Postoperative Horner Syndrome After Lung Transplantation

Horner syndrome arises from a disruption along the oculosympathetic efferent chain and can be caused by a variety of pathological and iatrogenic etiologies. We present 3 cases of postoperative Horner syndrome after bilateral lung transplantation.

Original Contribution Section Editors: Clare Fraser, MD Susan Mollan, MD Postoperative Horner Syndrome After Lung Transplantation Jonathan A. Go, BBA, Aroucha Vickers, DO, Thomas S. Kaleekal, MD, Hilary A. Beaver, MD, Andrew G. Lee, MD Background: Horner syndrome arises from a disruption along the oculosympathetic efferent chain and can be caused by a variety of pathological and iatrogenic etiologies. We present 3 cases of postoperative Horner syndrome after bilateral lung transplantation. Methods: The electronic health records of 3 patients with iatrogenic Horner syndrome after lung transplantation were examined, including notes from each patient’s medical history, operative and postoperative records, and ophthalmology consultation results. A literature review was performed. Results: All 3 of our patients displayed anisocoria and ptosis, symptoms consistent with Horner syndrome, and the patients from Cases 1 and 2 showed reversal of anisocoria after an application of topical apraclonidine. Conclusions: Ophthalmologists should be aware of the risk of Horner syndrome after lung transplantation. Journal of Neuro-Ophthalmology 2020;40:504–506 doi: 10.1097/WNO.0000000000000848 © 2019 by North American Neuro-Ophthalmology Society H orner syndrome (Horner syndrome) may arise from a lesion anywhere along the oculosympathetic efferent chain. Although Horner syndrome often does not have an identifiable cause, the literature reports that 35%–60% of Baylor College of Medicine (JAG, AGL), Houston, Texas; Department of Ophthalmology (AV, HAB, AGL), Blanton Eye Institute, Houston Methodist Hospital, Houston, Texas; Division of Pulmonary, Critical Care and Transplant Medicine (TSK), Houston Methodist Hospital, Houston, Texas; Department of Ophthalmology (HAB, AGL), University of Texas Medical Branch, Galveston, Texas; Department of Ophthalmology (AGL), University of Texas MD Anderson Cancer Center, Houston, Texas; Department of Ophthalmology (AGL), Texas A and M College of Medicine, Bryan, Texas; Departments of Ophthalmology, Neurology, and Neurosurgery (HAB, AGL), Weill Cornell Medicine, New York, New York; and Department of Ophthalmology (AGL), The University of Iowa Hospitals and Clinics, Iowa City, Iowa. The authors report no conflicts of interest. Address correspondence to Andrew G. Lee, MD, Blanton Eye Institute, Houston Methodist Hospital, 6560 Fannin Street, Suite 450, Houston, TX 77030; E-mail: [e-mail] 504 Horner syndrome etiologies are associated with neoplasms, 4%–13% arise from trauma and birth-related events, and 10%–18.5% are iatrogenic injuries (1). We present 3 cases of postoperative Horner syndrome after bilateral lung transplantation. To the best of our knowledge, this is the first series of Horner syndrome due to lung transplantation in the English ophthalmologic literature. METHODS The electronic health records of 3 former patients with iatrogenic Horner syndrome after lung transplantation were examined, including notes from each patient’s medical history, operative and postoperative records, and ophthalmology consultation results. A literature review was performed using the search terms “Horner syndrome,” “Horner’s syndrome,” “iatrogenic,” “anisocoria,” and “lung transplant.” Institutional review board approval was obtained for all study protocols with waiver of informed consent. RESULTS Case 1 A 60-year-old woman presented with a 2-day history of new onset mild ptosis of the right upper eyelid. The medical history was significant for coronary artery disease, diabetes mellitus, essential hypertension, hyperglycemia, and obesity. The patient’s surgical history was significant for bronchoscopy, cardiac catheterization, and bilateral breast