Objectives: To explore the clinical manifestations and pathological features in the biopsy of ovarian Sertoli-Leydig cell tumor, as well as to improve the clinical understanding of the disease. Methods: A case of pregnancy and childbirth after Sertoli-Leydig cell tumor resection was retrospectively analyzed. The patients’ clinical data were collected, including the clinical manifestations, postoperative biopsy results, auxiliary examination results, immunohistochemical results, treatment, and prognosis of the patient. Results: (1) SLCT occurred unilaterally; (2) according to the International Federation of Obstetrics and Gynecology (FIGO), the clinical staging was stage IA; according to the pathological classification of malignant tumors, it was grade ? (moderately differentiated); (3) a healthy female live baby was delivered. Conclusion: Such tumors are rare low-grade malignancies and are even rarer in pregnancy. An increase in preoperative testosterone levels with positive ultrasonography results can be used to assist diagnosis; however, postoperative biopsy pathology remains the “gold standard” for the diagnosis of SLCTs. The definite diagnosis of SLCTs is of great significance for surgical planning and prognostic evaluation.
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