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Background  Malignant endometriosis in an episiotomy scar is rare; only seven cases have been reported previously. Here, we compare two cases of benign endometriosis and clear cell carcinoma.    Case presentation  The first case was a 54-year-old woman who presented with a large perineal lesion in her episiotomy scar with high 18F-fluorodeoxyglucose uptake. This location had a history of endometriosis many years ago. She underwent radical excision of the mass and bilateral inguinal lymph node dissection. Histological and immunohistochemical analysis confirmed the presence of clear cell carcinoma arising from endometriosis. Assisted radiotherapy was performed after surgery due to a positive lymph node. No recurrence was detected over a 1-year follow-up period. The second case deals with a 3 × 2 cm mass in the episiotomy scar of a 33-year-old woman. Part of the anal sphincter was resected because of the close proximity of the lesion. Because the disease lay very close to the anus, she received anal sphincter reconstruction combined with mass excision. Pathology result showed typical endometrial glands and interstitial tissues.    Conclusions  Deleterious change only happens in patients experiencing perineal endometriosis. Complete excision is crucial for this form of disease; sometimes impairment of the anal sphincter is also necessary. Patients with malignancy required a combination of treatments in order to improve their prognosis.

Comparison of clear cell carcinoma and benign endometriosis in episiotomy scar - two cases report and literature review