hosted at The University of Utah. REDCap (Research Electronic Data Capture) is a secure, web-based application designed to support data capture for research studies, providing 1) an intuitive interface for validated data entry; 2) audit trails for tracking data manipulation and export procedures; 3) automated export procedures for seamless data downloads to common statistical packages; and 4) procedures for importing data from external sources (Harris et al., 2009). Missing data were handled using listwise deletion (small amounts) or mean imputation. Both methods are effective ways of handling missing data with sample sizes less than 40 and if the missing data does not exceed 15% (Myrtveit, Stensrud, & Olsson, 2001; Rubin, Witkiewitz, Andre, & Reilly, 2007). Analysis Plan for Each Aim Aim 1 Aim 1 was to describe the occurrence and severity of individual physical symptoms of children less than 6 years of age with cancer as reported by their parents. 36 Analysis Plan Daily symptom data were analyzed by describing the percentage of total monitored days with a report of a given symptom. Symptom severity was reported as mild, moderate, or severe. Symptom severity was further examined by identifying the number of days that parents reported symptom severity as none, mild, moderate, or severe for their child. Severity was further examined by identifying the number of days symptom severity for any symptom was reported at moderate or higher and mild or lower. Patient and group means and standard deviations were reported for continuous variables (e.g., pain). Frequencies were reported for categorical variables (e.g., diarrhea). Free-text entries were analyzed qualitatively using summative content analysis to identify common categories from the data as well as the frequencies of these categories (Hsieh & Shannon, 2005). A parent interview was administered at the end of each period to qualitatively explore how the symptom diaries were used. Data from these surveys were analyzed using content analysis. Aim 2 Aim 2 was to compare parents’ perceived usefulness of a smartphone application symptom diary to a paper and pencil symptom diary in reporting symptoms to their child’s provider. Analysis Plan Analysis of perceived usefulness will be measured through the use of the Technology Acceptance Model – Perceived Usefulness Questions at the end of each period with the symptom diaries. Wilcoxon-Signed Rank Tests were used to compare 37 perceived usefulness between each diary at the end of each period. Reliability of the adapted TAM questions was also evaluated using a test for internal consistency (Cronbach’s α). Aim 3 Aim 3 was to explore the usability of a smartphone application to report symptoms experienced by children with cancer as reported by their parents. Analysis Plan User satisfaction, a component of usability, was evaluated using the QUIS 7.0 instrument. This instrument was used to measure user satisfaction with each of the diaries to determine if the diary is acceptable for use by parents. Wilcoxon-Signed Ranks tests were used to compare the user satisfaction of each diary for use by parents at the end of each period. Additionally, data on the percentage of completed symptom diaries (electronic versus paper-and-pencil) were analyzed by comparing compliance rates for each of the symptom diaries at the end of each period. Wilcoxon-signed ranks tests analyses were also used to see if the frequency of responses differed between diary types. 38 Table 3.1 Description of Symptoms in the Symptom Diary and Corresponding Measurements Symptom Measurement Nausea Likert-type scale: none, mild, moderate, severe Pain Likert-type scale: none, mild, moderate, severe Fatigue Likert-type scale: none, mild, moderate, severe Vomiting Number of episodes Fever Degree in Celsius or Fahrenheit Lack of appetite Dichotomous Tingling in feet/toes Dichotomous Diarrhea Dichotomous Constipation Dichotomous Rash Dichotomous Other symptoms Free-text/content analysis 39 Table 3.2 Summary of Data Collection and Measures Data Collection Period 1 Clinic Visit #2 Consent and assignment to group Demographic information sheet iChemo Diary TAM Consent and assignment to group Demographic information sheet Paper-and-Pencil Diary Group Clinic Visit #1 1 2 Data Collection Period 2 Paper-and-Pencil Diary Clinic Visit #3 TAM QUIS QUIS Parent interview Parent interview TAM iChemo Diary TAM QUIS QUIS Parent interview Parent interview Note. TAM = Technology Acceptance Model questions (perceived usefulness); QUIS = Questionnaire for User Interface Satisfaction. 40 Clinic Visit #1 Informed Consent Randomization Group 1 iChemo Diary Clinic Visit #2 Switch to Paperand-Pencil Diary Group 2 Paper-and-Pencil Diary Clinic Visit #2 Switch to iChemo Diary Final Clinic Visit #3 Figure 3.1 Research design. CHAPTER 4 PARENTS’ REPORT OF PHYSICAL SYMPTOMS IN YOUNG CHILDREN WITH CANCER Abstract The purpose of this study was to describe the physical symptoms in young children with cancer as reported by their parents using two diaries and to explore how parents discerned symptoms and severity. Data were collected over two 7-day periods spanning three scheduled clinic visits. At visit #1, parents were randomly assigned to record their child’s symptoms using one of two symptom diaries. At visit #2, parents switched to the other diary. Parents also participated in interviews addressing how they determined the presence and severity of symptoms. Twenty parents of children 6 and younger with cancer were recruited from an outpatient oncology clinic. Symptom reports from the diaries were analyzed descriptively. Interview responses were analyzed using content analysis. Reported symptom occurrence did not differ based on diary type. Fatigue was the most commonly reported symptom (56.8%), followed by nausea (51.4%) and lack of appetite (37.8%). Symptoms most frequently reported as moderate or greater severity were fatigue (17.5%), nausea (6.4%), and pain (5%). Parents used their child’s activity level, verbal cues, and spousal discussion most often when identifying whether their child was experiencing a symptom. When determining severity, parents used the 42 degree of difference from their child’s “normal” self along with the child’s activity and verbal cues. Young children receiving chemotherapy in the ambulatory setting experience frequent, persistent symptoms, sometimes with a high degree of severity. Parents of young children utilize different ways to identify symptoms and severity. Understanding how parents identify these symptoms and what symptoms are commonly experienced can help guide future symptom assessment tools and symptom management strategies. Parents’ Report of Physical Symptoms in Young Children With Cancer Each year, more than 10,000 children under the age of 14 are diagnosed with cancer with the highest rates occurring among children 2-3 years of age (National Cancer Institute, 2014). To date, adolescents and school-age children have been the focus of the majority of the research addressing symptoms in childhood cancer (Pinheiro et al., 2018; Ruland, Hamilton, & Schjodt-Osmo, 2009; Tomlinson et al., 2017; Vatne, Slaugther, & Ruland, 2010; Walker, Gedaly-Duff, Miaskowski, & Nail, 2010). Younger children, those 6 years and younger, are often excluded from research due to challenges related to their age-related developmental abilities, often their limited ability to respond to selfreport scales (Linder, 2008). As a result, the symptom experiences of young children with cancer remain largely unexplored. Symptoms in Children With Cancer Identifying symptoms and their associated severity in children with cancer can be challenging, especially for young children. Young children can have difficulty understanding the meaning of symptoms and explaining symptoms to parents or healthcare providers. They may also refuse to participate in studies (Scolnik, Atkinson, 43 Hadi, Caulfeild, & Young, 2003; Tomlinson et al., 2017; Tomlinson et al., 2016; Vatne et al., 2010). Even older children may describe symptoms as feelings or “feeling states” rather than discrete, single symptoms (Woodgate, 2008). In studies which included younger children, the younger children were often combined with older age groups, and the studies were not powered to explore differences among different age groups. Because of these factors, evaluating differences based on age and developmental level is difficult (Kestler & Lobiondo-Wood, 2012; Linder, 2008; Ruland, Starren, & Vatne, 2008; Williams et al., 2012). Across all age groups of children and adolescents, the most commonly reported physical symptoms are fatigue, nausea, anorexia, drowsiness, and pain (Abu-Saad Huijer, Sagherian, & Tamim, 2013; Adlard et al., 2004; Dupuis, Ethier, Tomlinson, Hesser, & Sung, 2012; Hedström, Haglund, Skolin, & Von Essen, 2003; Kestler & Lobiondo-Wood, 2012; Dupuis, Lau, & Greenberg, 2001; Linder, Bratton, Nguyen, Parker, & Wawrzynski, 2018; Miller, Jacob, & Hockenberry, 2011; Pöder, Ljungman, & Von Essen, 2010; Rodgers, Krance, Stremayet, & Hockenberry, 2014; Williams, Schmideskamp, Ridder, & Williams, 2006). Of these studies, eight included children less than 6 years of age and half used parent or child report, two used parent report alone, and two utilized only child report (Adlard et al., 2004; Dupuis et al., 2012; Hedström et al., 2003; Kestler & Lobiondo-Wood, 2012; Dupuis et al., 2001; Linder et al., 2018; Pöder et al., 2010; Williams et al., 2006). In the few quantitative studies in which children less than 6 years were described separately from older children, only a few individual symptoms were examined. Nausea was self-reported by children as young as 3 and was reported at the same frequency as older children while vomiting was reported more frequently in 44 younger children than older children (Dupuis et al., 2001). The most distressing physical symptom reported was pain, along with the emotional symptom of confinement (Hedström et al., 2003). Qualitative work with children older than three years of age and their parents have also yielded information about differences in the symptom experience among different developmental groups particularly in how younger children express or verbalize specific symptoms (Hedström et al., 2003; Woodgate, 2008). Younger children often described symptoms collectively based on how it interfered with their day rather than delineating each symptom individually (Erickson et al., 2013; Hedström et al., 2003; Linder et al., 2018; Woodgate, 2008). Most of the literature that included younger children compared symptoms in children 4-7 years of age with those of children in other age groups. Disruptions in sleep and reports of pain were similar between 4-7-year-olds and older age groups (Adlard et al., 2004; Hedström et al., 2003; Zupanec et al., 2010). Studies examined individual symptoms like sleep and found that children within all age groups had disrupted sleep (Zupanec et al., 2010). In qualitative work, the feeling of alienation was experienced by 4-7-year-olds versus confinement in younger children (Hedström et al., 2003). For 0-7year-olds, physical symptoms tended to be expressed more than emotional symptoms than those of older ages (Hedström et al., 2003). The most bothersome symptoms varied among children. When parents provided proxy-reports of the child’s symptoms, missing activities, worry over the disease and aching bones were reported as most bothersome. Mood swings, fatigue, and missing activities were also among the most prevalent and severe symptoms (Dupuis et al., 2010). Parents also tended to report greater severity and bother of symptoms in older children 45 than the children’s’ reports, although the age groups were not delineated (Dupuis et al., 2010). Children of all ages often reported nausea and pain as the most severe and distressing symptoms (Hedström et al., 2003; Miller et al., 2011; Rodgers et al., 2014). Parents as Proxy When identifying symptoms, the self-report of the individual experiencing the symptom is regarded as the gold standard (Sherifali & Pinelli, 2007; Yeh et al., 2005; Zhukovsky et al., 2015). Using parent-proxy reporting for young children, however, is frequently used as an alternative approach to identifying and treating symptoms (Christina Baggott et al., 2014a; Gonzalez-Mercado, Williams, Williams, Pedro, & Colon, 2017; Hedström et al., 2003; Pöder et al., 2010; Quigg, Mahajerin, Sullivan, Pradhan, & Bauer, 2013; Williams et al., 2012; R. L. Woodgate et al., 2003). In studies with children 6 years or younger, parents were often used as a proxy to report symptoms to the provider. Young children are limited in their ability to report their symptoms to others because of their developmental abilities, including verbal and cognitive abilities. Young children are also present-oriented, limiting their ability to respond to traditional self-report instruments with varied recall periods such as the past week or past month (Linder, 2005; Sherifali & Pinelli, 2007). Additionally, other studies suggest that many parents of young children with cancer feel that their child may be too ill to report or that they tire easily (Ruland et al., 2009; Woodgate, Degner, & Yanofsky, 2003; Woodgate & McClement, 1998). Although self-report would be ideal for all patients with cancer, in the case of younger children, the parent-proxy report is a feasible and appropriate solution for symptom identification. Little knowledge is known about how parents decide a child is experiencing a 46 given symptom and how they determine the associated severity of the symptom. In the very limited literature, the beliefs of the family and child played a role in whether or not parents reported symptoms. Parents often did not report symptoms unless they felt their child was suffering (Woodgate et al., 2003). In one study, parents determined symptoms and identified severity by using open communication with their family, taking into account the personality of the child and determining if the symptoms were expected or not (Battista et al., 2017). Parent-proxy reporting, however, is not without limitations. When compared to child reports among children 8-18 years of age undergoing cancer treatment, parents tend to over-report rather than under-report symptoms (Christina Baggott et al., 2014a; Hyslop et al., 2018). Parents of older children also reported more symptoms with more severity and bothered than those with younger children (Dupuis et al., 2012; Pöder et al., 2010). Reports of a greater number of symptoms were also associated with the emotional distress of the parent providing the report (Pöder et al., 2010). Despite these issues, however, concordance among parents’ and children’s reports have been observed for many symptoms, primarily physical symptoms with a more directly observable component, among 10-18 year-olds (Christina Baggott et al., 2014a; Hyslop et al., 2018; Lo & Hayman, 1999; Speyer et al., 2009). Concordance between parent and child reports of psychosocial and other more subjectively experienced symptoms, however, is lower (Christina Baggott et al., 2014a; Hyslop et al., 2018; Lo & Hayman, 1999). Despite these potential limitations, parent proxy reporting may provide a reliable approach to physical symptom identification in young children with cancer. Parents serve as advocates for their children and are often the ones that speak on their behalf at medical appointments to 47 providers. Symptom Diaries Because symptoms can vary from day-to-day, supporting patients and families with a resource to track and report symptoms is could be beneficial. With the advent of smartphones, having an electronic symptom tracking resource could be appealing and convenient for patients and families. Symptom diaries have been utilized by children with other chronic illnesses such as cystic fibrosis and their parents (Bennett et al., 2010). When compared to paper-and-pencil diaries, mobile symptom diaries have shown greater compliance and accuracy (Palermo et al., 2004; D. Tomlinson et al., 2013). Mobile symptom diaries have also