Odontogenic Sinusitis Resulting in Abscess Formation Within the Optic Chiasm and Tract: Case Report and Review

A 33-year-old immunocompetent man developed rapid visual loss and a third nerve palsy secondary to acute rhinosinusitis and intracranial abscess formation. Despite endoscopic drainage of the ethmoid and sphenoid sinuses and empiric broad-spectrum antibiotics, the patient experienced progressive visual and neurological decline and ultimately required craniotomy for drainage of an optic apparatus abscess. Although odontogenic sinusitis rarely results in abscess formation of the visual pathways, early recognition and immediate treatment is imperative to decrease the risk of profound and permanent visual impairment.

Original Contribution Odontogenic Sinusitis Resulting in Abscess Formation Within the Optic Chiasm and Tract: Case Report and Review George M. Ghobrial, MD, Mary L. Pisculli, MD, James J. Evans, MD, Jurij R. Bilyk, MD, Christopher J. Farrell, MD Abstract: A 33-year-old immunocompetent man developed rapid visual loss and a third nerve palsy secondary to acute rhinosinusitis and intracranial abscess formation. Despite endoscopic drainage of the ethmoid and sphenoid sinuses and empiric broad-spectrum antibiotics, the patient experienced progressive visual and neurological decline and ultimately required craniotomy for drainage of an optic apparatus abscess. Although odontogenic sinusitis rarely results in abscess formation of the visual pathways, early recognition and immediate treatment is imperative to decrease the risk of profound and permanent visual impairment. Journal of Neuro-Ophthalmology 2016;36:393-398 doi: 10.1097/WNO.0000000000000430 © 2016 by North American Neuro-Ophthalmology Society S inusitis of odontogenic origin accounts for approximately 10% of cases of maxillary sinusitis and is typically related to polymicrobial bacterial infection (1). Intracranial extension of sinusitis rarely may occur due either to contiguous spread or retrograde extension through valveless diploic veins, potentially causing epidural abscess, subdural empyema, cavernous sinus thrombosis, or meningitis (2). A 33-year-old immunocompetent man experienced rapid visual decline secondary to acute rhinosinusitis and abscess formation within Department of Neurological Surgery (GMG, MLP, JJE, CJF), Thomas Jefferson University Hospital, Philadelphia, Pennsylvania; and Skull Base Division (JRB), Neuro-Ophthalmology Service, Wills Eye Hospital, Philadelphia, Pennsylvania. The authors report no conflicts of interest. Supplemental digital content is available for this article. Direct URL citations appear in the printed text and are provided in the full text and PDF versions of this article on the journal's Web site (www.jneuro-ophthalmology.com). Address correspondence to Christopher J. Farrell, MD, Division of Neuro-Oncology, Department of Neurological Surgery, Thomas Jefferson University Hospital, Philadelphia, PA 19107; E-mail: [e-mail] Ghobrial et al: J Neuro-Ophthalmol 2016; 36: 393-398 the intracranial anterior visual pathway. Despite endoscopic drainage of the ethmoid and sphenoid sinuses and empiric broad-spectrum antibiotics, the patient experienced progressive visual and neurological decline and ultimately required craniotomy and drainage of his intracranial abscess. CASE REPORT A 33-year-old man with an unremarkable medical history was evaluated in an emergency department complaining of left facial pain and purulent nasal drainage. He was treated empirically with amoxicillin/clavulanic acid and corticosteroids for a diagnosis of acute sinusitis. Despite oral antibiotic therapy, 5 days later he noted onset of blurred vision in his right eye and the following morning awoke completely blind and ophthalmoplegic in that eye. He was treated with intravenous antibiotics and transferred the next day to our institution. On initial examination, the patient's vision was no light perception, right eye, and 20/25, left eye. Findings included a mid-dilated, right amaurotic pupil, right ptosis, and eye movements consistent with a right third nerve palsy (Fig. 1). The anterior and posterior segments of each eye appeared normal. Confrontation visual field testing was full in the left eye. The remainder of his neurologic and physical examinations were normal, with the exception of poor dentition. The patient's temperature was 98.9°F. Laboratory evaluation was notable for an elevated peripheral white blood cell count of 16,900 mL with 92% neutrophils. Computed tomography (CT) of the sinuses revealed severe periodontal and dental disease with multiple carious