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Objective:To study the clinical presentation and treatment of patients with perineal endometriosis with anal sphincter involvement.Method:The clinical data of 26 patients with perineal endometriosis who were operated on during 1983-2007 in our hospital,and followed up for 0.5-14 years(average 32 months),were analyzed.Results:The incidence of perineal endometriosis with anal sphincter involvement was 0.21% among the total endometriosis cases in Peking Union Medical College Hospital,China.All patients had cyclical and painful lesions.The levels of CA125 were mostly normal except seven cases(7/26,26.9%)with anal sphincter involvement had re-current diseases because of incomplete excision.When the subsequent surgical resection was complete,there was no further recurrence diagnosed.Conclusions:Diagnosis of perineal endometriosis can be made correctly based on the patient's history and clini-cal manifestations.Complete excision of lesion is the first choice of treatment.The recurrence rate of incomplete ex-cision is high.The surgical excision of lesion is the best treatment for recurrence.Therefore it is important to evalu-ate preoperatively whether the anal sphincter is involved.