Abstract
PURPOSE: To report a case of severe bilateral coccidioidomycosis chorioretinitis in an immunosuppressed pediatric patient, demonstrating significant management challenges, therapeutic interventions, and a favorable outcome. OBSERVATIONS: A 14-year-old female with a history of 1p36 microdeletion and Takayasu Arteritis (TA), managed with adalimumab and mycophenolate, presented with acute respiratory symptoms and bilateral vision impairment. She had severe bilateral diffuse chorioretinitis and an anterior chamber sample revealed coccidioidomycosis. Over the next few months, she underwent 14 serial intravitreal injections in each eye (1 amphotericin, 13 voriconazole), intravenous amphotericin B and fluconazole, and restarted on immunosuppressants for her TA. At 8 months following initial presentation, her vision remains 20/100 in the right eye and 20/125 in the left eye. CONCLUSIONS AND IMPORTANCE: This case illustrates a rare occurrence of coccidioidomycosis with bilateral chorioretinal involvement, necessitating aggressive and prolonged antifungal therapy. The successful salvage of both globes and vision in such a severe bilateral infection highlights the potential of intensive, multi-modality treatment strategies in similar cases. Further studies are warranted to understand the optimal management of chorioretinal coccidioidomycosis and the impact of systemic immunosuppression.