been used successfully in adult cancer patients and adolescents with overall high adherence, and perceived benefit among participants (Christina Baggott et al., 2012; Blondin et al., 2010; Gaertner, Elsner, Pollmann-Dahmen, Radbruch, & Sabatowski, 2004; Rodgers et al., 2014). A mobile phone-based symptom diary created for adolescents and young adults showed feasibility and had daily adherence of greater than 90% (Christina Baggott et al., 2012). Study Purpose and Framework The purpose of this study was to describe the daily occurrence and severity of commonly occurring physical symptoms and their characteristics in young children with cancer. Parents provided proxy reports of their children’s symptoms using two different symptom diaries. The study also explored how parents identified their child’s symptoms and discerned symptom severity. This study was guided by the UCSF symptom management theory (Bender, M.S, 2018). The UCSF symptom management theory was 48 first proposed as a conceptual model in 1994 and was further refined in 2001. It was set forth as a theory in 2008 to describe the complex, multi-faceted symptom management process (Dodd et al., 2001; Bender, M.S., 2018). For this study, the focus was the “Symptom experience” portion of the model, which was the symptom prevalence and severity as reported by parents. Methods Study Design This study used a mixed-methods exploratory, crossover design. Data were collected prospectively over two time periods using two symptom-tracking diaries. These time periods corresponded to intervals between scheduled outpatient clinic visits for chemotherapy based on the child’s treatment protocol. A crossover design was used to match the children and parents with themselves and provided adequate power to distinguish differences. Symptoms were not anticipated to vary since children were receiving treatment of a similar intensity during each data collection period. A washout period was not used because no carryover effect based on diary type was anticipated. Study Sample and Setting Participants were recruited from the hematology/oncology clinic of the Cancer Transplant Center within a tertiary, free-standing children’s hospital in the Intermountain West of the United States. The clinic serves children with all types of cancer, hematological conditions, and those who have received hematopoietic stem cell transplants. This clinic provides treatment to approximately 40 to 50 children 6 years and younger, who are newly diagnosed with cancer each year. 49 Children and their parents were included as study participants. Inclusion criteria for child participants were a diagnosis of childhood cancer, between the birth to 6 years of age, and at least 30 days following an initial diagnosis of cancer or identification of relapsed disease. Child participants were receiving chemotherapy at the outpatient clinic with at least three scheduled visits with a similar degree of treatment intensity. Children who were receiving palliative therapy or end-of-life care were excluded. Parent participants were required to be the parent or legal guardian of the child who met inclusion criteria and is able to speak, read, and understand English. Parents who had physical or cognitive impairments that inhibited their ability to complete the study procedures were excluded. Study Measures Children’s symptoms were reported by their parents using two symptom diaries: the iChemo Diary version 1.1 app (Merck & Co., 2011) and a paper-and-pencil symptom diary. Parents also participated in a brief interview following each of the two data collection periods. iChemo Diary The iChemo Diary app is a free smartphone/tablet app developed by Merck (Merck & Co, 2011). This touch screen app supports tracking of commonly occurring physical symptoms related to treatment for cancer. Symptoms include nausea, pain, fatigue, vomiting, fever, lack of appetite, tingling in extremities, diarrhea, constipation, and rash. A free-text portion is also provided for identifying other symptoms. Additionally, the app provides a daily and weekly graphic representation of symptoms to 50 allow visualization of trends. Paper-and-Pencil Symptom Diary The paper-and-pencil diary was developed by the investigator. It includes the same items in a similar format, including rating scales, as the iChemo diary. Qualitative Interviews Qualitative interviews of parent participants were conducted after the use of each diary to determine how parents identified and determined severity for symptoms in their children. Questions for the interviews are listed in Table 4.1. Procedures Institutional review board approval was granted for this study. Potentially eligible parents and children were screened weekly by the principal investigator and an oncology clinic team member. The oncology team member approached that meeting eligibility to introduce the study and to obtain permission for the principal investigator to contact them with more information about the study. The principal investigator then met with the parents to explain the study and obtain both informed consent for the parent and parental permission for child participants. Because all children were less than 7 years of age, written assent was not obtained. Parent participants were randomly assigned to begin using either the paper-andpencil symptom diary or the electronic diary at their first visit. They were instructed to complete the diary each day between clinic visits and to bring it back to their next clinic appointment. At the second clinic visit, parents returned the initial diary. They then received the second diary to use and returned that diary at the third clinic visit. At the 51 second and third clinic visits, short parent interviews were also conducted using a set of investigator-developed, open-ended questions. Data from the iChemo diary were saved and stored locally within the iChemo app as the parent entered the information. An iPod Touch on which the app had been downloaded was provided to parents to use during the study period. The iPod Touch was provided to parents because the app was discontinued from the iOS App Store during the recruitment and data collection phase of the study. Twelve participants had been enrolled when the app was discontinued. Parents returned the iPod Touch at the subsequent scheduled clinic visit. Each diary was used daily for approximately two weeks during each study phase in between clinic visits for chemotherapy. Parents received daily reminders via text, telephone, or built-in reminder each day to complete the diary. Data Management and Analysis ID numbers were assigned to participant diary responses. App-generated data were downloaded directly from the app electronically and saved onto a passwordprotected, encrypted computer. Data were then downloaded into Microsoft Excel and imported into SPSS version 20 for Mac for data analysis. Data collected from previous parent users were deleted from the iChemo app before assigning the iPod touch to another parent. Paper-and-pencil diaries were retrieved from parents and stored in a locked cabinet in a private, locked office. Data were then entered into Microsoft Excel and imported into SPSS for data analysis. Because of the variability in the number of days in between clinic visits and because all participants had a minimum of 7 days, the first 7 days of each diary use were 52 included in the analyses. The total number of days with both diaries included was 280 total days (140 days for each diary type). Descriptive statistics were used to summarize the frequency of diary responses, the prevalence of symptoms by child, and demographic data. Free-text responses from the diaries were entered into a Microsoft Excel spreadsheet. Data from the interviews were downloaded from the digital audio recorder onto a password-protected, encrypted computer, and then transcribed verbatim. Responses to interview questions were entered into Microsoft Excel worksheets and analyzed using content analysis. Because of the inductive, exploratory nature of the symptom experience in children, a conventional approach towards content analysis was used (Hsieh & Shannon, 2005). This approach allows for the exploration of a particular phenomenon to serve as a stepping stone for future research. Data were reviewed and coded by two investigators working independently. The investigators then reviewed the codes together and decided upon categories and subcategories for the data. Discrepancies were resolved by meetings between investigators. Results Participants Participants included 20 children and 20 parents. Child and parent participant characteristics are presented in Table 4.2. The majority of children were being treated for acute lymphoblastic leukemia (n = 13), and their mean age was 3.05 years ( SD = 1.4; range 0-6 years). The majority of parent participants were female ( n = 18), and the mean age was 31 years (SD = 4.6; range 18-41 years). No families declined to be contacted about the study. After learning more about 53 the study, one parent declined to participate. The reason given was that she “did not have enough time to add another thing” to her life. No participants withdrew once enrolled in the study. Completion rates for the electronic diary and the paper-and-pencil diary were similar with temperature being the most frequently missing item with 12.1% missing from both diaries. One day of nausea (0.7%) reporting was also missing from a paperand-pencil-diary. Symptoms Tables 4.3 and 4.4 report the occurrence and prevalence of each symptom across the data collection periods along with the frequencies of the severity ratings for nausea, pain, and fatigue. Of the nine symptoms included in the two diaries, fatigue was the most commonly reported symptom (56.8% of reported days), followed by nausea (51.4%) and lack of appetite (37.8%). Of the three symptoms that also included a rating for severity, fatigue was most frequently reported as moderate or severe (17.5% of reported days), followed by nausea (6.4%), and pain (5%). Symptoms were analyzed descriptively by child to determine how many children experienced at least 1 day of a given symptom along with the how many children experienced at least one moderate or severe day. Across both the electronic and paperand-pencil diary, 95% (n = 19) of parents reported their child experiencing at least one daily symptom. Additionally, across both diaries, 50% (n = 10) of the 20 parents reported their child experiencing at least one day of a moderate or severe symptom. Free-text comments about other symptoms experienced were also examined. One parent added in information about going to the emergency room for a fever. Others reported symptoms not listed in the diaries such as “mouth sores” (n = 1) or “runny 54 nose” (n = 1) or “stomach hurts” (n = 1). Three parents included information about their child’s behavior like, “more irritable than usual” or “really tired and grumpy.” Parent Interviews From the interviews with parent participants, common categories and subcategories emerged from each of the questions related to parents’ assessment of symptoms. These are described in Tables 4.5 and 4.6. Identifying Symptoms Parents often reported using more than one way of determining if their child was experiencing a symptom. Parents most frequently stated that they used their child’s activity level (n = 53) and verbal cues (n = 23) as primary ways to determine if a symptom was present.. “I look at how she is acting. Is she crying a lot, always wanting me to hold her? Is she sleepy, you know, things that would be different for her.” For those children who could verbalize their feelings, parents would rely on their child’s cues, or they would ask the child about certain symptoms if the child’s activity suggested a symptom might be present. “He usually just tells me. He will say his tummy hurts.” Some parents also discussed other ways of determining symptom presence by discussing with their spouse or expecting that the child would have a particular symptom (e.g., after surgery). Wilcoxon signed-ranks test compared the frequency symptom identification responses by diary type. No significant difference was seen in the symptom identification responses between the electronic and paper-and-pencil diaries among all categories. The total mean of all responses for the electronic was 2.05 compared with 1.9 for the paper- 55 and-pencil (z = -.52, p = .6). All results for each category are presented in Table 4.6. Symptom Severity When deciding on the severity of a symptom a child was experiencing, many parents considered specific characteristics of their child’s behavior. Parents reported looking at the degree of difference from the usual their child’s normal behavior. “If he was more one way or another we put that as severe” or by comparing “her day to day so if one day she was really tired or overly hurting, I knew it was more.” One parent stated that she would look at “how much of the day she complained or I noticed she was sick. If it was most of the day, I figured it was moderate or severe.” Parents again relied on the child’s activity and verbal cues as well to inform their perceptions of a symptom’s severity. They most frequently reported monitoring for “how upset she was or tired” or by watching “the patterns and behaviors and see how it escalated.” Parents also looked at how well the child was playing as well as how easily their child could be consoled. One parent responded that she determined severity by how well the child was “able to calm down. If he was really upset or really crying then I knew it was more severe.” Symptom Relevance Most parents reported that the diaries addressed important symptoms related to their child’s experience (Table 4.7). Most parents related the diaries addressed “relevant symptoms” commonly experienced symptoms and symptoms commonly enquired about at outpatient appointments. A few parents mentioned that they wished some questions about “mood” or “sadness” could have been included in the diaries. 56 Discussion This study addresses an important gap in the science of symptom management for children with cancer, that of describing daily symptom experiences of young children with cancer who are receiving ambulatory care. Although children receiving ambulatory care for cancer are often perceived as having fewer symptoms than children in the inpatient setting, they still experience frequent daily symptoms. Children in this study experienced at least one symptom 46% of the monitored days. In terms of symptom prevalence, 19 out of 20 children experienced at least one daily symptom. This finding is clinically important because the experience of persistent, low severity symptoms is not a “normal” part of childhood. Of these reports, moderate or severe ratings were reported with on 10% of monitored days, and vomiting episodes were reported 11% of monitored days. Half of the parents reported their child experiencing at least 1 day with a symptom of moderate or greater severity. Again, this is important information for clinicians and parents alike because these children are at home in between ambulatory care visits, and parents are the ones who are identifying or managing the child’s symptoms. Symptoms exist outside of the hospital, and little has been done to understand how parents discern and ultimately manage their child’s symptoms. Additionally, symptom diaries may be useful for parents to have to record daily symptoms their children are experiencing. The diary could be paper-and-pencil or electronic since adherence and identification of symptoms did not vary between the two diaries. Remote monitoring in real-time by providers is a distinct advantage; however, of electronic diaries. Even though children treated in the ambulatory care setting do tend to report 57 fewer symptoms and less bothersome symptoms, many young children in the outpatient setting also experience symptoms that are moderately or highly rated in severity (Collins et al., 2000; Collins et al., 2002; Johnston et al., 2018). As with previous studies conducted in school-age children and adolescents, young children with cancer do experience physical symptoms persistently and sometimes with a high degree of severity (Hedström et al., 2003; Linder et al., 2018; Miller et al., 2011; Williams et al., 2006, 2012). More than half of diary reports indicated children were experiencing nausea or fatigue and 30% of reports indicated the presence of at least mild pain. These reports are key for providers and nurses because even though these children do not require inpatient hospital admissions, they still experience symptoms deserving of attention and intervention. These symptoms are important for ambulatory care clinicians to address when young children and their parents come in for scheduled chemotherapy. Understanding the prevalence and persistence of symptoms can ensure that palliation of these symptoms occurs. This study also provides insights into the cues, behaviors, or clues parents look for when discerning specific symptoms and their associated severity. Parents often gauged symptoms by the child’s behavior or change from “normal.” Parents at home must be able to identify the symptom, determine the severity, and then decide if and how it needs to be treated. This study helps to identify several of these components that parents use when identifying their child’s symptoms. Further exploring these identifiers could greatly improve the symptom experience in young children with cancer. 