Initial presentation of invasive fungal infections such as histoplasmosis can include non-specific clinical manifestations, especially in immunocompromised patients. A high index of suspicion is required to identify atypical manifestations of these diseases, which carry a high risk of mortality, if the diagnosis is delayed or missed. We describe a case of a kidney transplant recipient with cutaneous lesions as initial manifestation of progressive disseminated histoplasmosis where a skin biopsy was crucial to an early diagnosis.