【Klinefelter综合征合并典型先天性肾上腺皮质功能障碍的临床观察】。

N I Volkova, I Yu Davidenko, D P Stavitskaya, E V Kudinova
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引用次数: 0

摘要

先天性肾上腺增生症(CAH)是一种参与肾上腺皮质皮质醇合成的酶或转运蛋白的缺陷。以皮质醇缺乏和雄激素过多为特征的男性型CAH。Klinefelter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造成诊断和医疗困难,导致不良后果并影响生活质量。一位43岁的病人向医生抱怨4年来缺乏勃起,乳房增大。在3岁时,根据阴毛的生长,尿液中17-酮类固醇水平的下降和CAH的遗传分析诊断,怀疑是男性形式。处方强的松每日5毫克。5岁时,根据表型特征和核型诊断为Klinefelter综合征(XXY)。在13岁时,医生给他开了刺激绒毛膜促性腺激素的药物,每次注射10次,一个疗程。18岁时,患者自行停止使用强的松。此外,他没有接受CAH和Klinefelter综合征的药物治疗。42岁时,肾上腺CT显示左肾上腺形成。根据激素活性检查的结果,检测到高水平的醛固酮和肾素。诊断为左肾上腺醛固瘤并行左肾上腺切除术。组织学检查未确诊为醛固瘤。体格检查,BMI 30 kg/m2,基因型肥胖,右睾丸未触诊,左睾丸致密,体积减小。阴茎小。总睾酮水平下降,SHBG、LH、FSH正常。阴囊器官的超声检查显示睾丸和附属物尺寸减小,右侧睾丸体积增大。因此,诊断为CAH,阳刚型和Klinefelter综合征,原发性性腺功能减退,右侧隐睾。每日处方氢化可的松30毫克。在右睾丸肿瘤手术治疗之前,没有使用睾酮制剂进行激素替代治疗。在这个临床病例的例子中,我们已经证明了两种内分泌病理的结合,在这个病人的管理中犯了严重的错误。这类患者的管理需要多学科合作,避免失误,改善患者的预后和生活质量。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
[Combination of Klinefelter syndrome and the classic form of congenital dysfunction of the adrenal cortex: clinical observation].

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.

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