and periapical lucencies. Left maxillary sinusitis as well as bilateral ethmoid and sphenoid sinusitis were observed but there was no disruption of the bony skull base or orbits (Fig. 2). Magnetic resonance imaging (MRI) demonstrated an 393 Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited. Original Contribution FIG. 1. At presentation, the patient had right upper eyelid ptosis as well as limitation of elevation, depression, and adduction of the right eye. epidural abscess along the medial sphenoid wing and right orbital roof. There was enhancement of the right intracanalicular optic nerve, orbital apex, prechiasmatic segment of the right optic nerve, and portions of the optic chiasm (Fig. 3). Abnormal marrow signal involving the medial and superior aspect of the right sphenoid sinus was consistent with osteomyelitis. There were no signs of cavernous sinus involvement noted. Intravenous antibiotic therapy with vancomycin, ceftriaxone, and metronidazole was initiated empirically and the patient was taken emergently to the operating room for endoscopic sinus surgery and drainage of the sinus disease. Purulent material without obvious evidence of fungal disease was encountered throughout the paranasal sinuses. No direct source of intraorbital or intracranial extension was detected. The surgical pathology was consistent with acute FIG. 2. Ethmoid, maxillary, and sphenoid sinusitis are demonstrated on axial (A, B) and coronal (C, D) computed tomography. 394 Ghobrial et al: J Neuro-Ophthalmol 2016; 36: 393-398 Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited. Original Contribution FIG. 3. Initial postcontrast, fat-suppressed T1 MRI. A. Axial image shows enhancement of the right orbital apex extending intracranially. B. Coronal view reveals enhancement of right intracanicular optic nerve (arrow) and dura (arrowheads). C. There is ring enhancement of the prechiasmal right optic nerve (arrow). D. The anterior optic chiasm also enhances (arrows). sinusitis with absence of fungal elements and Gram stain revealed gram-variable rods and gram-positive cocci. After extensive discussion among the neurosurgery, infectious disease, and ophthalmology consultants, and due to the preserved vision in the left eye, the intracranial infection was initially managed solely with antibiotic therapy with serial ophthalmologic examination every 4 hours. A surveillance MRI performed 1-day after admission showed decrease in the sinus disease and stability of the epidural and optic apparatus abscesses. The third nerve palsy markedly improved on the first postoperative day. On the third day following admission, ophthalmologic examination revealed subtle temporal field loss in the left eye, although central vision was preserved. Weakness of the left arm also was noted. Brain MRI showed further expansion of the right optic nerve, optic chiasm, and optic tract, consistent with progressive infection (Fig. 4). Additionally, new abnormal T2 hyperintensity involving the right temporal lobe and cerebral peduncle was observed. The patient was taken emergently to the operating room for a pterional craniotomy and drainage of the epidural and optic apparatus infection. At the time of surgery, a swollen, hyperemic right optic nerve was observed with the abnormal swelling extending to the mid-aspect of the optic chiasm and posteriorly along the right optic tract. The lateral aspects of the optic nerve, chiasm, and tract were incised sharply with an arachnoid knife and suppurative Ghobrial et al: J Neuro-Ophthalmol 2016; 36: 393-398 material drained from within the neural structures (Fig. 5; also See Supplemental Digital Content, Video, http://links. lww.com/WNO/A210). A small epidural abscess in the region of the anterior clinoid and orbital roof was also drained. Following craniotomy, the patient experienced resolution of his left hemiparesis. He had a dense temporal visual defect on the left and 20/30 vision with eccentric fixation. No visual recovery was observed in either eye during hospitalization. Bacterial cultures from the sinuses grew methicillin-sensitive Staphylococcus aureus, Staphylococcus lugdunensis, Corynebacterium accolens, and Prevotella and Actinomyces species. Despite the purulence observed at the time of craniotomy, cultures taken were sterile. The patient subsequently was treated with surgical extraction of 12 carious teeth and a 6-week course of intravenous penicillin, ceftriaxone, and metronidazole for his polymicrobial infection. He was subsequently transitioned to oral amoxicillin for prolonged treatment of osteomyelitis and Actinomycosis infection