Angiokeratomas are benign vascular lesions that can occur anywhere in the body. Fordyce angiokeratomas, also known as genital angiokeratomas, often develop on the vulva in women and the scrotum in males. A subtype of genital angiokeratoma in males is called penile angiokeratoma (PEAKER). In females, clitoral angiokeratoma (CLANKER) is the embryologic equivalent. As a result of the underlying pathophysiology, these lesions are often bilateral. Unilateral Fordyce angiokeratoma instances are infrequent, and unilateral PEAKER cases have never been previously documented. We describe a case of Fordyce's unilateral angiokeratoma with unilateral PEAKER. To the best of our knowledge, such a variation in presentation of this rare disease has not been previously reported.
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