We report a unique case of unilateral incontinentia pigmenti in a male infant. Several days after birth, a few vesicles appeared on the right lower leg. After then, erythema with vesicles appeared, limited to the right inguinal area. Histopathological examination revealed eosinophilic spongiosis in the epidermis and an inflammatory cell infiltrate in company with scattered melanophages in the upper dermis. A thorough systemic examination did not show any other abnormalities of ectodermal organs, such as eyes, teeth, and central nervous systems. Based on all the results, we diagnosed the present case as a unilateral and mild case of incontinentia pigmenti. Herein we discuss the clinical features of male incontinentia pigmenti.
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