and has remained clinically stable without evidence of recurrent infection over 8 months of follow-up. DISCUSSION Intraorbital and intracranial complications of acute sinusitis are rare, with the published literature consisting primarily of case reports and small case series (3,4). Although the 395 Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited. Original Contribution FIG. 4. Follow-up postcontrast, fat-suppressed T1 magnetic resonance imaging. A and B. Axial scans show increased enhancement intracranially with evidence of abscess formation along the right optic tract (arrow). Coronal images reveal abscess formation (arrows) involving the right prechiasmal optic nerve (C) and right optic tract (D). mortality from sinusitis-related intracranial infections has been reduced to 2%-7% with advances in neuroimaging and antibiotic therapy, neurologic morbidity remains common (4). Complicated sinusitis has been most frequently reported in immunocompetent, young male patients and this predisposition has been attributed to their highly vascularized diploic venous system allowing for indirect intracranial extension through propagation of septic thrombophlebitis (5). However, in our patient, the spread of infection was likely due to osteomyelitis within the posterior and medial walls of the sphenoid sinus (6). Thin walls of bone anteriorly and posteriorly separate the sphenoid sinus from the orbital apex and intracranial cavity, respectively (7). Direct intraorbital spread of infection from sphenoethmoidal sinusitis most commonly results from disruption of the thin lamina papyracea separating the ethmoid sinuses from the orbit. Articulating with the frontal bone above and the maxillary bone below, the lamina papyracea is a thin boundary of bone that separates the medial orbit 396 from the ethmoid sinus. Although no imaging or intraoperative violation of the medial orbital wall was noted in our patient, the lamina papyracea contains multiple small, natural dehiscences that may allow for intraorbital spread of ethmoidal infection (6). The superior aspect of the posterior wall of the sphenoid sinus borders the medial wall of the optic canal and osteomyelitis of the sphenoid bone may extend through the optic canal or into the anterior clinoid process adjacent to the canalicular segment of the optic nerve. Visual loss related to acute sphenoethmoidal sinusitis most frequently results from orbital cellulitis characterized by proptosis, chemosis, and painful opthalmoplegia, often with redness and swelling of the periorbital structures. Depending on the segment of the optic nerve affected, visual loss may occur as a component of the orbital apex syndrome. This "posterior orbital cellulitis" results in impairment of the optic nerve and structures within the superior orbital fissure without accompanying orbital inflammatory signs (8). Damage to the optic nerve may result from Ghobrial et al: J Neuro-Ophthalmol 2016; 36: 393-398 Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited. Original Contribution to date, which included 70 cases of CNS Actinomyces infection, 21 cases (32%) originated contiguously from the ear, sinuses, or cervicofacial region. Most of the cervicofacial infections were considered to be of odontogenic origin (16). Despite aggressive surgical drainage in the majority of cases and prolonged antibiotic treatment (mean duration of 5 months), the overall mortality was 28%, with 54% of patients experiencing neurologic morbidity (14). Patients with delayed diagnosis and initiation of treatment experienced poorer outcomes. In the setting of Actinomyces CNS infection, the optimal duration of antibiotic therapy remains uncertain but there is general consensus for prolonged treatment for up to 12 months with penicillin antibiotics (16). FIG. 5. View at craniotomy, right pterional approach. The right optic nerve is swollen and inflamed with drainage of purulent material from the neurotomy defect: (A) optic nerve; (B) sphenoid bone. congestion and pressure within the orbital apex related to the inflammatory infiltrate with secondary vascular occlusion (9). Intraneural dissemination of infection through the optic nerve to the optic chiasm and optic tract has very rarely been reported (4,10). Odontogenic sinusitis is more likely to be polymicrobial with anaerobic organisms than non-odontogenic infection, and intraorbital and intracranial infectious complications can have devastating effects, even when timely appropriate therapy is initiated. Empiric antibiotic therapy should target aerobic gram-positive and anaerobic organisms. Abscessed