Idiopathic scrotal calcinosis is a rare, benign condition characterized by progressive calcification of the scrotal skin. A 29-year-old man who had undergone primary surgical excision of idiopathic scrotal calcinosis 7 years previously presented with recurrence that he had first noticed 3 years after surgery. Multiple yellowish nodules were observed in the scrotal skin and were confirmed by computed tomography. He underwent repeat resection without any postoperative complications. Histological examination of the surgical specimens revealed diffusely calcified areas within and beneath the squamous epithelium, some of which were associated with epithelial cysts. Immunopathological stains for antibodies against carcinoembryonic antigen, epithelial membrane antigen, and gross cystic disease fluid protein-15 were negative. This is the first reported case of recurrence of scrotal calcinosis. One possible reason for the relapse is that there were remnant seeds of calcification after the primary surgery. This case demonstrates the importance of careful identification and resection of all calcified areas, and of counseling patients about the possibility of relapse after surgical treatment.

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