The development of conjoined twins always catches the eyes of researchers and clinicians. Beyond the rareness of the cases, how they develop is a debatable issue. This report presented a case of ischiopagus conjoined twins who had two heads (dicephalus), four upper extremities (tetrabrachius), and were joined below the chest with two lower extremities (bipus). The twin's mother was referred from a primary hospital to Dessie Comprehensive Specialized Hospital for proper management of twin pregnancy, where a cesarean section was performed. The mother and her husband have no family history of birth defects or exposure to known teratogens. On imaging, the twins had separate hearts, lungs, and kidneys but a single liver, spleen, stomach, and intestine. They also shared genitourinary structures: a single penis with sub-coronal hypospadias and one imperforate anus. In addition, their placenta was single with one umbilical vein and two umbilical arteries. The conjoined twins had multiple accompanying cardiovascular anomalies but no external craniofacial, extremity, or brain anomalies. They passed away after 36 hours of follow-up in the neonatal intensive care unit at Dessie Comprehensive Specialized Hospital.