58 Clinical Implications Nurses caring for young children with cancer should anticipate persistent symptoms even in the outpatient setting. When the child is at home, utilizing a way to track and record symptoms could be a beneficial tool for providers to pick up on trends, prevalence and distressing symptoms that may be occurring. In the future, having a realtime way to report symptoms and receive advice, treatment intervention and more from a provider could be a useful way to decrease symptom prevalence and severity while in between outpatient visits. The increased use and prevalence of nursing care coordinators could also be utilized to support symptom assessment and communication with other members of the team. Having a designated nurse to follow-up with parents frequently to monitor symptoms at home could be useful information to pass along to the child’s healthcare team. Utilizing this role to help facilitate timely recognition, management, and follow-up could be crucial in minimizing symptoms in this population. Having an electronic ability for alerts and remote monitoring for providers is also important. Information gleaned from parents about symptoms their child experienced in between visits or is currently experiencing could be useful for clinicians. The symptoms experienced, as well as how parents identified symptoms, could help clinicians support other parents of children with cancer. Knowing common cues and behaviors that many parents report as being key to symptom identification could help newly diagnosed families better identify and thus, better manage symptoms. A study with adults using electronic diaries in an outpatient setting found that nearly a quarter of patients thought that their care did not improve based on real-time feedback to their provider (Marceau, 59 Link, Smith, Carolan, & Jamison, 2010). Only 15% felt that their treatment plan was changed. This result demonstrates the need to incorporate provider prompts, follow-up, and treatment thresholds. Although some children as young as three can self-report, this varies among children. Thus, despite the limitations, proxy-reporting may serve as the most reliable source of information on more objectively observed symptoms. Parents serve as advocates for their children and are often the ones that speak on their behalf at medical appointments to providers. Young children with cancer that have verbal abilities should be asked about their symptoms, so that self-report from the child is included in a symptom assessment. However, few symptom assessment tools have been created for children less than 6. Future research into symptom assessment tools and questions should be explored. Additionally, care providers should direct symptom education for parents in recognizing and responding to their child’s symptoms. Limitations This study included a small sample size from one institution which limits generalizability beyond this study setting. The study sample was also homogenous with regards to race/ethnicity and included only two diagnostic groups. This may limit the ability to make inferences about other diagnostic groups. Because the daily diary information was collected at the next clinic visit to minimize the burden on families, no record of when either diary was filled out was collected. The daily reminder provided to parents may have also have contributed to higher completion and adherence rates for both diaries. Also, since there was no time stamp for the electronic diary, the specific time the diary was completed may not be accurate. Since children were generally too young to 60 self-report, congruence between the child’s individual experience and the parent’s report cannot be verified. Therefore, the parents’ depiction of symptoms experience was utilized alone and may not be a full representation of the symptoms that their child experienced. The scope of the child’s symptom experience was limited to those included in the symptom diaries. Both diaries only contained physical symptoms for parents to report. Some parents did include other symptoms in the free response and noted that psychosocial symptoms, including the child’s mood, were missing from the diaries. Directions for Future Research Future research could include the initiation of a plan of care by providers to address symptoms and interventions at home. Additionally, following up with parents about their confidence in initiating home-based symptom interventions should also be examined. As many parents reported in the interviews, it was difficult to remember symptoms from 1or 2 weeks ago. A study utilizing the electronic diary as an intervention to track, monitor, and report, and symptoms could be beneficial for the patients as well as the parents and providers. Additionally, implementing processes to formalize symptom reporting in the clinical setting is important to help identify and manage symptoms in children with cancer. Further research should be conducted in children with cancer of younger ages. Knowledge regarding symptom experience of young children is still relatively limited with parent proxy used most commonly as a means of symptom reporting. Due to the persistence of low-severity symptoms, supporting parents in home-based interventions should be the focus of outpatient education. Additionally, it may be helpful to ask patents what they are doing at home to manage symptoms and how their day-to-day lives are 61 affected by their child’s symptoms. Conclusion Young children are receiving cancer treatment in the ambulatory care setting experience persistent symptoms, particularly fatigue and nausea, sometimes of moderate or greater severity. Because parents frequently serve as the main source for reporting the child’s symptoms, efforts to understand how they distinguish the presence and severity of a symptom and to support them in tracking their children’s symptoms are warranted. Parents often use their child’s behavior and verbal cues to identify if their child is experiencing a symptom. Having a tracking tool, whether electronic or paper-and-pencil, for parents can support symptom tracking, including identification of the presence and severity of symptoms as well as trends over time. This information could be used to guide interventions for their child. 62 Table 4.1 Parent Interview Questions Concepts Questions Identifying Symptoms and Severity o o How did you know your child was having a certain symptom? How did you decide how severe or mild the symptom was? Symptom Relevance o Do you think that the diaries addressed important questions related to your child’s symptom experience? § Why or why not? Are there questions that the diary should have asked about your child’s symptom experience? Yes No § If yes, what are they? o 63 Table 4.2 Participant Characteristics Characteristic Parent Gender Male Female n % 2 18 10% 90% Parent Age 18-25 26-35 36-45 3 14 3 15% 70% 15% Parent Marital Status Married 20 100% Parent Education Level High school/GED Some College College Graduate Post-Graduate 1 8 10 1 5% 40% 50% 5% Number of children 1 2 3 4+ 1 13 3 3 5% 65% 15% 15% Parent Race/Ethnicity White/non-Hispanic 20 100% Child Gender Male Female 7 13 35% 65% Child Diagnosis Acute lymphoblastic leukemia Wilms Tumor 13 7 65% 35% Child Age <2 years 2-3 years 4-6 years 2 12 6 10% 60% 30% Child Age Totals 0-6 years 3.05 (1.4) 3 64 Table 4.3 Symptom Frequency and Prevalence by Diary Symptom Prevalence by Child Symptom Frequency Symptom Electronic Diary n (%) Paper-andPencil Diary n (%) Overall n (%) Both Diaries n (%) Fatigue 76 (54.3) 83 (59.3) 159 (56.8) 19 (95) Nausea 74 (54.2) 69 (49.3) 144 (51.4) 19 (95) Lack of appetite 54 (38.6) 52 (37.1) 106 (37.8) 16 (80) Pain 45 (32.1) 39 (27.8) 84 (30) 15 (75) Constipation 30 (21.4) 32 (22.8) 62 (22.1) 11 (55) Vomiting 20 (14.3) 11 (7.8) 31 (11) 13 (63) Diarrhea 11 (7.8) 11 (7.8) 22 (7.8) 6 (30) Tingling in hands or feet 3 (2.1) 0 (0) 3 (1.1) 2 (10) 65 Table 4.4 Frequency and Prevalence of Symptom Severity Severity Prevalence by Child, Both Diaries n (%) Symptom Frequency n (%) Symptom None Mild Moderate Severe Moderate or Severe, All Symptoms 136 (48.5) 125 (44.6) 14 (5) 4 (1.4) 10 (50) Pain 196 (70) 70 (25) 10 (3.6) 4 (1.4) Fatigue 121 (43) 110 (39.3) 39 (13.9) 10 (3.6) Nausea 66 Table 4.5 Identifying Presence and Severity of Symptoms by Categories and Subcategories Categories and Subcategories Electronic Diary Frequency Paper-and-Pencil Diary Frequency Identifying Symptoms Activity Behavioral cues Different from normal Visual cues Playfulness 12 8 7 2 10 6 6 2 Verbal Communication Child verbalization Parent questioning 9 2 6 6 1 0 1 1 1 0 Other Discussion with spouse Expected symptoms for treatment/disease/procedure Using diary as a guide Determining Symptom Severity Symptom Characteristics Degree of difference from usual symptom presentation Symptom frequency Symptom persistence Consolability 9 10 0 2 1 1 2 0 Child Activity Visual Cues Behavioral cues Playfulness 4 6 2 10 4 3 Verbal Communication Child verbalization 3 4 Other Discussion with spouse Intuition 1 1 1 0 67 Table 4.6 Wilcoxon-Signed Ranks Comparing Symptom Identification Responses by Diary Type Category Diary Type M SD Electronic 1.45 .82 1 Paper 1.2 .7 1 Electronic .55 .69 0 Activity Verbal Median Paper .60 .94 0 Electronic .05 .22 0 Other Paper .10 .31 0 Electronic 2.05 .89 2 .91 2 Total Paper 1.9 z p –1.31 .19 –.31 .75 –.58 .56 –.52 .6 68 Table 4.7 Symptom Relevance Electronic Diary Frequency Paper-and-Pencil Diary Frequency Relevant Symptoms Addressed Yes No 18 2 17 3 Other Relevant Symptoms Mood Diaper rash 1 0 1 1 Question CHAPTER 5 PERCEIVED USEFULNESS AND USER SATISFACTION OF ELECTRONIC AND PAPER-AND-PENCIL DIARIES IN YOUNG CHILDREN WITH CANCER Abstract Each year over 10,000 children are diagnosed with cancer with the highest rates in children <6. Many of these younger children receive outpatient treatment, which means parents are identifying and managing symptoms at home. The purpose of this study was to explore aspects of usability: the user satisfaction and perceived usefulness of two daily approaches to tracking children’s symptoms. Twenty parents of children 0-6 years of age tracked their child’s symptoms using an electronic diary and a paper-and-pencil symptom diary. Parents completed the diaries over three outpatient clinic visits and were randomly assigned to use one of the diaries at visit #1 and then assigned the other diary at visit #2. After these visits, QUIS and TAM-adapted questionnaires were administered along with a brief parent interview. Wilcoxon Signed-Ranks Test compared parents’ satisfaction with and perceived usefulness of each diary type. Perceived usefulness and user satisfaction of the electronic diary was greater compared to the paper diary. Qualitative content analysis explored parents’ responses to interview questions, and although different reasons for diary use were reported, there was no difference based on 70 diary type. Nineteen parents reported the electronic diary to be more useful than the paper-and-pencil diary citing familiarity, features, and convenience, and all parents reported any type of diary useful for tracking their child’s symptoms at home. Having a way to track symptoms between outpatient visits could facilitate better symptom management and improve communication between parents and providers. Background Literature Review Each year in the United States, more than 10,000 children under the age of 14 are diagnosed with cancer. More than 55% of these childhood cancer diagnoses occur in children less than 6 years of age (Howlander et al., 2015). Cancer patients of all ages, including these young children, experience symptoms as a result of their disease and treatment that can adversely impact their quality of life (Kestler & Lobiondo-Wood, 2012; Pöder et al., 2010; Tomlinson et al., 2017). Of particular significance in these young children is that persistent, troublesome symptoms may interfere with the achievement of developmental milestones. These young children, however, can have difficulty expressing themselves verbally, recalling information, specifying symptom locations, and relating the severity of a given symptom. As a consequence, these younger children are often excluded from studies that include self-report of symptoms (L. A. Linder, 2008; Riggins et al., 2009). Because young children have been largely excluded from symptom studies, the symptom experience in these children is not well understood (Ruland et al., 2009; Walker et al., 2010). Most children with cancer receive treatment in an ambulatory setting (Conley et al., 2009). This means that they are at home between treatments, and initial efforts to 71 manage symptoms takes place at home. Symptoms are dynamic and can change because of different treatment schedules, medication regimens, and many other factors (Kirkova, Aktas, Walsh, & Davis, 2011) Because of the symptom identification and management occurring at home, children and their families can have trouble remembering the information at their next outpatient visit where the focus is often present-oriented. In children 6 years of age and younger, often it is the parent who identifies and manages symptoms as well as recalls symptoms experienced in between outpatient visits. Understanding how parents identify that their young child is experiencing a symptom and then deciding whether or not to intervene is something that has not been explored and could improve symptom management strategies in this population. Technology to Track Symptoms Studies addressing the use of technology for reporting symptoms in young children with cancer are sparse. Initial work has included adolescents and adults with cancer primarily (Baggott et al., 2012; Maguire et al., 2015; Rodgers et al., 2014; Tomlinson et al., 2014). However, some recent studies examining technology with symptoms have included school-age children. Children as young as 6 years of age reported symptoms, and many of them reported positive feedback regarding the technology to input symptoms (Ruland et al., 2008; Tsimicalis et al., 2018) Among adolescents, the work of Gibson and colleagues utilized a personal digital assistant (PDA) to deliver questions derived from symptoms included on the Memorial Symptom Assessment Scale before, during and after chemotherapy (Gibson et al., 2010). Another study, including adolescents and young adults (AYAs), reported daily adherence rates of >90% with electronic symptom diaries (Gibson et al., 2013). Additionally, AYAs 72 also reported few technical issues relating to the diary itself and reported it to be useful for themselves. Recent