Neuro endocrinology letters · 2025
Clinical Analysis of 14 Cases with Androgen-Secreting Ovarian Sex Cord-Stromal Tumors: Diagnosis, Treatment, and Literature Review.
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Sang Yi-Quan, Wang Xian-Ling, Lyu Zhao-Hui, Ba Jian-Ming, Pei Yu, Guo Qing-Hua
Аннотация
OBJECTIVE: To analyze the clinical data of 14 patients with androgen-secreting ovarian sex cord-stromal tumors (OSCSTs), a rare cause of female hyperandrogenism (HA), aiming to enhance differential diagnosis and reduce misdiagnosis of these diseases.
METHODS: Fourteen female patients with pathologically confirmed OSCSTs were retrospectively enrolled in this study.General clinical data were retrospectively collected from medical records, encompassingclinical manifestations, diagnostic and therapeutic interventions. A systematic literature review about the management of OSCSTs were performed.
RESULTS: Among 14 female patients with OSCSTs, age ranged from 12 to 69 years (mean ± SD: 34.4 ± 20.1 years), with symptom duration prior to diagnosis spanning 1-6 years (2.3 ± 1.4 years). Adolescent and reproductive-aged patients (n = 10) universally exhibited oligomenorrhea/amenorrhea with virilization, while postmenopausal patients (n = 4) presented with virilization alone. Preoperative testosterone levels were markedly elevated in all these patients (range 3.90-119.6 nmol/L; normal: 0.2-2.6). Imaging evaluation revealed ovarian masses on ultrasound in 78.6% (11/14) of cases. After complete tumors resection, testosterone reduced significantly to 0-6.51 nmol/L within 48 hours with complete resolution of hyperandrogenism symptoms. Adjuvant chemotherapy was administered in two patients with moderately/poorly differentiated Sertoli-Leydig cell tumors (Ki67 >30%). At one-month follow-up, testosterone normalized (0.5-2.6 nmol/L) in all patients with concomitant resolution of menstrual irregularities and improvement in virilizing symptoms.
CONCLUSION: OSCSTs are rare but clinically significant causes of hyperandrogenism, characterized by symptoms of HA such as oligomenorrhea, acne, hirsutism, and clitoromegaly. It has always been misdiagnosed due to overlapping features with PCOS and adrenal disorders. Early identification and complete surgical resection are critical for biochemical remission and symptom resolution.
METHODS: Fourteen female patients with pathologically confirmed OSCSTs were retrospectively enrolled in this study.General clinical data were retrospectively collected from medical records, encompassingclinical manifestations, diagnostic and therapeutic interventions. A systematic literature review about the management of OSCSTs were performed.
RESULTS: Among 14 female patients with OSCSTs, age ranged from 12 to 69 years (mean ± SD: 34.4 ± 20.1 years), with symptom duration prior to diagnosis spanning 1-6 years (2.3 ± 1.4 years). Adolescent and reproductive-aged patients (n = 10) universally exhibited oligomenorrhea/amenorrhea with virilization, while postmenopausal patients (n = 4) presented with virilization alone. Preoperative testosterone levels were markedly elevated in all these patients (range 3.90-119.6 nmol/L; normal: 0.2-2.6). Imaging evaluation revealed ovarian masses on ultrasound in 78.6% (11/14) of cases. After complete tumors resection, testosterone reduced significantly to 0-6.51 nmol/L within 48 hours with complete resolution of hyperandrogenism symptoms. Adjuvant chemotherapy was administered in two patients with moderately/poorly differentiated Sertoli-Leydig cell tumors (Ki67 >30%). At one-month follow-up, testosterone normalized (0.5-2.6 nmol/L) in all patients with concomitant resolution of menstrual irregularities and improvement in virilizing symptoms.
CONCLUSION: OSCSTs are rare but clinically significant causes of hyperandrogenism, characterized by symptoms of HA such as oligomenorrhea, acne, hirsutism, and clitoromegaly. It has always been misdiagnosed due to overlapping features with PCOS and adrenal disorders. Early identification and complete surgical resection are critical for biochemical remission and symptom resolution.