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首页> 外文期刊>Sao Paulo Medical Journal >Testicular adrenal rest tumor in infertile man with congenital adrenal hyperplasia: case report and literature review
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Testicular adrenal rest tumor in infertile man with congenital adrenal hyperplasia: case report and literature review

机译：不育男性先天性肾上腺增生的睾丸肾上腺休息肿瘤：病例报告及文献复习

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CONTEXT: Synthesis of cortisol and aldosterone is impaired in patients with congenital adrenal hyperplasia (CAH) because of 21-hydroxylase deficiency. Men with CAH have low fertility rates compared with the normal population, and this is related to testicular adrenal rest tumors. Findings of azoospermia in combination with a testicular tumor on ultrasound are likely to have a mechanical cause, especially when in the testicular mediastinum. The preferred treatment method consists of intensive corticoid therapy. However, when the tumor is unresponsive to steroid therapy, surgical treatment should be considered. CASE REPORT: We present the case of a male patient with CAH due to 21-hydroxylase deficiency who presented a testicular tumor and azoospermia. Treatment with low daily corticoid doses had previously been started by an endocrinologist, but after 12 months, no significant change in sperm count was found. Although the adrenocorticotrophic hormone and 17-hydroxyprogesterone levels returned to normal values, the follicle-stimulating hormone (FSH), luteinizing hormone and testosterone levels remained unchanged. Ultrasound examination confirmed that the testicles were small and heterogenous bilaterally, and revealed a mosaic area at the projection of the testis network bilaterally. Magnetic resonance imaging confirmed the finding. Testicular biopsy revealed the presence of preserved spermatogenesis and spermiogenesis in 20% of the seminiferous tubules in the right testicle. The patient underwent testis-sparing tumor resection. After 12 months of follow-up, there was no tumor recurrence but the patient still presented azoospermia and joined an intracytoplasmic sperm injection program.
机译：背景：由于21-羟化酶缺乏症，先天性肾上腺皮质增生（CAH）患者皮质醇和醛固酮的合成受到损害。与正常人群相比，CAH男性的生育率低，这与睾丸肾上腺休息肿瘤有关。超声检查发现无精症合并睾丸肿瘤可能是机械性原因，尤其是在睾丸纵隔中。优选的治疗方法包括强化皮质激素治疗。但是，当肿瘤对类固醇治疗无反应时，应考虑手术治疗。病例报告：我们报道了一例男性男性，由于21-羟化酶缺乏症导致CAH，并出现睾丸肿瘤和无精子症。以前由内分泌学家开始使用每日低剂量的类固醇激素治疗，但在12个月后，未发现精子数量有明显变化。尽管肾上腺皮质营养激素和17-羟孕酮水平恢复正常，但促卵泡激素（FSH），促黄体生成激素和睾丸激素水平保持不变。超声检查证实双侧睾丸小而异质，并在双侧睾丸网投影处显示出马赛克区域。磁共振成像证实了这一发现。睾丸活检显示在右睾丸中有20％的曲细精管中存在保留的精子发生和精子发生。该患者接受了保留睾丸的肿瘤切除术。随访12个月后，没有肿瘤复发，但患者仍表现出无精子症，并参加了胞浆内精子注射计划。

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