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Mayer-Rokitansky-Kuster-Hauser syndrome (MRKHS) is characterized by vaginal agenesis with variable Müllerian duct abnormalities. We report here a case of uterine adenomyosis which developed from a hypoplastic uterus in a patient with MRKHS. A 55-year-old postmenopausal woman visited a university hospital for pelvic mass. She had underwent vaginoplasty via the McIndoe procedure for MRKHS at 15 years of age. Pelvic magnetic resonance imaging showed a 5.4 × 4.8 × 4.7 cm mass suspicious for a uterine myoma. She received total abdominal hysterectomy with bilateral salpingo-oophorectomy, and neither the cervix nor endometrium was found grossly in the surgical specimen. The final histologic diagnosis was uterine adenomyosis.

Uterine Adenomyosis Which Developed from Hypoplastic Uterus in Postmenopausal Woman with Mayer-Rokitansky-Kuster-Hauser Syndrome: A Case Report