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[克兰费尔特综合征与经典型先天性肾上腺皮质功能不全的合并症:临床观察]

[Combination of Klinefelter syndrome and the classic form of congenital dysfunction of the adrenal cortex: clinical observation].

作者信息

Volkova N I, Davidenko I Yu, Stavitskaya D P, Kudinova E V

机构信息

Rostov State Medical University.

出版信息

Probl Endokrinol (Mosk). 2024 Mar 11;71(1):27-31. doi: 10.14341/probl13298.

Abstract

Congenital adrenal hyperplasia (CAH) is a defect in one of the enzymes or transport proteins involved in the synthesis of cortisol in the adrenal cortex. Virile form of CAH characterized by cortisol deficiency and hyperandrogenism. Klinefelter syndrome is one of the most frequent chromosomal diseases leading to the development of primary hypogonadism. The manifestation of these two diseases could cause difficulties in diagnosis and medical treatment that lead to adverse consequences and affect the quality of life.A 43-years-old patient consulted a physician complaining about the lack of erections for 4 years, breast enlargement. At the age of 3 years based on experienced growth of pubic hair, decreased level of 17-ketosteroids in the urine and genetic analysis diagnosis of CAH, virile form was suspected. Prednisone 5 mg daily was prescribed. At the age of 5, based on phenotypic features and karyotyping Klinefelter Syndrome (XXY) was diagnosed. At the age of 13, stimulating hormonal chorionic gonadotropin drug with only one course of 10 injections was prescribed. At the age of 18, the patient independently canceled the use of prednisone. Further, he did not receive medication therapy for CAH and Klinefelter syndrome. At the age of 42, adrenal CT revealed formation of the left adrenal gland. According to the results of the hormonal activity examination, high levels of aldosterone and renin were detected. A diagnosis of left adrenal aldosteroma was made and a left-sided adrenalectomy was performed. Histological examination diagnosis of aldosteroma did not confirmed. On physical examination, BMI 30 kg/m2, genoid type of obesity, right testicle isn`t palpated, left testicle is dense, reduced in size. Small penis size. Decreased level of total testosterone, normal level of SHBG, LH and FSH was revealed. Ultrasound of the scrotum organs revealed decrease in the size of the testicles and appendages, a volumetric formation of the right testicle. Thus, diagnosis of CAH, virile form and Klinefelter syndrome, primary hypogonadism, right-sided cryptorchidism was confirmed. Hydrocortisone 30 mg daily was prescribed. Hormone replacement therapy with testosterone preparations was not prescribed until surgical treatment of neoplasm of the right testicle will be performed. On the example of this clinical case, we have demonstrate a combination of two endocrine pathologies and serious mistakes were made in the management of this patient. The management of such patients requires a multidisciplinary approach, which will avoid mistakes and improve the prognosis and quality of life of these patients.

摘要

先天性肾上腺皮质增生症(CAH)是肾上腺皮质中参与皮质醇合成的一种酶或转运蛋白存在缺陷。CAH的男性化形式表现为皮质醇缺乏和雄激素过多。克兰费尔特综合征是导致原发性性腺功能减退的最常见染色体疾病之一。这两种疾病的表现可能会导致诊断和治疗困难,进而产生不良后果并影响生活质量。一名43岁患者咨询医生,称其4年来一直无法勃起且乳房增大。3岁时,根据阴毛提前生长、尿中17-酮类固醇水平降低以及基因分析,怀疑诊断为CAH男性化形式,遂每日服用5毫克泼尼松。5岁时,根据表型特征和染色体核型分析诊断为克兰费尔特综合征(XXY)。13岁时,仅注射一个疗程共10针的促性腺激素绒毛膜促性腺激素药物。18岁时,患者自行停用泼尼松。此后,他未接受针对CAH和克兰费尔特综合征的药物治疗。42岁时,肾上腺CT显示左肾上腺有肿物。根据激素活性检查结果,检测到醛固酮和肾素水平升高。诊断为左肾上腺醛固酮瘤并进行了左侧肾上腺切除术。组织学检查未确诊为醛固酮瘤。体格检查发现,体重指数(BMI)为30kg/m²,为类人猿型肥胖,右侧睾丸无法触及,左侧睾丸质地硬且体积缩小。阴茎短小。总睾酮水平降低,性激素结合球蛋白(SHBG)、促黄体生成素(LH)和促卵泡生成素(FSH)水平正常。阴囊器官超声检查显示睾丸和附睾体积减小,右侧睾丸有一个占位性病变。因此,确诊为CAH男性化形式、克兰费尔特综合征、原发性性腺功能减退、右侧隐睾症。遂每日服用30毫克氢化可的松。在对右侧睾丸肿瘤进行手术治疗之前,未给予睾酮制剂进行激素替代治疗。以该临床病例为例,我们展示了两种内分泌疾病的合并情况,并且在该患者的治疗过程中犯了严重错误。对此类患者的管理需要多学科方法,这将避免错误并改善这些患者的预后和生活质量。

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本文引用的文献

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The role of hypogonadism in Klinefelter syndrome.性腺功能减退在克兰费尔特综合征中的作用。
Asian J Androl. 2014 Mar-Apr;16(2):185-91. doi: 10.4103/1008-682X.122201.

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