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We report the clinical course of a 47-year-old diabetic and hypertensive male who presented to us with diminution of vision in both eyes (OU) for 4 months. He was diagnosed with granulomatosis with polyangiitis and was treated with cyclophosphamide and steroids elsewhere. Before this, he was on steroids and methotrexate for probable sarcoidosis based on cavitary lesions in the lungs. During tapering of the steroids and so in relatively immunosuppressed status, he developed fever and maculopapulo-nodular skin lesions. Skin lesion biopsy confirmed Histoplasma capsulatum and he was started on systemic antifungals. Later, he developed bilateral endogenous endophthalmitis and was managed with intravitreal medications followed by vitrectomy with lensectomy OU. The patient regained vision and is maintaining the same until 1 year of follow-up. Thus, in spite of its rare occurrence, H. capsulatum should be considered as a cause of endogenous endophthalmitis in an immunosuppressed host with systemic histoplasmosis.