studies with adults have also included various forms of current technology to report symptoms (Bender, Yue, To, Deacken, & Jadad, 2013; Mirkovic, Kaufman, & Ruland, 2014; Pandey et al., 2013). Apps are being developed and utilized for cancer patients to not only track symptoms but also to receive answers to questions, learn about different cancers and treatments, and support medication adherence, among other reasons (Carroll et al., 2017; Pandey et al., 2013) Other tracking apps are currently used by many people in the United States. Many smartphone owners use fitness tracking apps such as Fitbit and MyFitnessPal with 23.6 and 11.7 million monthly users, respectively (“Verto Index Health and Fitness Apps,” 2017). With the number of smartphone users increasing, this number is only expected to increase. Approximately 77% of American adults own smartphones with the highest percentage of ownership among 18-49-year-olds (Rainie & Perrin, 2017). Potential benefits of using smartphone apps to support the care of younger childhood cancer patients and their parents, however, have not been explored at this date. Furthermore, studies evaluating the feasibility and acceptability of mobile technology to support symptom tracking by families of children with cancer are sparse. Smartphones have been used in adults with various types of cancer to support reporting chemotherapy-related symptoms/toxicity to their healthcare provider directly (McCann et al., 2009) Additionally, and integrative symptom monitoring and management system, called SymptomCare@Home© has been created and used in the outpatient setting for patients receiving chemotherapy (Beck et al., 2017; Mooney et al., 2014, 2017). In both studies, 73 participants reported that technology-based symptom reporting improved communication with providers, improved symptom management, and provided a feeling of security in knowing someone was aware of their symptoms. Both studies also reported high satisfaction with each tool. Electronic Versus Paper-and-Pencil Diaries Several studies have examined the differences between electronic and paper-andpencil diaries in a variety of conditions, including adult cancers, cystic fibrosis, asthma, and sleeping problems. Paper-and-pencil and electronic diaries have also been used in several study samples, including children, and even ill infants, in which parents provided proxy reports. (Kelly, Young, Lane, & Shames, 2004) Potential advantages of electronic diaries include a reduction in missing data, decreased burden for participants, convenience, and the ability to time-stamp diary entries (Gwaltney et al., 2008; Morren et al., 2009; Müller et al., 2011; Stone et al., 2003). However, the most significant advantage reported is the increase in adherence rates with electronic diaries with reports ranging from 83-94% compared to 11-20% for paper-and-pencil diaries (Gwaltney et al., 2008; Stone et al., 2003). Although reported adherence rates are greater for electronic diaries, a recent review of the literature reported a high level of agreement overall between electronic and paper-and-pencil diaries (Gwaltney et al., 2008; Müller et al., 2011). Whether using paper-and-pencil or electronic diaries to track symptoms in teenagers and adults, increased awareness of symptoms was often an unanticipated result of using the diary(Bennett et al., 2010; Gwaltney et al., 2008; Kelly et al., 2004; Stone et al., 2003). 74 Theoretical Framework The overarching framework for this study is the sociotechnical framework, adapted from the approach described by Barber, Cornford, and Klecun (Barber et al., 2007). The sociotechnical framework describes how humans and technology interact and how this interaction can affect outcomes. For this study, the Barber and colleagues’ description has been modified to exclude the organizational component of the model because the emphasis of this study was to examine aspects of usability (Table 5.1). The concept of “Perceived Usefulness” from the Technology Acceptance Model has also been included to gain parent perspectives on the use of the two types of diaries (Davis, 1989). User satisfaction, an aspect of usability, will also be evaluated in this study (usability.gov, n.d.). Purpose The purpose of the study was to compare parents’ perceived usefulness of and satisfaction with a smartphone app symptom diary to a paper-and-pencil symptom diary to report their child’s symptoms and to explore how parents used each diary and their preferences for a given diary type. Methods Study Design This study used a mixed-methods exploratory, crossover design. Data were collected prospectively over two visits. These visits corresponded to intervals between scheduled outpatient clinic visits for chemotherapy based on the child’s treatment protocol. A crossover design was implemented to increase the power of the study by 75 matching participants with themselves to control for variations between participants by having them serve as their own controls (Garcia et al., 2004). Power analysis for a Wilcoxon signed-rank test was conducted in G*Power to determine a sufficient sample size using an alpha of 0.05, a power of 0.80, a large effect size (dz = 0.8), and two tails (Faul, Erdfelder, Lang, & Buchner, 2007). Based on the assumptions above, the desired sample size was 15. Study Setting and Sample Setting The study setting was a hematology/oncology clinic that is part of the Cancer Transplant Center within a tertiary, free-standing children’s hospital in the Intermountain West of the United States. The clinic serves children with all types of cancer and hematological conditions as well as those who have received hematopoietic stem cell transplants. This cancer transplant center sees approximately 40-50 new cancer diagnoses in children less than 6 years of age each year. Sample Inclusion criteria for child participants were a diagnosis of childhood cancer and between the ages of birth to 6 years. Child participants also had to be treated at the outpatient clinic with chemotherapy for cancer with at least three scheduled visits and be at least 30 days post initial diagnosis or relapse. Children were also required to be receiving chemotherapy with a similar degree of intensity at each of the three visits to reduce the likelihood of children’s conditions changing significantly during the course of the study. Children who were receiving palliative or end-of-life care were excluded. 76 Parent participants were required to be the parent or legal guardian of the child who met inclusion criteria and be able to speak, read, and understand English. Parents who had physical or cognitive impairments that inhibited their ability to complete the study procedures were excluded. Measures Symptom Diaries Two symptom diaries were used in this study: the iChemo Diary version 1.1(Merck & Co, 2011) and a paper-and-pencil symptom diary. The iChemo Diary app is a free smartphone/tablet app developed by Merck (Merck & Co, 2011). This touch screen app supports daily reporting and tracking of commonly occurring physical symptoms related to cancer treatment. Symptoms include nausea, pain, fatigue, vomiting, fever, lack of appetite, tingling in extremities, diarrhea, constipation, and rash. A free-text portion is also provided for listing other symptoms. If parents did not have a smartphone/tablet or a compatible device, one was loaned to them to use during the study. Data from the iChemo diary were saved locally on the device as the parent entered the information. Additionally, the iChemo diary provided a line-graph representation of symptoms that the parent could use as a reference if desired. The paper-and-pencil diary was developed by the principal investigator and included the same items and format as the iChemo electronic version. Parent Interviews Brief interviews were conducted with parents after the use of each diary. Interviews addressed parents’ actual use of the diary and its perceived usefulness. 77 Questionnaire for User Interface Satisfaction At the end of each visit, part 3 of the Questionnaire for User Interaction Satisfaction (QUIS) 7.0 was administered to the parent electronically evaluate diary user satisfaction (“Questionnaire for User Interface Satisfaction,” n.d.) The QUIS was developed at the University of Maryland to measure user satisfaction with computers and electronic tools. The QUIS is a modular tool composed of several semantic differential scales measuring user satisfaction. Because of its hierarchical nature, certain sections may be included or omitted with no major effects on the reliability of the tool. (“Questionnaire for User Interface Satisfaction,” n.d.) For this study, the portion “Part 3: Overall User Reactions” of the QUIS was used to measure users’ overall reactions. For each item, a score of 1 indicates a negative response, and a score of 9 indicates a positive response. This 5-item questionnaire overall has demonstrated acceptable internal consistency (Cronbach’s alpha = .88 -.96) and high overall user satisfaction with the questionnaire. (Chin et al., 1988; Harper, Slaughter, & Norman, 1988; Slaughter & Norman, K. L. Schniederman, 1995) Construct validity has been established for each of the six scales within the questionnaire. The QUIS has also been used to evaluate users’ reactions to other computer tools in previous nursing research (Hortman & Thompson, 2005; Johnson et al., 2004; Staggers et al., 2007). The QUIS questionnaire took less than 5 minutes to complete. Technology Acceptance Model: Perceived Usefulness Questions adapted from the perceived usefulness component of the Technology Acceptance Model Measurement (TAM) scales were administered electronically using the electronic capture tool, RedCap (described below) to gauge parents’ perceived 78 usefulness of both diaries (Davis, 1989). This tool includes five items ranked on a 7-point semantic differential scale ordered from extremely likely to extremely unlikely. In previous studies, reliability (α = .87 to .98) was demonstrated (Venkatesh & Davis, 2000). Parents completed the TAM items in less than 5 minutes. Item scores were averaged to create one score with higher/lower scores indicating greater perceived usefulness of each diary. Procedures Institutional Review Board (IRB) approval was granted for this study. Potentially eligible participants were screened weekly by the principal investigator and an oncology clinic team member. The oncology team member approached the potential participants with the study purpose and to obtain permission for the principal investigator to contact them. The principal investigator then met with the parents to explain the study and obtain both informed consent for the parent and parental permission for child participants. Because all children were less than 7 years of age, written assent was not obtained. At visit 1, participants were enrolled and randomly assigned one of the diaries to use until their next appointment. At visit 2, parents returned the completed diary and took the other diary home to complete. Parents also completed the two measurement tools and a short interview. At visit 3, parents returned the other diary, completed the two measurement tools, and again participated in a short interview. At this visit, parents were also asked an additional question about which diary they preferred and why. For both diaries, parents received daily reminders to complete the diary based on their preference of text, phone call, email, or smartphone calendar. Each diary took between 5-10 minutes to complete each day. All interviews were digitally audio-recorded and transcribed. Data 79 obtained from these interviews were analyzed qualitatively using content analysis. Data Management and Analysis Data were obtained from parents per the protocol schedule by the principal investigator at each clinic visit. Data from all surveys and instruments were entered into and managed using REDCap at the University of Utah (Harris et al., 2009). REDCap (Research Electronic Data Capture) is a secure, web-based application designed to support data capture for research studies, providing 1) an intuitive interface for validated data entry; 2) audit trails for tracking data manipulation and export procedures; 3) automated export procedures for seamless data downloads to common statistical packages; and 4) procedures for importing data from external sources. REDCap provides a secure place to create electronic surveys and store data and is available for use at the University of Utah. Quantitative data were downloaded into IBM SPSS Statistics Software version 23.0 for analysis. Qualitative data were organized into Microsoft Excel spreadsheets to support content analysis procedures. ID numbers were assigned to participant diary responses to ensure confidentiality. All electronic data were transferred to an encrypted partition on the hard drive of a password-protected computer. Paper-and-pencil diary responses were stored in a locked cabinet in a locked office. Wilcoxon Signed-Rank test compared the user satisfaction (QUIS 7.0) and perceived usefulness (TAM) of each diary type. A Wilcoxon Signed-Rank test was used because of the small sample size and because the dependent variable was not normally distributed. For ease of interpretability, means and standard deviations will be reported instead of positive and negative mean ranks. The average score per question for both the 80 QUIS and the TAM was used as input to the Wilcoxon Signed-Rank Test, for n = 20 participants. Qualitative Data Management and Analysis Qualitative data were analyzed using qualitative content analysis to summarize and organize parent interview responses (Elo & Kyngäs, 2008). Parent responses to each item were used as the unit of analysis. Responses were transcribed verbatim and organized into categories and subcategories. Wilcoxon Signed-Rank test was used to compare the frequency of responses between diaries regarding diary use. Again, for interpretability purposes, means and standard deviations are reported instead of positive and negative ranks. Results Participants Study participants were twenty children and twenty parents (Table 5.2). Thirteen child participants (65%) were female with a mean age of 3.05 years (range <1-6 years). Children were receiving treatment for a primary diagnosis of acute lymphoblastic leukemia (n = 13) and Wilms tumor (n = 7). Ninety percent (n = 18) of parent participants were female with a median age of 31 years (range 18-45 years). All child and parent participants were White, non-Hispanic. All parents reported using electronic devices, including smartphones, computers, and tablets. One parent also reported using an “Xbox.” After hearing about the study, one parent declined to participate. The reason given was “did not have enough time to add another thing” to her life. No participants withdrew once enrolled in the study. 