Lichiardopol Corina, Herlea V, Ioan Virginia, Tomulescu V, Mixich F
Department of Endocrinology, University of Medicine and Pharmacy of Craiova, Romania.
Rom J Morphol Embryol. 2006;47(3):295-9.
Both hypergonadotropic hypogonadism and myasthenia gravis can be parts of type II autoimmune polyendocrine syndrome and association between the two disorders has been reported in few cases. A 14 year old male patient with a personal history of bilateral cryptorchidism and ptosis was referred for delayed puberty. Clinical examination revealed eunuchoid habitus, small, soft testes, gynecomastia, ptosis, a myasthenic deficit score of 22.5 points and an IQ of 84 points. Decreased testosterone (0.064 ng/mL) and elevated LH (64.5 mUI/mL) were consistent with hypergonadotropic hypogonadism and karyotype was normal: 46,XY. Thyroid function, haematologic evaluation, BUN, electrolytes, and glycemia were in the normal range. Therapy consisted of anticholinesterase inhibitors, immunosuppressants, corticotherapy, testosterone; thoracoscopic thymectomy was performed showing thymic lymphoid hyperplasia on histopathologic examination. Myasthenic score improved (12.5 points), progressive virilization occurred, and a year later the patient presented with cushingoid features and obesity.
高促性腺激素性性腺功能减退症和重症肌无力均可为Ⅱ型自身免疫性多内分泌综合征的组成部分,且已有少数病例报道了这两种疾病之间的关联。一名14岁男性患者,有双侧隐睾和上睑下垂病史,因青春期发育延迟前来就诊。临床检查发现其具有类无睾体型、小而软的睾丸、乳腺增生、上睑下垂、重症肌无力缺陷评分为22.5分以及智商为84分。睾酮水平降低(0.064 ng/mL)和促黄体生成素升高(64.5 mUI/mL)符合高促性腺激素性性腺功能减退症,核型正常:46,XY。甲状腺功能、血液学评估、血尿素氮、电解质和血糖均在正常范围内。治疗包括抗胆碱酯酶抑制剂、免疫抑制剂、皮质激素治疗、睾酮治疗;进行了胸腔镜胸腺切除术,组织病理学检查显示胸腺淋巴样增生。重症肌无力评分改善(12.5分),出现进行性男性化,一年后患者出现库欣样特征和肥胖。