Endometriosis is a frequent gynecological disease among women of reproductive age, a benign condition with an uncertain etiology, but with multiple theories being proposed as main mechanisms of development. Perineal endometriosis is an uncommon condition, with various theories about its pathogenesis including the contiguous propagation of endometrial tissue over the episiotomy perineal scar. A 36-year-old woman, G2 P2, presented with cyclic inflammation and pain in the episiotomy area, with significant impairment of her daily quality of life. Clinical examination revealed a palpable mass in the episiotomy area and a probable clinical diagnosis of perineal scar endometriosis was considered, with further investigation being recommended. The patient was surgically treated by local excision of the perineal mass and the histopathologic examination of the mass confirmed the diagnosis of perineal endometriosis in the episiotomy scar. Although perineal endometriosis remains a rare condition, it should be considered in all patients with an anterior vaginal delivery and a painful perineal mass during menstrual cycle, a wide excision of the affected tissue remaining the best option for a permanent cure.