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性腺功能减退症:腭裂青少年一定存在促性腺激素低下吗?一例克氏综合征的意外发现。

Hypogonadism: Is It Always Hypogonadotropic in an Adolescent With a Cleft Palate? A Surprising Case of Klinefelter Syndrome.

机构信息

HSU, Division of Pediatric Endocrinology, Department of Pediatrics, 147023Zeynep Kamil Women and Children's Diseases Training and Research Hospital, Istanbul, Turkey.

Department of Pediatrics, Yeni Yuzyil Medical School, Istanbul, Turkey.

出版信息

Cleft Palate Craniofac J. 2021 Jun;58(6):787-790. doi: 10.1177/1055665620967601. Epub 2020 Oct 27.

Abstract

Pituitary hormone deficiencies may occur in children with midline defects; in these cases, hypogonadism is usually hypogonadotropic. Herein, we report a boy at the age of 13.8 years with mild mental retardation, previously operated for complete cleft palate (isolated) and presented with hypoglycemia due to isolated secondary adrenal insufficiency, who further had a decrease in testicular size with increased follicle-stimulating hormone level (hypergonadotropic hypogonadism) and diagnosed with Klinefelter syndrome. Klinefelter syndrome in childhood is rarely diagnosed and cases are observed in a wide spectrum. Although some regional duplications of the X chromosome also show midline defects such as spina bifida-neural tube defects, mental retardation, hypopituitarism (mostly growth hormone deficiency); coexistence of Klinefelter syndrome and isolated secondary adrenal deficiency/midline defect in our case may also be coincidental. However, to our knowledge, this is the first case in literature with this association in a patient with a 47, XXY karyotype.

摘要

垂体激素缺乏症可能发生在中线缺陷的儿童中;在这些情况下,性腺功能减退症通常是促性腺激素低下型的。在此,我们报告了一名 13.8 岁的男孩,有轻度智力障碍,曾因完全腭裂(孤立性)接受手术治疗,因孤立性继发性肾上腺功能不全而出现低血糖,进一步出现睾丸体积缩小,卵泡刺激素水平升高(促性腺激素性性腺功能减退症),并诊断为克氏综合征。儿童期克氏综合征很少被诊断,病例观察范围广泛。虽然 X 染色体的一些区域重复也会出现中线缺陷,如脊柱裂-神经管缺陷、智力障碍、垂体功能减退(主要是生长激素缺乏症);但在我们的病例中,克氏综合征和孤立性继发性肾上腺缺乏/中线缺陷的共存也可能是偶然的。然而,据我们所知,这是文献中首例在 47,XXY 核型患者中出现这种关联的病例。

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