Tehran University of Medical Sciences Case Reports in Clinical Practice 2538-2683 8 6 2024 09 29 275 280 EN Azadeh Tarafdari Department of Obstetrics and Gynecology, Imam Khomeini Hospital Complex, Vali-e-Asr Hospital, Tehran University of Medical Sciences, Tehran, Iran Sanaz Ghashghaee Department of Obstetrics and Gynecology, Imam Khomeini Hospital Complex, Vali-e-Asr Hospital, Tehran University of Medical Sciences, Tehran, Iran Zeinab Mansouri Department of Obstetrics and Gynecology, Imam Khomeini Hospital Complex, Vali-e-Asr Hospital, Tehran University of Medical Sciences, Tehran, Iran Mohammadamin Parsaei Maternal, Fetal & Neonatal Research Center, Family Health Research Institute, Tehran University of Medical Sciences, Tehran, Iran 2024 01 15 2024 05 05 Gestational choriocarcinoma, a rare variant of gestational trophoblastic disease, typically arises from abnormal trophoblastic cell proliferation post-pregnancy, often associated with a hydatidiform mole. While most choriocarcinoma cases develop within the uterine cavity, an exceedingly rare manifestation occurs within a previous cesarean section scar. In our study, a 31-year-old woman with a history of hydatidiform mole presented with amenorrhea and spotting. Initial assessments revealed elevated beta-human chorionic gonadotropin (βhCG) levels and a heteroechoic mass at her prior cesarean section scar in sonographic examination. Histopathologic findings and the metastatic workup categorized the patient as FIGO stage I, indicating no metastasis. Due to the absence of metastasis, adjuvant chemotherapy was omitted. Total abdominal hysterectomy confirmed choriocarcinoma. Post-surgery, βhCG levels notably decreased, remaining negative during the two-year follow-up with no reported symptoms. Our findings suggest that surgical resection and meticulous βhCG monitoring may be a promising treatment strategy for non-metastatic choriocarcinoma. https://crcp.tums.ac.ir/index.php/crcp/article/view/918 https://crcp.tums.ac.ir/index.php/crcp/article/download/918/596