Skin lesions are common in renal transplant recipients (RTR) and the clinical distinction of malignancy versus infection may be difficult in this patient population, with the need for further histological and biological investigations. We report here on a 73-year-old male RTR who presented with Alternaria infectoria phaeohyphomycosis of 1 year's duration. Mycological cultures were negative, and the diagnosis was performed by real-time polymerase chain reaction assay and direct sequencing. The extension of the lesion under itraconazole treatment required its surgical excision. Alternaria are ubiquitous plant-inhabiting saprobes, which are increasingly associated with opportunistic phaeohyphomycosis in immunocompromised individuals.