cyst excision. She was an ex-smoker with a 40-pack-year history. She denied excessive alcohol and illicit drug use. Her father had diabetes; her mother had hypertension, and her sister had lung cancer. A review of systems was unremarkable. One month before presentation, the patient underwent a pulmonary allograft transplantation without cardiopulmonary bypass due to end-stage lung disease, secondary to chronic obstructive pulmonary disease (COPD). She was induced with general double-lumen endotracheal Go et al: J Neuro-Ophthalmol 2020; 40: 504-506 Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited. Original Contribution anesthesia, and a bilateral anterolateral thoracotomy incision followed. Hilar dissection was completed, followed by ligation and division of the right pulmonary vessels and bronchus. The right lung was resected, and the donor lung was prepared. Anastomosis and deairing, then ventilation, was performed. The left lung was also resected and replaced with the same procedure. Bilateral 28 French chest tubes were placed at anterior and posterior positions. No complications were noted. Examination revealed best-corrected visual acuities of J1+ at near. Visual fields were full in both eyes. The pupils measured 4 mm on the right and 2 mm on the left in the dark and measured 2.5 mm on the right and 1.5 mm on the left in the light. No afferent pupillary defect was present. Extraocular motility examination was full for both eyes. Intraocular pressure measurements were normal. External examination revealed a 1–2 mm of right ptosis. Slit-lamp examination revealed 2+ nuclear sclerosis of the lens bilaterally. Fundus examination was unremarkable. Off-label application of topical apraclonidine reversed the anisocoria, consistent with a right Horner syndrome. Case 2 A 60-year-old woman presented with a 3-day history of new onset, mild right ptosis. Her medical history was significant for bronchitis, COPD, hypothyroidism, and osteoporosis. She was an ex-smoker with a 39 pack-year history. A bilateral pulmonary allograft transplantation without cardiopulmonary bypass was performed 1 month before presentation due to end-stage lung disease and COPD. The patient was induced with general doublelumen endotracheal anesthesia, and a bilateral anterolateral thoracotomy incision and hilar dissection followed. Adhesions were very dense, requiring 2 additional hours of dissection. The right pulmonary vessels and bronchus were ligated and divided, followed by right lung resection. The donor lung was prepared. Anastomoses were completed, along with deairing. The left lung transplant was also completed with the same methodology. Bilateral 28 French chest tubes were placed anteriorly and posteriorly. Five days after surgery, a right-sided pneumothorax was noted, with an atypical air gap of 2.5 cm. The patient’s water-seal chest tube was switched to a suction chest tube. Two days later, a chest radiograph showed resolution of the right pneumothorax; however, a new, small, loculated left basal pneumothorax was noted. At discharge, the small left, apical pneumothorax was reported as stable. Examination revealed best-corrected visual acuities of 20/50-2 bilaterally. Color plates were correctly identified (14/14 bilaterally). Visual fields to confrontation were full. The pupils measured 4 mm on the right and 2 mm on the left in the light; In the dark, pupil sizes were 5 mm on the right and 2 mm on the left. No afferent pupillary defect was Go et al: J Neuro-Ophthalmol 2020; 40: 504-506 present. The extraocular motility and intraocular pressure examinations were normal. External examination showed a 2-mm right ptosis. Slit-lamp examination showed nuclear sclerotic cataracts consistent with the patient’s 20/50 visual acuities. Fundus examination was normal bilaterally. Offlabel application of topical apraclonidine reversed the anisocoria, consistent with a right Horner syndrome. Case 3 A 60-year-old man presented with a 1-day history of new onset left ptosis. Her