Li Yan, Zhao Junfeng, Chen Yi, Zhang Jing, Chen Hehe
Department of Pediatrics Surgery, Women and Children's Hospital of Ningbo University, Ningbo, Zhejiang, China.
Department of Surgical Nursing, Women and Children's Hospital of Ningbo University, Ningbo, Zhejiang, China.
Front Endocrinol (Lausanne). 2025 May 27;16:1545449. doi: 10.3389/fendo.2025.1545449. eCollection 2025.
Adenomatoid tumors, rare benign mesothelial neoplasms primarily involving the reproductive tract, account for 30% of paratesticular masses and predominantly localize to the epididymal head. Although typically diagnosed in adults (mean age: 33 years), pediatric cases are exceptionally rare. Conservative excision with testicular preservation is optimal, yet diagnostic ambiguity often leads to unnecessary orchidectomy. A 5.2-year-old boy presented with asymptomatic left testicular enlargement. MRI identified a 13 × 14 mm heterogeneous mass on the lateral testis. Intraoperative frozen section analysis suggested benign/low-grade malignancy, confirmed postoperatively as a parenchymal adenomatoid tumor via immunohistochemistry. A 5-year prospective study assessed testicular function through serial measurements of serum hormones [follicle-stimulating hormone (FSH), luteinizing hormone (LH), testosterone, inhibin B (INHB), and anti-Müllerian hormone (AMH)] and bilateral testicular volume (TV). Our findings were as follows: Left TV exceeded age-adjusted norms by 2.1-fold (4.2 mL vs. 2.0 mL), contrasting with right TV at 0.5× mean (1.0 mL). AMH declined to a nadir (2.1 ng/mL) at 48 months, recovering to 3.8 ng/mL by 60 months. INHB reached minimal levels (45 pg/mL) at 6 months postresection, peaking at 128 pg/mL by study endpoint. FSH (1.2-1.5 IU/L) and testosterone (T, 0.15-0.18 ng/mL) remained prepubertal, while LH (0.3-0.5 IU/L) persisted near lower normative limits. This study demonstrates the progressive decline of Sertoli cell function in pediatric adenomatoid tumor survivors, detectable through longitudinal monitoring of INHB, AMH, and TV. The 2.1-fold compensatory hypertrophy in the affected testis suggests adaptive mechanisms requiring further investigation. Conservative resection guided by frozen section analysis prevents orchidectomy, while biomarker surveillance enables early detection of testicular dysfunction for timely intervention.
腺瘤样瘤是一种罕见的良性间皮肿瘤，主要累及生殖道，占睾丸旁肿块的30%，主要位于附睾头部。虽然通常在成人中诊断（平均年龄：33岁），但儿科病例极为罕见。保留睾丸的保守性切除是最佳选择，但诊断不明确常导致不必要的睾丸切除术。一名5.2岁男孩出现无症状的左侧睾丸肿大。磁共振成像（MRI）在睾丸外侧发现一个13×14毫米的异质性肿块。术中冰冻切片分析提示为良性/低级别恶性，术后通过免疫组化确诊为实质性腺瘤样瘤。一项为期5年的前瞻性研究通过连续测量血清激素[促卵泡激素（FSH）、促黄体生成素（LH）、睾酮、抑制素B（INHB）和抗苗勒管激素（AMH）]以及双侧睾丸体积（TV）来评估睾丸功能。我们的研究结果如下：左侧睾丸体积超过年龄校正标准2.1倍（4.2毫升对2.0毫升），而右侧睾丸体积仅为平均值的0.5倍（1.0毫升）。AMH在48个月时降至最低点（2.1纳克/毫升），到60个月时恢复至3.8纳克/毫升。INHB在切除术后6个月达到最低水平（45皮克/毫升），到研究终点时峰值为128皮克/毫升。FSH（1.2 - 1.5国际单位/升）和睾酮（T，0.15 - 0.18纳克/毫升）仍处于青春期前水平，而LH（0.3 - 0.5国际单位/升）持续接近较低的正常范围下限。本研究表明，通过对INHB、AMH和睾丸体积的纵向监测可发现，儿科腺瘤样瘤幸存者的支持细胞功能呈进行性下降。患侧睾丸2.1倍的代偿性肥大提示存在需要进一步研究的适应性机制。冰冻切片分析指导下的保守性切除可避免睾丸切除术，而生物标志物监测能够早期发现睾丸功能障碍以便及时干预。
