[46, PPP1R12A基因变异致XY性发育障碍1例报告]。

Q3 Medicine
Wei Su, Zhe Su, Jing-Yu You, Hui-Ping Su, Li-Li Pan, Shu-Min Fan, Jian-Chun Yin
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引用次数: 0

摘要

患者是一名1岁零9个月的男孩,表现为46,xy性发育障碍(DSD),伴有严重的外生殖器男性化不足。实验室检查和超声检查显示睾丸间质细胞和支持细胞功能正常。基因检测发现PPP1R12A基因中存在一种新的致病性杂合变异c.1186dupA (p.T396Nfs*17)。此前已有13例PPP1R12A基因变异的报道。这些变异可能导致孤立的泌尿生殖系统或神经系统受累,或影响其他系统/器官,包括消化道、眼睛、心脏等。DSD患者典型表现为46xy核型和不同程度的男性化不足,包括外生殖器、性腺和生殖道。本文报道了一例由PPP1R12A基因杂合变异引起的46,XY DSD患儿伴生长迟缓,扩大了PPP1R12A基因变异相关的临床疾病谱系。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
[46,XY disorder of sex development caused by PPP1R12A gene variants: a case report].

The patient was a boy aged 1 year and 9 months who presented with 46,XY disorder of sex development (DSD), with severe undermasculinization of the external genitalia. Laboratory tests and ultrasound examinations showed normal functions of Leydig cells and Sertoli cells in the testes. Genetic testing revealed a novel pathogenic heterozygous variant, c.1186dupA (p.T396Nfs*17), in the PPP1R12A gene. Thirteen cases of PPP1R12A gene variants have been reported previously. These variants may cause isolated involvement of the genitourinary or neurological systems, or affect other systems/organs including the digestive tract, eyes, heart, etc. Patients with DSD typically present with a 46,XY karyotype and variable degrees of undermasculinization involving the external genitalia, gonads, and reproductive tract. This article reports a child with 46,XY DSD accompanied by growth retardation caused by a heterozygous variant in the PPP1R12A gene, which expands the clinical disease spectrum associated with PPP1R12A gene variants.

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来源期刊
中国当代儿科杂志
中国当代儿科杂志 Medicine-Pediatrics, Perinatology and Child Health
CiteScore
1.50
自引率
0.00%
发文量
5006
期刊介绍: The Chinese Journal of Contemporary Pediatrics (CJCP) is a peer-reviewed open access periodical in the field of pediatrics that is sponsored by the Central South University/Xiangya Hospital of Central South University and under the auspices of the Ministry of Education of China. It is cited as a source in the scientific and technological papers of Chinese journals, the Chinese Science Citation Database (CSCD), and is one of the core Chinese periodicals in the Peking University Library. CJCP has been indexed by MEDLINE/PubMed/PMC of the American National Library, American Chemical Abstracts (CA), Holland Medical Abstracts (EM), Western Pacific Region Index Medicus (WPRIM), Scopus and EBSCO. It is a monthly periodical published on the 15th of every month, and is distributed both at home and overseas. The Chinese series publication number is CN 43-1301/R;ISSN 1008-8830. The tenet of CJCP is to “reflect the latest advances and be open to the world”. The periodical reports the most recent advances in the contemporary pediatric field. The majority of the readership is pediatric doctors and researchers.
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