Endometriosis is an ectopic occurrence of tissue morphologically and functionally resembling endometrial tissue in regions outside the uterine cavity. Although scar endometriosis after surgery has been shown to be most common among all the extrapelvic forms of endometriosis, endometriosis after bladder exstrophy surgery has not been reported, and here we present the first known case. A 26-year-old woman with a history of bladder exstrophy was aware of a painful induration at the operative scar located in the left lower abdominal wall, and presented at our hospital. Although the symptoms resolved, recurring exacerbation was observed after 9 months. Abdominal magnetic resonance imaging showed a heterogeneous mass 16 mm in diameter in the left abdominal wall with high signal intensity on T1W1 and T2W1 images. She underwent excisional biopsy of the lesion under general anesthesia. Histopathology confirmed the diagnosis of endometriosis. Eighteen months after surgery, she was well and free from recurrence.
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