81 Diary Completion Rates Completion rates were similar between the two diaries. Completion rates, as well as the most frequently reported symptoms and their associated characteristics, have been previously reported (in Chapter 4). Perceived User Satisfaction and Usefulness QUIS 7.0 Perceived user satisfaction with the electronic diary was greater compared with that of the paper diary (Table 5.3).The mean for user satisfaction was TAM was 2.02 for the electronic diary compared with 3.52 for the paper-and-pencil diary (z = -3.454, p = .001). For this instrument, lower scores indicated more agreement. Internal consistency was acceptable with a Cronbach’s alpha coefficient of .753. TAM Questions Parents’ perceived usefulness of the electronic diary was also greater than that of the paper diary (Table 5.3). The mean for perceived usefulness was 8.1 for the electronic diary compared with 5.39 for the paper-and-pencil diary (z = -3.061, p = .002). Parent Interviews Each question answered by parents was analyzed as a unit of measurement. One of the questions was only asked after the electronic diary (use of electronic devices). Table 5.4 summarizes the questions and the categories represented in parents’ responses. Usage and perceived usefulness were similar across diary type. 82 Diary Use When deciding how to use the symptom diary, three categories and eight subcategories emerged from the interviews with the most frequently described being routine or as directed (n = 32). One parent using the electronic diary described that they “…used it every day just to see how he was doing.” Parents using the paper-and-pencil dairy also reported that they “…filled it in after the reminder.” Parents also reported using the diary for symptom tracking and identification (n = 14). One parent reported showing the electronic diary to an emergency room provider when their child was sick. Another parent using the paper-and-pencil dairy reported that they used the diary to “...see how sick he was after his chemo and stuff.” Three categories also emerged regarding when symptom information was entered/filled out in the symptom diary: Evenings/before bed after the reminder was sent, and at home together. One parent utilizing the electronic diary stated that they would fill out the diary “Just before I went to sleep I would do it.” The majority of parents reported using a similar time of day to complete the diaries. Some parents also reported that they waited for the reminder from the study to remember to complete the diary, and one parent reported waiting until her spouse was home to complete the symptom information together. Using the paper-and-pencil diary, one parent stated that “…I think it made more sense to see how things went through the day, so I would do it right before bed.” Wilcoxon Signed-Rank test was used to compare differences in the frequency of category responses regarding diary use. The means between diaries showed no significant difference in diary use based on diary type for all categories with a total mean for the electronic of 2.05 and the paper-and-pencil at 1.9 (z =-. -1.13, p = .26). All results are 83 depicted in Table 5.5. When parents were asked about what they did with information from the diary, parents identified four categories. These included symptom trends/review, used as directed, shared, and other. Most parents identified that they used the diary for examining symptom trends and to review symptoms before attending their child’s next outpatient visit. “I just looked at it myself to see how she was during the week since we go in every week for chemo,” said one parent completing the paper-and-pencil diary. Another parent using the electronic dairy stated that “…We just looked at it to see you know patterns or things like that.” Some parents also reported just using the diary as directed by the study. One parent using the electronic diary stated that they “…We didn’t think we were supposed to for the study. Were we?” Some parents also reported sharing the symptom information with someone. Often this was a spouse or family member. Usefulness of Diary All parents reported that either diary would be useful in reporting their child’s symptoms to their child’s provider. One parent commented, “Yeah, I honestly thought it's a really good idea to just keep it to show to the provider…” As to why the diary would be useful, parents identified five categories. Most of these related to reminding parents of what to check for or to remember what symptoms their child experienced in between outpatient visits. Parents once again identified assessing symptom frequency and trends as important reasons for why the diaries were useful. A few parents also felt that the diary might best be used for patients at diagnosis or during times of increased symptoms (timing). Another parent commented that the idea of a diary was “great” but that “…not everyone would use it because it does require a few minutes every day…” The timing of 84 tracking symptoms may be an important aspect clinically for healthcare providers. Diary Preference Nineteen parents reported the electronic diary to be more useful than the paperand-pencil diary. The most common response that emerged in support of the electronic diary was usability (n = 18). Parents reported that the electronic diary had more features like portability and more timely completion, which made it easier to use (n = 8). They also commented on the electronic diary being more familiar to them (n = 5). The one parent that preferred both of the diaries reported that the paper-and-pencil diary was “easier to figure out” than the electronic diary. These are all components of usability which impact user satisfaction. Parents reported ease and little time needed when reporting data in each diary (Table 5.6). The electronic diary was reported as being more convenient, portable, and easier to use than the paper-and-pencil diary by the majority of parents. Other parents reported that they were able to input data quickly, and each diary was easy to use when recording their child’s symptom information. Discussion This study evaluated the perceived satisfaction and parent user reactions to the iChemo app and a paper-and-pencil diary. Results indicate that parents’ uses for each diary did not differ based on the type of diary. However, there were different reasons for why and how parents used the diaries in general. Parents also reported finding both diaries useful but had greater user satisfaction with the iChemo diary and greater perceived usefulness compared with the paper-and-pencil diary. 85 The iChemo diary demonstrated the ability to capture meaningful and helpful data into the symptom experience of young children with cancer. Perceived usefulness and perceived user satisfaction scores were greater for the electronic diary when compared with the paper-and-pencil diary. Parents expressed that the diary was important because they were managing their child’s care at home and not in the hospital. The features of the electronic diary also made it more appealing than the paper-and-pencil dairy. Parents found the convenience and the ability to help remember symptoms for upcoming chemotherapy appointments helpful. The visual data was also used by many of the parents to see how often their child was experiencing a symptom. Although most parents preferred the electronic diary, many parents commented that having any diary would be useful with one parent commenting that a choice of diary should be offered. This finding could be generalizable to other apps offering similar symptom tracking benefits since this study focused on the process rather than specific app features. Most parents used the diary as requested per study procedures or at a certain time of day. However, many parents reported using the diary for symptom tracking, a reminder in between visits, or a cue to remind them to ask about symptoms. Parents also noted that they would have shown it to someone if they had been suggested to do so. Incorporating processes in the clinical setting to prompt providers to ask about symptoms and patterns could be important. Parent responses to interview items suggest that tracking symptoms between visits could improve communication between parents and clinicians, which, in turn, could facilitate symptom management. Parents of young children with cancer perceived the electronic diary to be more convenient yet also reported that having some sort of 86 symptom tracking system in place would be beneficial. Many parents reported using the diaries to help track symptoms in between outpatient visits and found the diaries helpful to remember which symptoms occurred on which days, and how often symptoms were occurring. Electronic and paper-and-pencil symptom diaries showed potential for families of young children with cancer to record and track daily physical symptoms while at home in between clinic visits. This study builds upon limited research involving young children with cancer and mobile health apps. Because many young children with cancer are treated in ambulatory settings, parents are often required to remember symptoms as well as if they intervened upon those symptoms. This study contributes to the body of knowledge by supporting the use of mobile apps or diaries to help parents track and view their child’s symptoms (Baggott et al., 2012; Gibson et al., 2013; Smaldone, Tsimicalis, & Stone, 2011). All parents acknowledged that having some way to track would be useful and found the iChemo diary to be the most useful because of its portability, accessibility, visual representation of symptoms, and other reasons. Providing parents with a way to track symptoms could lead to better and more timely symptom interventions by parents and healthcare providers. Future Directions Parents of young children with cancer who are treated in the outpatient setting have to monitor and manage their child’s symptoms at home. Having a resource to help support parents in reporting symptoms to clinicians could be valuable in reducing the frequency, severity, and symptom burden of their children. Possible future studies include integrating parent-reported symptoms into their child’s Electronic Health Record (EHR) 87 in real time by utilizing symptom data entered into patient portals like MyChart. Clinicians could then respond to symptoms directly is needed to support parents via a mobile app. Further evaluation of clinician responses to symptoms entered by parents could also be done to help standardize common symptom management protocols among clinicians. Having this data integrated into the EHR would also be beneficial for parents because this data would be available to all clinicians that they may encounter during their child’s treatment. This data would be helpful if their child is admitted to the inpatient unit, emergency room, or another unit. Some parents also reported that the timing of symptom diary use might be important. Utilizing symptom diaries at times of diagnosis, surgeries, or other treatments could be especially useful in monitoring and managing symptoms. These are times when patients often report more symptom occurrence and severity and may need additional support from providers. Additionally, with a new diagnosis, parents are still learning what symptoms often occur, how to identify them, and when provider notification may be necessary. Having a diary to prompt parents on what to anticipate or look for in their children could be helpful, especially while managing the new diagnosis and symptoms at home. The findings from this study provide support for future interventional studies using symptom diaries in outpatient settings. This would help parents to remember their child’s past symptoms, especially when there are weeks between outpatient chemotherapy visits. Care providers should offer parents a choice of diary that they prefer to record symptoms. Additionally, a system could be set up to link the diary to providers 88 so that symptoms could be reported in real time. Having a threshold where a provider or nurse could be notified when symptom severity increased or if the frequency and occurrence of symptoms increased could help with symptom management at home. This system could even start with app alerts to support parents in recognizing when a call to a provider might be appropriate. Technology, such as an automated phone monitoring system has been used with success in adult cancer populations. (Beck et al., 2017; Mooney et al., 2014, 2017). Limitations Limitations of this study include generalizability, time-recording for the symptom diaries, and parent familiarity with technology. Although this study does meet recommendations for usability pilot studies, the sample size is still relatively small, which makes generalizability beyond this study difficult. The study was also cross-sectional and only included participants with two different cancer diagnoses and treatment plans. The racial and ethnic diversity of the sample was also limited since all patients were white and non-Hispanic. Additionally, because the daily diary information was collected at the next clinic visit to decrease the burden on families, no time stamp of when the diary was filled out and if the diary was completed each day was identified. Additionally, the majority of parents were younger and familiar with technology, which may have made the electronic diary more favorable than the paper-and-pencil diary. Conclusion Young children with cancer treated in the outpatient setting experience symptoms that their parents must monitor, track, and respond. Parents in this study overwhelmingly 89 found the electronic symptom diary more useful for reporting their child’s symptoms than a traditional paper-and-pencil diary. Offering symptom diaries to parents of children with cancer could provide more timely symptom identification and ultimately, intervention. Providers should be routinely asking parents about symptoms at home and the management of symptoms. Future use of implementing a real-time tracking and reporting tool could ultimately lead to better symptom management and symptom relief. 90 Table 5.1 Application of the Sociotechnical Framework Concepts Technical System Function Social Human Perspective Structure Symptom diaries Children Parents Process Electronic diary Paper-and-pencil diary Interaction Outcome Feasibility/acceptance Diary compliance Symptoms Perceived usefulness 91 Table 5.2 Parent Demographics Characteristics Gender Male Female n % 2 18 10% 90% Age 18-25 26-35 36-45 3 14 3 15% 70% 15% Marital Status Married 20 100% Education Level High school/GED Some College College Graduate Post-Graduate 1 8 10 1 5% 40% 50% 5% Number of children 1 2 3 4+ 1 13 3 3 5% 65% 15% 15% Ethnicity White 20 100% Reminder Requested Email Text Message Phone calendar reminder Ipod Calendar reminder Phone call 5 8 2 5 0 25% 40% 10% 25% 0% 92 Table 5.3 Wilcoxon Signed-Rank Test Results Comparing TAM and QUIS 7.0 Responses Between Diaries Survey Diary Type M SD z p TAM Electronic Paper 2.02 3.52 1.55 1.19 –3.061 .002 QUIS 7.0 Electronic Paper 8.1 5.39 .75 2.15 –3.45 .001 93 Table 5.4 Categories and Subcategories of Parents’ Responses to Interview Questions Category Electronic Diary Frequency Paper-and-Pencil Diary Frequency Using the Symptom Diary How did you decide to use the symptom diary? Routine/as directed 19 Symptom Tracking & Identification 5 Other – showed ER provider 1 13 9 0 When did you fill out/enter information into the diary? Evenings/before bed After reminder At home together 20 3 0 19 3 1 What did you do with the information you filled in/entered into the symptom diary? Symptom trends & review 14 Used as directed 4 Shared 4 Other 1 Usefulness of Diary Do you think this diary could be useful in reporting symptoms to your child’s provider? Yes 20 No 0 Why or why not? Reminder 9 Trends 3 Symptom Frequency 2 Timing 2 Useful, no reason given 6 Was information shared with oncology provider Yes Used as a reminder No, used as directed 0 0 20 12 7 2 1 20 0 9 6 2 1 6 1 1 19 94 Table 5.5 Wilcoxon-Signed Ranks Comparing Diary Use Responses by Diary Type Category Diary Type M SD Median Electronic .95 .61 1 Paper .65 .49 1 Electronic .25 .44 0 Paper .45 .51 0 Electronic 1 .58 1 Paper 0 0 0 Electronic 1.25 .44 1 Paper 1.1 .31 1 Routine Symptom tracking Other Total z p –1.73 .08 –1.41 .16 –1 .32 –1.13 .26 95 Table 5.6 Diary Preference and Rationale Categories and Sub-Categories N Diary Preference Electronic Paper and pencil Both 19 0 1 Rationale Usability Features Familiarity Paper diary more intuitive 18 8 5 1 Other Comments Choice should be given for type of diary 1 CHAPTER 6 DISCUSSION AND CONCLUSIONS Discussion This study expands on the limited symptom literature of young children with cancer and advances the understanding of symptoms in children less than 6 years of age receiving treatment. Children less than 6 years of age represent the largest group of children with cancer under the age of 14, and yet, descriptions of their symptom experience are under-represented in the literature. Understanding the symptom experience of young children with cancer can lead to future interventions and prompter identification of symptoms. Additionally, since children with cancer often receive many treatments in the outpatient setting, exploring how parents identify symptoms in their young children can further guide an understanding of the complete symptom experience in this population. Key findings from this study indicate that young children experience frequent symptoms while at home in between outpatient visits. Sometimes these symptoms are moderate or severe, and parents use different methods of identifying and assessing symptoms. This study also reveals that any sort of symptom diary would be useful for parents of young children with cancer, but parents did prefer the electronic symptom diary. 