Background.: Although uterine anomalies are uncommon, gynecologists must be aware of the anatomical challenges that may be encountered from these anomalies. Cervical cancer in the context of a mullerian lateral fusion defect is rare. Case.: A case of a unilateral IB1 squamous cell carcinoma of the cervix in the setting of a complete uterovaginal septum and cervix duplex is described. Conclusion.: Uterine anomalies compound diagnostic difficulty of routine pathology. Thorough examination and evaluation are crucial for timely diagnosis and treatment. Genital tract duplication, although rare, should always be considered. © 2006 Elsevier Inc. All rights reserved.