medical history was significant for Stage IV systemic sarcoidosis, unspecified arrhythmia, borderline diabetes mellitus, coronary artery disease, hypertension, hypercholesteremia, hypothyroidism, ischemic cardiomyopathy, pulmonary fibrosis, and obesity. The patient had experienced a cardiac arrest with ventricular fibrillation 1 year earlier and was cardioverted into a viable rhythm with no recurrence. Echocardiogram showed significantly depressed left ventricle function, and an automated implantable cardioverter defibrillator (AICD) was implanted. An arch aortogram showed a major clot in the aortic arch, near the origin of the left subclavian artery, so anticoagulants were prescribed. His surgical history was significant for laryngoscopy 1 week earlier, confirming left true vocal cord paralysis. He also completed cardiac electrophysiology, cardiac catheterization, and a coronary angioplasty with stent placement. He denied excessive alcohol and illicit drug use. He was an ex-smoker with a 19-pack-year history. His father had Alzheimer disease and heart disease, his mother had unspecified lymphoma, and his brother had hypertension. One week before presentation, the patient underwent an orthotopic heart and bilateral lung transplantation for systolic heart and respiratory failure stemming from sarcoidosis. The patient was induced with general endotracheal anesthesia, followed by a midline sternotomy incision. The heart and lungs were resected using a standard protocol, with no damage to the phrenic nerves. The trachea was resected above the carina, along with all level 7 nodes and greater. Anastomosis was completed. Although pacemaker wires were placed, the patient established his own heart rhythm. Hemostasis was verified, and chest tubes were placed at an unknown location. An AICD was resected through a left subclavicular incision and Bovie electrocauterization. Venous entry points were sutured. No complications were noted. Examination revealed best-corrected visual acuities of 20/30 bilaterally. Visual fields were full. The right pupil measured 4.5 mm in the dark and 3.5 mm in the light, whereas the left pupil measured 3.5 mm and 2.5 mm, respectively. No afferent pupillary defect was noted. Motility was full. Pressure was normal bilaterally by palpation. A 1-mm left ptosis was noted. Slit-lamp examination revealed nuclear cataracts consistent with his 505 Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited. Original Contribution visual acuities of 20/30. Fundus examination was normal bilaterally. A diagnosis of left Horner syndrome was made clinically. Harlequin syndrome, a sympathetic disruption similar to Horner syndrome, has been reported after lung transplantation (14). Ophthalmologists should be aware of the risk of Horner syndrome after lung transplantation. CONCLUSION The oculosympathetic pathway is a three-neuron pathway that travels caudally from the hypothalamus (first-order neuron) to the spinal cord (C8-T2), then rostrally in the cervical sympathetic chain (second-order neuron) to its synapse in the superior cervical ganglion (third-order neuron), then intracranially to the internal carotid artery to reach cranial nerve V1 in the cavernous sinus, and finally to the eye (2). A disruption along any point in this pathway can result in Horner syndrome; up to 84% of iatrogenic Horner syndrome cases involve the second-order neuron (1). Iatrogenic Horner syndrome has been reported due to a variety of causes including thyroid surgery (3), pneumothorax (4), lumbar epidural anesthesia (5), sympathectomy (6), tube thoracostomy (7), thoracotomy for cystotomy (8), chest tube placement (9), cervical nerve root block (10), and carotid endarterectomy (11). In a review of 151 cases of iatrogenic Horner syndrome, peridural obstetric anesthesia, cervical sympathetic chain surgery, chest tube insertion, and tonsillectomy were cited as the most common etiologies. Although 100% of the chest tube insertions and tonsillectomies reported full or partial