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Purpose: To report a case of intraocular sporotrichosis presenting with bilateral granulomatous panuveitis. Methods: Observational case report and literature review. Case presentation: A 62-year-old woman with a history of polycythemia vera presented with a non-healing ulcer at the left index finger, generalized erythematous papules, and bilateral granulomatous panuveitis. Cultures of skin and amputated finger identified Sporothrix schenckii. The diagnosis of intraocular sporotrichosis secondary to disseminated sporotrichosis was made. Intravenous liposomal Amphotericin B and intravitreal Amphotericin B were used to control systemic and ocular disease resulting in resolution of the skin lesions and intraocular inflammation. Conclusions: Intraocular sporotrichosis can manifest as bilateral granulomatous panuveitis in the setting of disseminated sporotrichosis. Treatment with intravenous and intravitreal antifungal therapy is useful for controlling intraocular infection. [ABSTRACT FROM AUTHOR]