Abstract
Background/Objectives: Heterotopic cesarean scar pregnancy (HCSP) is an exceptionally rare and potentially life-threatening form of ectopic pregnancy, characterized by the coexistence of a viable intrauterine pregnancy and an ectopic implantation within a previous cesarean section scar. Its incidence has risen in recent years, primarily due to the increased rate of cesarean deliveries and the widespread use of assisted reproductive technologies (ART). This systematic review aims to provide a comprehensive synthesis of published evidence on HCSP, with a focus on epidemiology, diagnostic challenges, therapeutic strategies, complications, and maternal-fetal outcomes. Methods: A systematic literature search was conducted in PubMed, Scopus, and Web of Science up to May 2025, in accordance with PRISMA guidelines. Included studies comprised case reports, case series and retrospective reviews documenting confirmed HCSP cases. Data were extracted on clinical presentation, imaging, treatment approaches, outcomes, and complications. Results: Thirty studies reporting 40 confirmed HCSP cases were included. Transvaginal ultrasonography was the primary diagnostic tool, although diagnosis was often delayed by the presence of a viable intrauterine pregnancy. Management strategies included surgical intervention, local medical therapy and conservative approaches or expectant management. Maternal complications included hemorrhage and uterine rupture, while fetal outcomes were variable. In selected cases, intrauterine pregnancy continued to term. Conclusions: HCSP is a rare but high-risk obstetric entity requiring early recognition and multidisciplinary management. Prompt ultrasound-based diagnosis and individualized treatment can significantly reduce maternal morbidity and improve fetal outcomes. Further multicenter studies are warranted to establish standardized diagnostic and management protocols.