Purpose: The study aim was to report diagnostic and therapeutic challenges in treatment of a patient with cervical dysplasia and congenital uterine anomaly. Case Report: A 53-year-old women with Mullerian anomaly – uterus duplex (bicorporal septate uterus) and Yshaped endocervical canal was referred due to repeated abnormal Pap smears. She underwent endocervical curettage of both canals and the endocervical septum biopsy which revealed presence of cervical intraepithelial neoplasia (CIN) III. Cervical conization was considered technically unfeasible because of abnormal cervical anatomy (lesions deep in the cervical canal on the cervical bifurcation where the cervical wall is the thickest). Classical open abdominal hysterectomy was performed. Patient had two almost equally-sized, symmetrical uterine bodies connected in the isthmico-cervical region, with normal left and obstructed right hemi-vagina. Postoperative histopathological findings confirmed that dysplasia was located in the region where two endocervical canals conjoined. Conclusion: Diagnostic and therapeutic approach to patients with uterine anomalies has to be individualized, based on anomaly type, patient's age, reproductive history, and patient's preferences.