A syndrome complex consisting of didelphys uteruses, obstructed hemivagina and ipsilateral renal agenesis is one of the most complex and rare congenital anomalies of the female urogenital system. In most of the cases patients remain asymptomatic till puberty only to present with dysmenorrhea, pelvic pains, and intermenstrual bleeding or abdominal lumps in the period following menarche. We present one of the atypical delayed presentations of this syndrome at 26 years of age with dysmenorrhea and irregular vaginal bleeding. Appropriate combination of imaging, clinical examination, and hysteroscopic evaluation confirmed the diagnosis followed by appropriate surgical correction to relieve the patient of her complaints.
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