teeth should be extracted and the paranasal sinuses drained for source control. If there is radiographic evidence of an intraorbital abscess, drainage should be performed in the setting of visual impairment (9). With regard to the optimal treatment of an intracranial abscess within the optic apparatus itself, there is no information in the literature to guide therapy. Multiple reports of successful treatment of small intracranial abscesses within the brain parenchyma with antibiotic therapy alone have been described (10,11). Additionally, reversal of vision loss in the setting of odontogenic orbital infections has been reported with early initiation of antibiotic therapy at the time of visual decline, although the prognosis for vision recovery is usually poor if vision loss is severe on presentation (3,12,13). An additional consideration in our patient is the appropriate management of intracranial Actinomyces infection. Actinomyces are non-spore forming, branching anaerobic bacteria that are normal flora of the oral cavity. Although Actinomyces species are generally of low pathogenicity, central nervous system (CNS) infection does not require an immunocompromised host and typically occurs in patients having prior neurosurgery, head trauma, or otorhinolaryngeal infections (14,15). Within the largest review Ghobrial et al: J Neuro-Ophthalmol 2016; 36: 393-398 STATEMENT OF AUTHORSHIP Category 1: a. Conception and design: G. M. Ghobrial, C. J. Farrell, J. J. Evans, and J. R. Bilyk; b. Acquisition of data: G. M. Ghobrial, C. J. Farrell, J. J. Evans, and J. R. Bilyk; c. Analysis and interpretation of data: G. M. Ghobrial, C. J. Farrell, J. J. Evans, and J. R. Bilyk. Category 2: a. Drafting the manuscript: G. M. Ghobrial, C. J. Farrell, J. J. Evans, and J. R. Bilyk; b. Revising it for intellectual content: G. M. Ghobrial, C. J. Farrell, J. J. Evans, and J. R. Bilyk. Category 3: a. Final approval of the completed manuscript: G. M. Ghobrial, C. J. Farrell, J. J. Evans, and J. R. Bilyk. REFERENCES 1. Mehra P, Jeong D. Maxillary sinusitis of odontogenic origin. Curr Infect Dis Rep. 2008;10:205-210. 2. Younis RT, Lazar RH, Anand VK. Intracranial complications of sinusitis: a 15-year review of 39 cases. Ear Nose Throat J. 2002;81:636-638. 640-642. 644. 3. Gold RS, Sager E. Pansinusitis, orbital cellulitis, and blindness as sequelae of delayed treatment of dental abscess. J Oral Surg. 1974;32:40-43. 4. Osborn MK, Steinberg JP. Subdural empyema and other suppurative complications of paranasal sinusitis. Lancet Infect Dis. 2007;7:62-67. 5. DelGaudio JM, Evans SH, Sobol SE, Parikh SL. Intracranial complications of sinusitis: what is the role of endoscopic sinus surgery in the acute setting. Am J Otolaryngol. 2010;31:25-28. 6. Jain A, Rubin PA. Orbital cellulitis in children. Int Ophthalmol Clin. 2001;41:71-86. 7. Budu V, Mogoanta CA, Fanuta B, Bulescu I. The anatomical relations of the sphenoid sinus and their implications in sphenoid endoscopic surgery. Rom J Morphol Embryol. 2013;54:13-16. 8. Allan K, Atkinson H, Agada F. Posterior orbital cellulitis: case report and literature review. J Laryngol Otol. 2013;127:1148- 1151. 9. Youssef OH, Stefanyszyn MA, Bilyk JR. Odontogenic orbital cellulitis. Ophthal Plast Reconstr Surg. 2008;24:29-35. 10. Osmanovic S, Al-Heeti OM, Lin AY, Zivin SP, Justo JA, Mayer SM, Aakalu VK, Moss HE, Patel MC. Staphylococcus aureus infection of the optic nerve. J Neuroophthalmol. 2015;35:48-50. 11. Leyngold I, Olivi A, Ishii M, Blitz A, Burger P, Subramanian PS, Gallia G. Acute chiasmal abscess resulting from perineural extension of invasive sino-orbital aspergillosis in an immunocompetent patient. World Neurosurg. 2014;81:203. e201-e206. 12. Caruso PA, Watkins LM, Suwansaard P, Yamamoto M, Durand ML, Romo LV, Rincon SP, Curtin HD. Odontogenic 397 Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited. Original Contribution orbital inflammation: clinical and CT findings-initial observations. Radiology. 2006;239:187-194. 13. Harris GJ. Subperiosteal abscess of the orbit. Arch Ophthalmol. 1983;101:751-757. 14. Smego RA Jr. Actinomycosis of the central nervous system. Rev Infect Dis. 1987;9:855-865. 398 15. Akhaddar A, Elouennass M, Baallal H, Boucetta M. Focal intracranial infections due to Actinomyces species in immunocompetent patients: diagnostic and therapeutic challenges. World Neurosurg. 2010;74:346-350. 16. Oostman O, Smego RA. Cervicofacial Actinomycosis: diagnosis and management. Curr Infect Dis Rep. 2005;7:170-174. Ghobrial et al: J Neuro-Ophthalmol 2016; 36: 393-398 Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited.