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Congenital perineal groove is a rare and likely underrecognized congenital malformation occurring almost exclusively in females. It presents as a non-epithelialized mucosal sulcus located along the midline of the perineum, extending from the posterior vaginal fourchette to the anterior margin of the anus. Diagnosis is clinical, and the condition is generally benign and self-limiting, with spontaneous epithelialization during early childhood. Early recognition is essential to avoid misdiagnosis and unnecessary investigations or interventions.