recovery, only 56% of cervical sympathetic chain surgeries and 5% of peridural obstetric anesthesia cases achieved full or partial recovery (1). In addition, a study of 933 thoracic surgery patients revealed 12 cases of iatrogenic Horner syndrome: 5 cases from chest tube insertion pressure injuries, 2 cases from surgical complications, and 5 cases from thoracic trauma. Only chest tube placement cases showed clinical improvement over time, after the tubes were repositioned (12). In our 3 Horner syndrome cases, it is not possible to completely rule out chest tube placement as the etiology. However, in our cases, a lung transplant procedure is the more likely etiology because our patients presented 1 week to 1 month after surgery. Most chest tube-related Horner syndrome cases in the literature had improvement after chest tube removal or repositioning. Because our patients presented past the chest tube drainage recovery stage, chest tubes are less likely to be the underlying cause. All 3 of our patients displayed anisocoria and ptosis, symptoms consistent with Horner syndrome, and the patients from Cases 1 and 2 showed reversal of anisocoria after an off-label application of topical apraclonidine (13). 506 STATEMENT OF AUTHORSHIP Category 1: a) Conception and design: J. A. Go, A. Vickers, T. S. Kaleekal, H. A. Beaver, and A. G. Lee; b) Acquisition of data: J. A. Go, A. Vickers, T. S. Kaleekal, H. A. Beaver, and A. G. Lee; c) Analysis and interpretation of data: J. A. Go, A. Vickers, T. S. Kaleekal, H. A. Beaver, and A. G. Lee. Category 2: a) Drafting the manuscript: J. A. Go, A. Vickers, and A. G. Lee; b) Revising it for intellectual content: J. A. Go, A. Vickers, T. S. Kaleekal, H. A. Beaver, and A. G. Lee. Category 3: a) Final approval of the completed manuscript: J. A. Go, A. Vickers, T. S. Kaleekal, H. A. Beaver, and A. G. Lee. REFERENCES 1. Knyazer B, Smolar J, Lazar I, Rosenberg E, Tsumi E, Lifshitz T, Levy J. Iatrogenic horner syndrome: etiology, diagnosis and outcomes. IMAJ. 2017;19:34–38. 2. Martin TJ. Horner syndrome: a clinical review. ACS Chem Neurosci. 2018;9:177–186. 3. Hu X, Zhang X, Gan H, Yu D, Sun W, Shi Z. Horner syndrome as a postoperative complication after minimally invasive videoassisted thyroidectomy: a case report. Medicine. 2017;96:e8888. 4. Mutalib M, Vandervelde C, Varghese A, Sallomi DF, de Silva P, Hickman Casey JM, Howlett DC. Horner’s syndrome secondary to asymptomatic pneumothorax in an adolescent. Eur J Pediatr. 2007;166:507–508. 5. Biousse V, Guevara RA, Newman NJ. Transient Horner’s syndrome after lumbar epidural anesthesia. Neurology. 1998;51:1473–1475. 6. Singh B, Moodley J, Allopi L, Cassimjee HM. Horner syndrome after sympathectomy in the thoracoscopic era. Surg Laparosc Endosc Percutan Tech. 2006;16:222–225. 7. Baird R, Al-Balushi Z, Wackett J, Bouchard S. Iatrogenic Horner syndrome after tube thoracostomy. J Pediatr Surg. 2009;44:2012–2014. _ Karaca M, Yazici Ü, Tanir G. A case of Horner’s 8. Bayhan GI, syndrome after the surgical treatment of pulmonary hydatid cyst. Turkiye Parazitol Derg. 2010;34:196–199. 9. Levy M, Newman-Toker D. Reversible chest tube horner syndrome. J Neuroophthalmol. 2008;28:212–213. 10. Kaplowitz K, Lee AG. Horner syndrome following a selective cervical nerve root block. J Neuroophthalmol. 2011;31:54–55. 11. Perry C, James D, Wixon C, Mills J, Ericksen C. Horner’s syndrome after carotid endarterectomy—a case report. Vasc Surg. 2001;35:325–327. 12. Kaya SO, Lima ST, Bir LS, Yuncu G, Erbay HR, Unsal S. Horner’s syndrome as a complication in thoracic surgical practice. Eur J Cardiothorac Surg. 2003;24:1025–1028. 13. Morales J, Brown SM, Abdul-Rahim AS, Crosson CE. Ocular effects of apraclonidine in horner syndrome. Arch Ophthalmol. 2000;118:951–954. 14. Boling B, Key C, Wainscott J, Rebel A. Harlequin syndrome as a complication of epidural anesthesia. Crit Care Nurse. 2014;34:57–61. Go et al: J Neuro-Ophthalmol 2020; 40: 504-506 Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited.