| Summary: | Abstract Background Mullerian duct anomalies are a broad spectrum of congenital anomalies of the female genital tract presenting with variable symptoms like infertility, amenorrhoea, dysmenorrhea, pelvic endometriosis, and poor obstetric outcomes. Unicornuate uterus or hemiuterus occurs as a result of abnormal formation or failure of formation of the contralateral part. Case presentation We present a rare case report of hemiuterus with functional non-communicating horn along with hematometra in a 15-year-old female who presented with severe dysmenorrhoea since her menarche. Pelvic Ultrasonography demonstrated inconclusive findings of a heterogenous lesion in the right pelvic cavity adjacent to the uterus. Further, pelvic MRI revealed a hemiuterus on the left side with a normal endometrial cavity and a well-defined functional non-communicating horn on right side. The diagnosis of European Society of Human Reproduction (ESHRE) Classification U4a uterine anomaly (left hemiuterus with functional non-communicating horn on right side) was established. Conclusions ESHRE Class U4a comprises of hemiuterus with a functional rudimentary horn. This is considered clinically significant as it may lead to further complications, such as hematometra or ectopic pregnancy in the rudimentary horn. Hence, the correct identification of this entity is essential as laparoscopic removal is the current recommended management.
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