97 Theoretical Frameworks This study was guided by the sociotechnical framework as described by Barber, et al. (2007), the UCSF symptom management theory and the Technology Acceptance Model (TAM) (Barber et al., 2007). The sociotechnical framework proved useful as an over-arching framework when evaluating the individual human-technical interaction. This framework supported this study since the usability of two different diaries and the parent interaction and preference with these diaries intertwine. Since perceived usefulness was an aim of this study, the TAM was also incorporated. This model supported the aim by describing the importance of perceived usefulness in making the behavioral choice to use a particular symptom. This was key since parents were evaluating diary preference and if a diary was at all useful. Finally, the “symptom experience” portion of the UCSF symptom management theory addressed the symptoms experienced by young children. This portion of the theory provided a reference as to how symptoms are perceived, evaluated, and responded to. The theory recognizes self-report as the gold standard for symptom perception but acknowledges the need for parent-report with younger children (Bender, M.S., 2018). Although this study did not focus on the response to symptoms, the importance of how parents identified symptoms was a main component of the symptom experience (Bender, M.S., 2018 Venkatesh & Davis, 2000). According to the theory, the way a person identifies differences in how they feel or they vary from normal is important for the perception of a symptom. In this study, the way that parents often identified that their child was experiencing a symptom was noticing changes in behavior, activity, or consolability. Within the symptom experience, evaluation of the symptom refers to how a 98 person interprets the severity, cause, impact on life, or interventions needed for a particular symptom. Parents in this study often relied on how different from “normal” their child was behaving to determine severity. This does not correlate perfectly with this theory, but the impact on a parent’s day to day life would most likely be affected by behavioral changes seen in their child. Summary of Results Symptom Experience and Identification Results from this study indicate that young children with cancer experience frequent, daily symptoms. These findings are similar to other studies conducted with children of all ages with cancer with some children reporting >10 cooccurring symptoms (Baggott, Dodd, Kennedy, Marina, & Miaskowski, 2009; Torres et al., 2019). Fatigue and nausea were present more than half of the reported diary days followed by lack of appetite 37.8% of the time. Parents of 19 children also reported their child experiencing at least 1 day of both fatigue and nausea followed by lack of appetite (16 children) and pain (15 children). Of these three symptoms that included a rating for severity, fatigue was most frequently reported as moderate or severe (17.5% of reported days), followed by nausea (6.4%), and pain (5%). Parents of 10 children reported at the child experienced at least one day of moderate or severe ratings for both fatigue and nausea, and six children experienced at least 1 day of moderate or greater pain. Although ratings of moderate or greater severity for these three symptoms were present on fewer than 20% of reported days, any symptom experienced even at a mild rating is not a “normal” occurrence for a child. Two of the children had 11 reports of moderate or higher symptoms, followed by one with nine. The other children had three or 99 fewer reports of moderate or higher severity. These findings indicate that some children experience more frequent and more severe symptoms than other children despite having similar diagnoses, ages, and treatment. Young children living with chronic illnesses that also produce persistent symptoms struggle with child development. Impacts on gross and fine motor skills, physical and psycho-social development, and cognitive development have all been seen in children living with various chronic illnesses (Curtis & Luby, 2008; Goldberg et al., 1997; Ohan, Glauert, Bell, Harrison, & Bayliss, 2016). Addressing symptoms, even if they are not severe, is important for clinicians to assess in order to prevent developmental delays later on. Parents serve as advocates for their child and are vital to how symptoms are reported and managed in young children with cancer. Results from parent interviews regarding symptoms revealed that parents used more than one way to identify symptoms and severity in their children. Symptom identification is important because it may provide clues on how best to obtain symptom information from young children. Several studies have shown that utilizing different methods of gathering symptom data in young children may be required in order to truly understand their experience (Battista et al., 2017; L. A. Linder & Wawrzynski, 2018; Pinheiro et al., 2018; D. Tomlinson et al., 2017). Parents often relied on the child’s activity level & consolability, verbal cues in older children, or discussing with their spouse if they thought a particular symptom might be present. Although different ways were utilized in identification, the frequencies of responses did not differ based on diary type (z = -.52, p = .6). 100 Usability and Perceived Usefulness of Symptom Tracking Diaries Parents reported greater perceived usefulness and user satisfaction with the electronic symptom diary than with the paper-and-pencil diary. Parents preferred the electronic diary because of the features, the ease, portability, and familiarity of the electronic diary. These findings are consistent with other studies that have utilized both diary modalities in various ages and populations (Baggott et al., 2012; Jensen, 2014; Palermo, Valenzuela, & Stork, 2004; Stone, Shiffman, Schwartz, Broderick, & Hufford, 2003; Whalen et al., 2006). When describing how parents used the symptom diaries, most parents stated that they used the diary as requested or to track and identify symptoms. The frequency of categories reflected in responses did not differ based on diary type (z = -1.13, p = .26). Most parents reported completing the diary at the same time each day and often in the evening. Parents also appreciated a reminder each day to remind them to complete the diary. When parents were asked about what they did with the information in the diary, the majority explained that they used the diary to examine symptom trends and to review symptoms before an outpatient appointment. Some parents also reported sharing the information with someone which was usually a spouse or family member. Parents liked both diaries but preferred the electronic diary. Parents reported that they found both to be useful to track their child’s symptoms. Although the iChemo app is no longer available other electronic symptom apps and resources are available for symptom tracking. Recommendations for Future Research Results from this study indicate that young children with cancer experience symptoms, whether self-reported or reported by their parents. Because of age, very few 101 self-report measures exist for young children although visual measures could be a possibility for children as young as 3 (Dupuis et al., 2001; Pinheiro et al., 2018). Very few studies exist regarding symptoms in children 6 and younger. Exploring this age group separately from others could help shed light on this area. Additionally, studies examining differences in gender, race, and other types of pediatric cancers are also warranted. Parents of young children with cancer perceived the electronic diary to be more convenient yet also reported that having some symptom tracking system in place would be beneficial. Many parents reported using the diaries to help track symptoms in between outpatient visits. Electronic and paper-and-pencil symptom diaries showed potential for families of young children with cancer to record and track daily physical symptoms while at home in between clinic visits. Some parents also reported the diaries might be useful at certain times during their child’s treatment. At diagnosis or during more rigorous treatment protocols could be times when symptom tracking could be used instead of during a child’s entire treatment. Timing is important for clinicians to consider since they could recommend symptom tracking to parents at certain time points during their child’s treatment. The findings from this study provide support for future interventional studies using symptom diaries in the outpatient setting. Diaries could help parents to remember their child’s past symptoms, especially when there are weeks between outpatient chemotherapy visits. Care providers should offer parents a choice of diary that they prefer to record symptoms. Additionally, a connected system in which providers could receive symptom information in real-time could help to support parents in managing symptoms at 102 home. Additionally, evaluating the clinician’s response to the symptom data could also be useful for future research. Recommendations for Practice Incorporating processes in the clinical setting to prompt providers to ask about symptoms and patterns could be important. Outpatient visits are often present-focused on current symptoms and not on trends or past symptom experiences. A systematic approach to examining these trends could be beneficial for families since the recall of past symptoms is often difficult. This approach could also be advantageous for some parents who may be reluctant to report symptoms or increases in severity because they believe it is a part of the cancer experience (R. L. Woodgate et al., 2003). Parent responses to interview items suggest that tracking symptoms between visits could improve communication between parents and clinicians, which, in turn, could facilitate symptom management. Nurses caring for young children with cancer should anticipate persistent symptoms even in the outpatient setting. Information gleaned from parents about symptoms their child experienced in between visits or is currently experiencing could be useful for clinicians. The symptoms experienced, as well as how parents identified symptoms could help clinicians support other parents of children with cancer. Knowing common cues and behaviors that many parents report as being key to symptom identification could help newly diagnosed families better identify and thus, better manage symptoms. Additionally, studies regarding symptom identification in this population could also help to set institutional or national standards regarding symptoms similar to what is incorporated in many adult hospitals and institutions. 103 Recommendations for Policy Integrating more patient data into EHR systems from symptom diary reports at home could support parents and clinicians alike. Clinicians at outpatient visits often see only a snapshot of what symptoms a child has experienced since the last visit. Examining daily symptoms can help prompt clinicians to discuss how parents are identifying these symptoms and if and when they manage them. Utilizing tools like patient portals where patients or parents could record symptoms could be better integrated into EHR systems to support daily symptom entry. Clinical decision support utilizing best practice advisory/alerts (BPAs) could be implemented to guide clinicians on when and how to intervene with symptoms. Decision support should be done using evidence-based guidelines, which often are not present or scarce when it comes to guiding care for children. Formalizing symptom assessment within the clinical setting is also important to implement for children with cancer. Additionally, in studies where clinicians received real-time symptom data, many clinicians did not act upon the symptoms or patients felt that their symptoms were not addressed (Marceau et al., 2010; Mooney et al., 2014). Having integrated infrastructure to support clinicians in decision-making, triage, and interventions are necessary to make system-wide changes. Recent work utilizing a triage symptom management system for nurses in adult cancer patients has shown some promise in identifying barriers to developing symptom protocols and use (Stacey et al., 2016). Limitations Limitations of this study include generalizability, time-recording with the symptom diaries, proxy-reporting, and the lack of reliability/validity measures with the 104 iChemo Diary. Although this study does meet recommendations for usability pilot studies, the sample size is still relatively small, which makes generalizability beyond this study difficult. This study used a cross-section design and included only white, nonHispanic participants. Additionally, because the daily diary information was collected at the next clinic visit to decrease the burden on families, no record to confirm when the diary was filled out and whether the diary was completed each day was available. Proxy-reporting is another recognized limitation of this study. Although the literature shows, proxy-reporting may be the most reliable source of information in young children since they may be too young to self-report, congruence between self-report and parent-report cannot be verified. In this study, the parents’ depiction of symptoms experience was utilized alone. The other limitation involves the iChemo Diary. To date, no studies or testing have been done using this application. Thus, the reliability of this tool cannot be assured. However, this study is exploratory, and investigating the usability of this tool is a primary aim of this study. Summary This study fills a gap in the existing body of symptom literature in young children with cancer by not only exploring symptoms in this population but by examining the usefulness of different symptom diaries for parents to track symptoms. This study also advances the science by demonstrating the perceived usefulness of symptom diaries for parents of young children with cancer. These benefits align with the National Institute of Nursing Research (NINR) initiatives in symptom management research, which focus on symptom identification, management, and symptom causations (National Institute of 105 Nursing Research, 2016). This study could greatly impact symptom care and treatment in the outpatient setting in the future since emerging technology is becoming more accessible. Future research could include actual implementation of the electronic diary to inform healthcare providers of the child’s status or symptoms that were experienced. APPENDIX A PARENT INTERVIEW QUESTIONS 1. Using the Symptom Diary • How did you decide to use the symptom diary? • How did you know your child was having a certain symptom? • How did you decide on how severe or mild the symptom was? • When did you fill out/enter information into the diary? • What did you do with the information you filled in/entered into the symptom diary? • Did you share the dairy information with anyone? Yes No o If yes, who? 2. Usefulness of the Symptom Diary • Do you think this diary could be useful in reporting symptoms to your child’s provider? Yes No § Why or why not? § If yes, which diary did you find more useful and why? 3. Experience • What electronic devices have you used previously? Tablet Smartphone Computer Others:____________________________________________________ 4. Improvements • Do you think that the diaries addressed