Mycobacterium hemophilum Infection Presenting as Filamentary Keratopathy in an Immunocompromised Adult

PURPOSE: To report a case of Mycobacterium hemophilum of the eye. METHODS: Case report with pathologic correlation. A 55-year-old Malaysian man with a 3-year history of graft-versus-host disease presented with dry eye and keratopathy. RESULTS: The diagnosis was not initially evident, despite biopsy specimens of the conjunctiva. Definitive diagnosis was made after dermatology consultation suggested a histoid variant of lepromatous leprosy, prompting Ziehl-Neelsen staining of the initial and subsequent conjunctival biopsies with subsequent polymerase chain reaction testing. Anti-M. hemophilum treatment resulted in prompt resolution of ocular signs. CONCLUSIONS: Mycobacterium hemophilum is a rare condition, affecting mainly immunocompromised patients. Although filamentary keratopathy has been described as common manifestations of leprosy, to date, no ocular manifestations of M. hemophilum have been described. Conjoint management with infectious disease and clinical microbiology is imperative to ensure accurate diagnosis and appropriate early intervention. The effect of systemic immunosuppression is relevant in such patients.