important questions related to your child’s symptom experience? o Why or why not? • Are there questions that the diary should have asked about your child’s symptom experience? Yes No o If yes, what are they? APPENDIX B PAPER-AND-PENCIL VERSION OF THE ICHEMO DIARY 1) Please rate and circle the severity of: Nausea None Mild Moderate Severe Moderate Severe Moderate Severe Pain None Mild Fatigue None Mild 2) Vomited? Yes No If yes, number of times 3) Temperature (in Fahrenheit or Celsius) 4) Lack of Appetite? Yes No 5) Tingling in Feet/Toes? Yes No 6) Diarrhea? Yes No 7) Constipation? Yes No 8) Rash? Yes No 9) Please list and describe any other symptoms: APPENDIX C QUIS 7.0 SECTION PART 3: Overall User Reactions Please circle/select the numbers which most appropriately reflect your impressions about using this diary Not Applicable = NA. 3.1 Overall reactions to the diary: terrible 1 3.2 5 6 7 2 8 3 4 5 6 7 8 NA 9 NA stimulating 2 3 4 5 6 7 8 2 9 NA easy 3 4 5 6 7 8 rigid 1 9 satisfying difficult 1 3.5 4 dull 1 3.4 3 frustrating 1 3.3 2 wonderful 9 NA flexible 2 3 4 5 6 7 8 9 NA APPENDIX D PERCEIVED USEFULNESS QUESTIONS ADAPTED FROM THE TAM 1. Using the iChemo/Paper-and-Pencil diary would enable me to report my child’s symptoms more quickly. Likely |_________|_________|_________|_________|_________|_________|_________| Unlikely Extremely Quite Slightly Neither Slightly Quite Extremely 2. Using the iChemo/Paper-and-Pencil diary would improve the symptom experience for my child. Likely |_________|_________|_________|_________|_________|_________|_________| Unlikely Extremely Quite Slightly Neither Slightly Quite Extremely 3. Using the iChemo/Paper-and-Pencil diary would make reporting my child’s symptoms easier. Likely |_________|_________|_________|_________|_________|_________|_________| Unlikely Extremely Quite Slightly Neither Slightly Quite Extremely 4. Using the iChemo/Paper-and-Pencil diary is effective for reporting my child’s symptoms. Likely |_________|_________|_________|_________|_________|_________|_________| Unlikely Extremely Quite Slightly Neither Slightly Quite Extremely 5. I find the iChemo/Paper-and-Pencil diary useful for reporting my child’s symptoms. Likely |_________|_________|_________|_________|_________|_________|_________| Unlikely Extremely Quite Slightly Neither Slightly Quite Extremely REFERENCES Abu-Saad Huijer, H., Sagherian, K., & Tamim, H. (2013). Quality of life and symptom prevalence as reported by children with cancer in Lebanon. European Journal of Oncology Nursing, 17(6), 704–710. https://doi.org/10.1016/j.ejon.2013.09.004 Adlard, K., Savedra, M. C., Van Cleve, L., Bossert, E., Beecroft, P., Alvarez, O., … Savedra, M. C. (2004). The pain experience of children with leukemia during the first year after diagnosis. Nursing Research, 53(1), 1–10. https://doi.org/10.1097/00006199-200401000-00001 Baggott, C., Dodd, M., Kennedy, C., Marina, N., & Miaskowski, C. (2009). Multiple symptoms in pediatric oncology patients: A systematic review. Journal of Pediatric Oncology Nursing, 26(6), 325–339. https://doi.org/10.1177/1043454209340324 Baggott, C., Cooper, B. A., Marina, N., Matthay, K. K., & Miaskowski, C. (2014). Symptom assessment in pediatric oncology: How should concordance between children’s and parents’ reports be evaluated? Cancer Nursing, 37(4), 252–262. https://doi.org/10.1097/NCC.0000000000000111 Baggott, C., Dodd, M., Kennedy, C., Marina, N., Matthay, K. K., Cooper, B. A., & Miaskowski, C. (2010). Changes in children’s reports of symptom occurrence and severity during a course of myelosuppressive chemotherapy. Journal of Pediatric Oncology Nursing, 27(6), 307–315. https://doi.org/10.1177/1043454210377619 Baggott, C., Gibson, F., Coll, B., Kletter, R., Zeltzer, P., & Miaskowski, C. (2012). Initial evaluation of an electronic symptom diary for adolescents with cancer. Journal of Medical Internet Research, 1(2), e23. https://doi.org/10.2196/resprot.2175 Barber, N., Cornford, T., & Klecun, E. (2007). Qualitative evaluation of an electronic prescribing and administration system. Quality & Safety in Health Care, 16(4), 271– 278. https://doi.org/10.1136/qshc.2006.019505 Battista, A., Dupuis, L. L., Cassidy, M., Portwine, C., Johnston, D. L., Pradier Silva, M., … Barrera, M. (2017). Parent attributions about child symptoms related to cancer therapy. Journal of Pediatric Oncology Nursing, 34(1), 44–50. https://doi.org/10.1177/1043454215628002 Beck, S. L., Eaton, L. H., Echeverria, C., & Mooney, K. H. (2017). 111 SymptomCare\@Home: Developing an Integrated symptom monitoring and management system for outpatients receiving chemotherapy. Computers, Informatics, Nursing, 35(10), 520–529. https://doi.org/10.1097/CIN.0000000000000364 Bender, J. L., Yue, R. Y. K., To, M. J., Deacken, L., & Jadad, A. R. (2013). A lot of action, but not in the right direction: Systematic review and content analysis of smartphone applications for the prevention, detection, and management of cancer. Journal of Medical Internet Research, 15(12), e287. https://doi.org/10.2196/jmir.2661 Bender, M. S., Janson, S. L., Franck, L. S., & Lee, K. A. (2018). Theory of symptom management. In M. J. Smith & P. R. Liehr (Eds.), Middle range theory for nursing (4th ed., pp. 147–178). New York, NY: Springer Publishing Co., LLC. Bennett, A. V., Patrick, D. L., Lymp, J. F., Edwards, T. C., & Goss, C. H. (2010). Comparison of 7-day and repeated 24-hour recall of symptoms of cystic fibrosis. Journal of Cystic Fibrosis, 9(6), 419–424. https://doi.org/10.1016/j.jcf.2010.08.008 Blondin, J. M., Abu-Hasaballah, K. S., Tennen, H., & Lalla, R. V. (2010). Electronic versus paper diaries: A pilot study of concordance and adherence in head and neck cancer patients receiving radiation therapy. Head and Neck Oncology, 2(1). https://doi.org/10.1186/1758-3284-2-29 Carroll, J. K., Moorhead, A., Bond, R., LeBlanc, W. G., Petrella, R. J., & Fiscella, K. (2017). Who uses mobile phone health apps and does use matter? A secondary data analytics approach. Journal of Medical Internet Research, 19(4), e125. https://doi.org/10.2196/jmir.5604 Children’s Oncology Group: About Us. (n.d.). Retrieved from https://www.childrensoncologygroup.org/index.php/about-us Chin, J. P., Diehl, V. a, & Norman, L. K. (1988). Development of an instrument measuring user satisfaction of the human-computer interface. CHI ’88- Proceedings of the SIGCHI Conference on Human Factors in Computing Systems, 213–218. https://doi.org/10.1145/57167.57203 Collins, J. J, Byrnes, M. E., Dunkel, I. J., Lapin, J., Nadel, T., Thaler, H. T., … Portenoy, R. K. (2000). The measurement of symptoms in children with cancer. Journal of Pain and Symptom Management, 19(5), 363–377. https://doi.org/10.1016/S08853924(00)00127-5 Collins, John J., Devine, T. D., Dick, G. S., Johnson, E. A., Kilham, H. A., Pinkerton, C. R., … Portenoy, R. K. (2002). The measurement of symptoms in young children with cancer: The validation of the memorial symptom assessment scale in children aged 7-12. Journal of Pain and Symptom Management, 23(1), 10–16. https://doi.org/10.1016/S0885-3924(01)00375-X 112 Conley, S., Curry, J. O. H., Hines, M., Baker, K., Schmidt, K., Zwier, K., … Kline, N. (2009). APHON Position Paper on Ambulatory Pediatric Hematology / Oncology Nursing Practice, 1–5. Retrieved from http://aphon.org/UPLOADS/Education/pp2.pdf Curtis, C. E., & Luby, J. L. (2008). Depression and social functioning in preschool children with chronic medical conditions. Journal of Pediatrics. https://doi.org/10.1016/j.jpeds.2008.03.035 Davis, F. D. (1989). Perceived usefulness, perceived ease of use, and user acceptance of information technology. MIS Quarterly, 13(3), 319. https://doi.org/10.2307/249008 Docherty, S. L. (2003). Symptom experiences of children and adolescents with cancer. Annual Review of Nursing Research, 21, 123–149. Donabedian, A. (1988). The quality of care: How can it be assessed? JAMA: The Journal of the American Medical Association, 260(12), 1743. https://doi.org/10.1001/jama.1988.03410120089033 Dupuis, L. L., Ethier, M.-C. C., Tomlinson, D., Hesser, T., & Sung, L. (2012). A systematic review of symptom assessment scales in children with cancer. BMC Cancer, 12, 430. https://doi.org/10.1186/1471-2407-12-430 Dupuis, L. L., Lau, R., & Greenberg, M. L. (2001). Delayed nausea and vomiting in children receiving antineoplastics. Medical and Pediatric Oncology, 37(2), 115–121. https://doi.org/10.1002/mpo.1179 Dupuis, L. L., Milne-Wren, C., Cassidy, M., Barrera, M., Portwine, C., Johnston, D. L., … Sung, L. (2010). Symptom assessment in children receiving cancer therapy: The parents’ perspective. Supportive Care in Cancer, 18(3), 281–299. https://doi.org/10.1007/s00520-009-0651-1 Elo, S., & Kyngäs, H. (2008). The qualitative content analysis process. Journal of Advanced Nursing, 62(1), 107–115. https://doi.org/10.1111/j.13652648.2007.04569.x Erickson, J. M., MacPherson, C. F., Ameringer, S., Baggott, C., Linder, L., & Stegenga, K. (2013). Symptoms and symptom clusters in adolescents receiving cancer treatment: A review of the literature. International Journal of Nursing Studies, 50(6), 847-869. https://doi.org/10.1016/j.ijnurstu.2012.10.011 Faul, F., Erdfelder, E., Lang, A. G., & Buchner, A. (2007). G*Power 3: A flexible statistical power analysis program for the social, behavioral, and biomedical sciences. Behavior Research Methods, 39(2), 175-191. https://doi.org/10.3758/BF03193146 113 Faulkner, L. (2003). Beyond the five-user assumption: Benefits of increased sample sizes in usability testing. Behavior Research Methods, Instruments, and Computers, 35(3), 379-383. https://doi.org/10.3758/BF03195514 Gaertner, J., Elsner, F., Pollmann-Dahmen, K., Radbruch, L., & Sabatowski, R. (2004). Electronic pain diary: A randomized crossover study. Journal of Pain and Symptom Management, 28(3), 259-267. https://doi.org/10.1016/j.jpainsymman.2003.12.017 Garcia, R., Benet, M., Arnau, C., & Cobo, E. (2004). Efficiency of the cross-over design: An empirical estimation. Statistics in Medicine, 23(24), 3773–3780. https://doi.org/10.1002/sim.2072 Gibson, F., Aldiss, S., Taylor, R. M., Maguire, R., McCann, L., Sage, M., & Kearney, N. (2010). Utilization of the Medical Research Council evaluation framework in the development of technology for symptom management: The ASyMS-YG Study. Cancer Nursing, 33(5), 343–352. https://doi.org/10.1097/NCC.0b013e3181cb4bad Gibson, P., Tomlinson, D., Sung, L., Watson, J., Kuczynski, S., Dupuis, L. L., … Spiegler, B. (2013). Initial development of the Symptom Screening in Pediatrics Tool (SSPedi). Supportive Care in Cancer, 22(1), 71–75. https://doi.org/10.1007/s00520-013-1945-x Goldberg, S., Janus, M., Washington, J., Simmons, R. J., Maclusky, I., & Fowler, R. S. (1997). Prediction of preschool behavioral problems in healthy and pediatric samples. Journal of Developmental and Behavioral Pediatrics, 18(5), 304-313. https://doi.org/10.1097/00004703-199710000-00004 Gonzalez-Mercado, V. J., Williams, P. D., Williams, A. R., Pedro, E., & Colon, G. (2017). The symptom experiences of Puerto Rican children undergoing cancer treatments and alleviation practices as reported by their mothers. International Journal of Nursing Practice, 23(1), e12500. https://doi.org/10.1111/ijn.12500 Gwaltney, C. J., Shields, A. L., & Shiffman, S. (2008). Equivalence of electronic and paper-and-pencil administration of patient-reported outcome measures: A metaanalytic review. Value in Health, 11(2), 322-333. https://doi.org/10.1111/j.15244733.2007.00231.x Hagan, J. F., Duncan, P. M., & Shaw, J. . (2008). Bright futures: Guidelines for health supervision of infants, children, and adolescents, 3rd edition, pocket guide. Elk Grove Village, IL: The American Academy of Pediatrics. Harper, B., Slaughter, L., & Norman, K. (1988). Questionnaire administration via the WWW : A validation & reliability study for a user satisfaction questionnaire. In Proceeding of WebNet 97 (pp. 3–6). Harris, P. A., Taylor, R., Thielke, R., Payne, J., Gonzalez, N., & Conde, J. G. (2009). Research electronic data capture (REDCap)-A metadata-driven methodology and 114 workflow process for providing translational research informatics support. Journal of Biomedical Informatics, 42(2), 377–381. https://doi.org/10.1016/j.jbi.2008.08.010 Hedström, M., Haglund, K., Skolin, I., & Von Essen, L. (2003). Distressing events for children and adolescents with cancer: child, parent, and nurse perceptions. Journal of Pediatric Oncology Nursing, 20(3), 120–132. https://doi.org/10.1053/jpon.2003.76 Hendershot, E., Murphy, C., Doyle, S., Van-Clieaf, J., Lowry, J., & Honeyford, L. (2005). Outpatient chemotherapy administration: Decreasing wait times for patients and families. Journal of Pediatric Oncology Nursing, 22(1), 31-37. https://doi.org/10.1177/1043454204272539 Hinds, P. S., Quargnenti, A. G., & Wentz, T. J. (1992). Measuring symptom distress in adolescents with cancer. Journal of Pediatric Oncology Nursing, 9(2), 84–86. https://doi.org/10.1177/104345429200900238 Holden, H., & Rada, R. (2011). Understanding the influence of perceived usability and technology self-efficacy on teachers’ technology acceptance. Journal of Research on Technology in Education, 43(4), 343–367. https://doi.org/10.1080/15391523.2011.10782576 Hortman, P. A., & Thompson, C. B. (2005). Evaluation of user interface satisfaction of a clinical outcomes database. CIN - Computers Informatics Nursing, 23(6), 301–307. https://doi.org/10.1097/00024665-200511000-00004 Howlader, N., AM, N., Krapcho, M., Miller, D., Bishop, K., SF, A., … EJ, F. (2015). SEER Cancer Statistics Review, 1975-2013. Based on November 2015 SEER Data Submission, Posted to the SEER Web Site, April 2016. Bethsada, MD: National Cancer Institute. Retrieved from http://seer.cancer.gov/csr/1975_2013/ Hsieh, H.-F., & Shannon, S. E. (2005). Three approaches to qualitative content analysis. Qualitative Health Research, 15(9), 1277–1288. https://doi.org/10.1177/1049732305276687 Hyslop, S., Dupuis, L. L., Baggott, C., Dix, D., Gibson, P., Kuczynski, S., … Sung, L. (2018). Validation of the proxy version of symptom screening in pediatrics tool in children receiving cancer treatments. Journal of Pain and Symptom Management, 56(1), 107–112. https://doi.org/10.1016/j.jpainsymman.2018.03.025 Jensen, J. (2014). Use of electronic diaries versus paper diaries in tracking alcohol and birth control behaviors in female college students. Alcoholism: Clinical and Experimental Research, 38, 73A. Retrieved from http://ovidsp.ovid.com/ovidweb.cgi?T=JS&PAGE=reference&D=emed12&NEWS= N&AN=71503374 115 Johnson, T. R., Zhang, J., Tang, Z., Johnson, C., & Turley, J. P. (2004). Assessing informatics students’ satisfaction with a web-based courseware system. In International Journal of Medical Informatics, 73, 181-187. https://doi.org/10.1016/j.ijmedinf.2003.12.006 Johnston, D. L., Hyslop, S., Tomlinson, D., Baggott, C., Gibson, P., Orsey, A., … Sung, L. (2018). Describing symptoms using the Symptom Screening in Pediatrics Tool in hospitalized children with cancer and hematopoietic stem cell transplant recipients. Cancer Medicine, 7(5), 1750-1755. https://doi.org/10.1002/cam4.1433 Kelly, M. D., Young, D. Y., Lane, N. M., & Shames, R. S. (2004). Validation of electronic versus paper subject diaries. Journal of Allergy and Clinical Immunology, 113(2). https://doi.org/http://dx.doi.org/10.1016/j.jaci.2004.01.654 Kestler, S. A., & Lobiondo-Wood, G. (2012). Review of symptom experiences in children and adolescents with cancer. Cancer Nursing, 35(2), E31-49. https://doi.org/10.1097/NCC.0b013e3182207a2a Kirkova, J., Aktas, A., Walsh, D., & Davis, M. P. (2011). Cancer Symptom clusters: clinical and research methodology. Journal of Palliative Medicine, 14(10), 11491166. https://doi.org/10.1089/jpm.2010.0507 Lewis, J. R. (1994). Sample sizes for usability studies: Additional considerations. Human Factors. https://doi.org/10.1177/001872089403600215 Linder, L. A. (2005). Measuring physical symptoms in children and adolescents with cancer. Cancer Nursing, 28(1), 16-26. https://doi.org/10.1097/00002820200501000-00003 Linder, L. A. (2008). Developmental diversity in symptom research involving children and adolescents with cancer. Journal of Pediatric Nursing, 23(4), 296-309. https://doi.org/10.1016/j.pedn.2007.10.003 Linder, L. A., & Wawrzynski, S. E. (2018). Staff Perceptions of symptoms, approaches to assessment, and challenges to assessment among children with cancer. Journal of Pediatric Oncology Nursing, 35(5), 332-341. https://doi.org/10.1177/1043454218767888 Linder, L., Bratton, H., Nguyen, A., Parker, K., & Wawrzynski, S. (2018). Symptoms and self-management strategies identified by children with cancer using drawand-tell Interviews. Oncology Nursing Forum, 45(3), 290–300. https://doi.org/10.1188/18.ONF.290-300 Lo, L. H., & Hayman, L. L. (1999). Parents associated with children in measuring acute and delayed nausea and vomiting. Nursing & Health Sciences, 1(3), 155–161. https://doi.org/10.1046/j.1442-2018.1999.00020.x 116 Magnusson, D., & Cairns, R. B. (2009). Developmental science: Toward a unified framework. In R. B. Cairns, G. H. Elder, & E. J. Costello (Eds.), Developmental science (pp. 7–30). Cambridge, UK: Cambridge University Press. https://doi.org/10.1017/CBO9780511571114.003 Marceau, L. D., Link, C. L., Smith, L. D., Carolan, S. J., & Jamison, R. N. (2010). Inclinic use of electronic pain diaries: Barriers of implementation among pain physicians. Journal of Pain and Symptom Management. https://doi.org/10.1016/j.jpainsymman.2009.12.021 McCann, L., Maguire, R., Miller, M., & Kearney, N. (2009). Patients’ perceptions and experiences of using a mobile phone-based advanced symptom management system (ASyMS) to monitor and manage chemotherapy related toxicity. European Journal of Cancer Care, 18(2), 156–164. https://doi.org/10.1111/j.1365-2354.2008.00938.x Merck & Co. (2011). iChemoDiary. Retrieved from http://itunes.apple.com/us/app/ichemodiary/id334253464?mt=8 Merriam Webster. (2016). Merriam Webster. Online Dictionary. https://doi.org/10.1016/j.bmcl.2010.12.080 Miller, E., Jacob, E., & Hockenberry, M. J. (2011). Nausea, pain, fatigue, and multiple symptoms in hospitalized children with cancer. Oncology Nursing Forum, 38(5), E382–E393. https://doi.org/10.1188/11.ONF.E382-E393 Mirkovic, J., Kaufman, D. R., & Ruland, C. M. (2014). Supporting cancer patients in illness management: Usability evaluation of a mobile app. Journal of Medical Internet Research, 2(3), e33. https://doi.org/10.2196/mhealth.3359 Mooney, K. H., Beck, S. L., Friedman, R. H., & Farzanfar, R. (2002). Telephone-linked care for cancer symptom monitoring: A pilot study. Cancer Practice, 10(3), 147– 154. https://doi.org/10.1046/j.1523-5394.2002.103006.x Mooney, K. H., Beck, S. L., Friedman, R. H., Farzanfar, R., & Wong, B. (2014). Automated monitoring of symptoms during ambulatory chemotherapy and oncology providers’ use of the information: A randomized controlled clinical trial. Support Care Cancer, 22(9), 2343–2350. https://doi.org/10.1007/ s00520-014-2216-1 Mooney, K. H., Beck, S. L., Wong, B., Dunson, W., Wujcik, D., Whisenant, M., & Donaldson, G. (2017). Automated home monitoring and management of patientreported symptoms during chemotherapy: Results of the symptom care at home RCT. Cancer Medicine, 6(3), 537–546. https://doi.org/10.1002/cam4.1002 Morren, M., Dulmen, V. S., Ouwerkerk, J., & Bensing, J. (2009). Compliance with momentary pain measurement using electronic diaries: A systematic review. European Journal of Pain, 13(4), 354-365. 117 https://doi.org/10.1016/j.ejpain.2008.05.010 Müller, S., Hemmi, M. H., Wilhelm, F. H., Barr, R. G., & Schneider, S. (2011). Parental report of infant sleep behavior by electronic versus paper-and-pencil diaries, and their relationship to actigraphic sleep measurement. Journal of Sleep Research, 20(4), 598–605. https://doi.org/10.1111/j.1365-2869.2011.00926.x Myrtveit, I., Stensrud, E., & Olsson, U. H. (2001). Analyzing data sets with missing data: An empirical evaluation of imputation methods and likelihood-based methods. IEEE Transactions on Software Engineering. https://doi.org/10.1109/32.965340 National Cancer Institute. (n.d.). NCI Dictionary of Cancer Terms. Retrieved from https://www.cancer.gov/publications/dictionaries/cancer-terms/def/cure National Cancer Institute. (2014). Age-Specific Ratesa and Counts for the Top 5 Cancer Sites by Single Year of Age at Diagnosis SEER Cancer Incidence 2010-2014. Retrieved from https://seer.cancer.gov/archive/csr/1975_2014/browse_csr.php?sectionSEL=28&pag eSEL=sect_28_table.13 National Cancer Institute. (2018). Cancer in Children and Adolescents. Retrieved from https://www.cancer.gov/types/childhood-cancers/child-adolescent-cancers-fact-sheet National Center on Caregiving. (2015). Caregiver Health | Family Caregiver Alliance. Retrieved from https://www.aarp.org/content/dam/aarp/ppi/2015/caregiving-in-theunited-states-2015-report-revised.pdf National Institute of Nursing Research. (2016). The NINR Strategic Plan: Advancing science, improving lives. Retrieved from https://www.ninr.nih.gov/sites/www.ninr.nih.gov/files/NINR_StratPlan2016_reduce d.pdf Nielsen, J. (2000). Why you only need to test with 5 users. Retrieved from https://www.nngroup.com/articles/why-you-only-need-to-test-with-5-users/ Noone, A.M., Howlader, N, Krapcho, M, Miller, D, Brest, A, Yu, M,… Cronin K.A. (Eds). (2017). SEER Cancer Statistics Review, 1975-2015. Bethesda, MD: National Cancer Institute. https://doi.org/10.1109/CEC.2012.6256410 Ohan, J. L., Glauert, R., Bell, M. F., Harrison, A., & Bayliss, D. M. (2016). Chronic illness and developmental vulnerability at school entry. Pediatrics, 137(5), e20152475. https://doi.org/10.1542/peds.2015-2475 Palermo, T. M., Valenzuela, D., & Stork, P. P. (2004). A randomized trial of electronic versus paper pain diaries in children: Impact on compliance, accuracy, and acceptability. Pain, 107(3), 213–219. https://doi.org/10.1016/j.pain.2003.10.005 118 Pandey, A., Hasan, S., Dubey, D., & Sarangi, S. (2013). Smartphone apps as a source of cancer information: Changing trends in health information-seeking behavior. Journal of Cancer Education. https://doi.org/10.1007/s13187-012-0446-9 PDQ Pediatric Treatment Editorial Board. (2018). Childhood Acute Lymphoblastic Leukemia Treatment (PDQ®): Patient Version. PDQ Cancer Information Summaries. Retrieved from http://www.ncbi.nlm.nih.gov/pubmed/26389385 Pinheiro, L. C., McFatrich, M., Lucas, N., Walker, J. S., Withycombe, J. S., Hinds, P. S., … Reeve, B. B. (2018). Child and adolescent self-report symptom measurement in pediatric oncology research: A systematic literature review. Quality of Life Research. https://doi.org/10.1007/s11136-017-1692-4 Pizzo, P.A., & Poplack, D.G. (2010). Principles and practice of pediatric oncology. Philadelphia, PA: Lippincott, Williams and Wilkins. Pöder, U., Ljungman, G., & Von Essen, L. (2010). Parents’ perceptions of their children’s cancer-related symptoms during treatment: A prospective, longitudinal study. Journal of Pain and Symptom Management, 40(5), 661–670. https://doi.org/10.1016/j.jpainsymman.2010.02.012 Questionnaire for User Interface Satisfaction. (n.d.). Retrieved from http://lap.umd.edu/quis/ Quigg, T. C., Mahajerin, A., Sullivan, P. D., Pradhan, K., & Bauer, N. S. (2013). Ages and Stages Questionnaires-3 developmental screening of infants and young children with cancer. Journal of Pediatric Oncology Nursing, 30(5), 235–241. https://doi.org/10.1177/1043454213493510 Rainie, L., & Perrin, A. (2017). 10 facts about smartphones as the iPhone turns 10. Retrieved from http://www.pewresearch.org/fact-tank/2017/06/28/10-facts-aboutsmartphones/ Riggins, T., Miller, N. C., Bauer, P. J., Georgieff, M. K., & Nelson, C. A. (2009). Electrophysiological indices of memory for temporal order in early childhood: Implications for the development of recollection. Developmental Science, 12(2), 209–219. https://doi.org/10.1111/j.1467-7687.2008.00757.x Rodgers, C. C., Krance, R., Street, R. L., & Hockenberry, M. J. (2014). Symptom prevalence and physiologic biomarkers among adolescents using a mobile phone intervention following hematopoietic stem cell transplantation. Oncology Nursing Forum, 41(3), 229–236. https://doi.org/10.1188/14.ONF.229-236 Rubin, L. H., Witkiewitz, K., Andre, J. S., & Reilly, S. (2007). Methods for handling missing data in the behavioral neurosciences: Don’t Throw the baby rat out with the bath water. Journal of Undergraduate Neuroscience Education, 5(2), A71-7. bath water. Journal of Undergraduate Neuroscience Education, 5(2), A71-7. 119 Ruland, C. M., Hamilton, G. A., & Schjodt-Osmo, B. (2009). The complexity of symptoms and problems experienced in children with cancer: A review of the literature. Journal of Pain and Symptom Management, 37(3), 403–418. https://doi.org/http://dx.doi.org/10.1016/j.jpainsymman.2008.03.009 Ruland, C. M., Starren, J., & Vatne, T. M. (2008). Participatory design with children in the development of a support system for patient-centered care in pediatric oncology. Journal of Biomedical Informatics, 41(4), 624-635. https://doi.org/10.1016/j.jbi.2007.10.004 Scolnik, D., Atkinson, V., Hadi, M., Caulfeild, J., & Young, N. L. (2003). Words used by children and their primary caregivers for private body parts and functions. Canadian Medical Association Journal, 169(12), 1275–1279. Sherifali, D., & Pinelli, J. (2007). Parent as proxy reporting. Journal of Family Nursing, 13(1), 83–98. https://doi.org/10.1177/1074840706297789 Slaughter, L., & Norman, K. L. Schniederman, B. (1995). Assessing users’ subjective satisfaction with the information system for youth services (ISYS). In Proceeds of Third Annual Mid-Atlantic Human Factors Conference (pp. 164–170). Blacksburg, VA. Smaldone, A., Tsimicalis, A., & Stone, P. (2011). Measuring resource utilization in patient-oriented comparative effectiveness research: A psychometric study of the resource utilization questionnaire. Research Theory Nursing Practice, 25(2), 80-106. Speyer, E., Herbinet, A., Vuillemin, A., Chastagner, P., & Briançon, S. (2009). Agreement between children with cancer and their parents in reporting the child’s health-related quality of life during a stay at the hospital and at home. Child: Care, Health and Development, 35(4), 489–495. https://doi.org/10.1111/j.13652214.2009.00972.x Stacey, D., Green, E., Ballantyne, B., Tarasuk, J., Skrutkowski, M., Carley, M., … Howell, D. (2016). Implementation of symptom protocols for nurses providing telephone-based cancer symptom management: A comparative case study. Worldviews on Evidence-Based Nursing, 13(6), 420-431. https://doi.org/10.1111/wvn.12166 Staggers, N., Kobus, D., & Brown, C. (2007). Nurses’ evaluations of a novel design for an electronic medication administration record. CIN - Computers Informatics Nursing, 25(2), 67–75. https://doi.org/10.1097/01.NCN.0000263981.38801.be Stone, A. A., Shiffman, S., Schwartz, J. E., Broderick, J. E., & Hufford, M. R. (2003). Patient compliance with paper and electronic diaries. Controlled Clinical Trials, 24(2), 182–199. https://doi.org/10.1016/S0197-2456(02)00320-3 Tomlinson, D., Dupuis, L. L., Gibson, P., Johnston, D. L., Portwine, C., Baggott, C., … 120 Sung, L. (2014). Initial development of the Symptom Screening in Pediatrics Tool (SSPedi). Supportive Care in Cancer, 22(1), 71–75. https://doi.org/10.1007/s00520013-1945-x Tomlinson, D., Tigelaar, L., Hyslop, S., Lazor, T., Dupuis, L. L., Griener, K., … Sung, L. (2017). Self-report of symptoms in children with cancer younger than 8 years of age: A systematic review. Supportive Care in Cancer, 25(8), 2663–2670. https://doi.org/10.1007/s00520-017-3740-6 Tomlinson, D., De Mol Van Otterloo, S., O’Sullivan, C., Gibson, P., Johnston, D. L., Portwine, C., … Sung, L. (2016). Methodological issues identified during cognitive interviews in the development of a pediatric cancer symptom screening tool. Psycho-Oncology, 25(3), 349-353. https://doi.org/10.1002/pon.3821 Torres, V., Nunes, M. D. R., Silva-Rodrigues, F. M., Bravo, L., Adlard, K., Secola, R., … Jacob, E. (2019). Frequency, severity, and distress associated with physical and psychosocial symptoms at home in children and adolescents with cancer. Journal of Pediatric Health Care,34(4), 404-414. https://doi.org/10.1016/j.pedhc.2018.11.007 Tsimicalis, A., Rennick, J., Stinson, J., May, S. L., Louli, J., Choquette, A., … Ruland, C. (2018). Usability testing of an interactive communication tool to help children express their cancer symptoms. Journal of Pediatric Oncology Nursing, 35(5), 320331. https://doi.org/10.1177/1043454218777728 usability.gov. (n.d.). Usability Evaluation Basics. Retrieved from https://www.usability.gov/what-and-why/usability-evaluation.html Vatne, T. M., Slaugther, L., & Ruland, C. M. (2010). How children with cancer communicate and think about symptoms. Journal of Pediatric Oncology Nursing, 27(1), 24–32. https://doi.org/10.1177/1043454209349358 Venkatesh, V., & Davis, F. D. (2000). A Theoretical extension of the technology acceptance model: Four longitudinal field studies. Management Science, 46(2), 186– 204. https://doi.org/10.1287/mnsc.46.2.186.11926 Verto Index Health and Fitness Apps. (2017). Retrieved from http://www.vertoanalytics.com/verto-index-health-fitness-apps/ Virzi, R. A. (1992). Refining the test phase of usability evaluation: How many subjects is enough? Human Factors, 34(4), 457-468. https://doi.org/10.1177/001872089203400407 Walker, A. J., Gedaly-Duff, V., Miaskowski, C., & Nail, L. (2010). Differences in symptom occurrence, frequency, intensity, and distress in adolescents prior to and one week after the administration of chemotherapy. Journal of Pediatric Oncology Nursing, 27(5), 259–265. https://doi.org/10.1177/1043454210365150 Nursing, 27(5), 259–265. https://doi.org/10.1177/1043454210365150 121 Whalen, C. K., Henker, B., Jamner, L. D., Ishikawa, S. S., Floro, J. N., Swindle, R., … Johnston, J. A. (2006). Toward mapping daily challenges of living with ADHD: Maternal and child perspectives using electronic diaries. Journal of Abnormal Child Psychology, 34(1), 115–130. https://doi.org/10.1007/s10802-005-9008-5 Williams, P. D., Schmideskamp, J., Ridder, E. L., & Williams, A. R. (2006). Symptom monitoring and dependent care during cancer treatment in children: Pilot study. Cancer Nursing, 29(3), 188–197. https://doi.org/10.1097/00002820-20060500000004 Williams, P. D., Williams, A. R., Kelly, K. P., Dobos, C., Gieseking, A., Connor, R., … Del Favero, D. (2012). A symptom checklist for children with cancer: The therapyrelated symptom checklist-children. Cancer Nursing, 35(2), 89–98. https://doi.org/10.1097/NCC.0b013e31821a51f6 Woodgate, R., & McClement, S. (1998). Symptom distress in children with cancer: The need to adopt a meaning-centered approach. Journal of Pediatric Oncology Nursing : Official Journal of the Association of Pediatric Oncology Nurses, 15(1), 3–12. https://doi.org/10.1177/104345429801500102 Woodgate, R. L. (2008). Feeling states: A new approach to understanding how children and adolescents with cancer experience symptoms. Cancer Nursing, 31(3), 229–238. https://doi.org/10.1097/01.NCC.0000305731.95839.ca Woodgate, R. L., Degner, L. F., & Yanofsky, R. (2003). A different perspective to approaching cancer symptoms in children. Journal of Pain and Symptom Management, 26(3), 800–817. https://doi.org/10.1016/S0885-3924(03)00285-9 Yeh, C.-H., Chang, C.-W., & Chang, P.-C. (2005). Evaluating quality of life in children with cancer using children’s self-reports and parent-proxy reports. Nursing Research, 54(5), 354–362. https://doi.org/10.1097/00006199-200509000-00010 Zhukovsky, D. S., Rozmus, C. L., Robert, R. S., Bruera, E., Wells, R. J., Chisholm, G. B., … Cohen, M. Z. (2015). Symptom profiles in children with advanced cancer: Patient, family caregiver, and oncologist ratings. Cancer, 121(22), 4080-4087. https://doi.org/10.1002/cncr.29597 Zupanec, S., Jones, H., & Stremler, R. (2010). Sleep habits and fatigue of children receiving maintenance chemotherapy for all and their parents. Journal of Pediatric Oncology Nursing, 27(4), 217–228. https://doi.org/10.1177/1043454209358890