Suppr超能文献

一名患有亚端粒不平衡易位t(10;17)(p15.3;p13.3)的患者中Miller-Dieker关键区域的重复。

Duplication of the Miller-Dieker Critical Region in a Patient with a Subtelomeric Unbalanced Translocation t(10;17)(p15.3;p13.3).

作者信息

Ruiz Esparza-Garrido R, Velázquez-Wong A C, Araujo-Solís M A, Huicochea-Montiel J C, Velázquez-Flores M Á, Salamanca-Gómez F, Arenas-Aranda D J

机构信息

Unidad de Investigación Médica en Genética Humana (UIMGH), Hospital de Pediatría, Instituto Mexicano del Seguro Social (IMSS), Mexico City, Mexico.

出版信息

Mol Syndromol. 2012 Aug;3(2):82-8. doi: 10.1159/000339639. Epub 2012 Jul 10.

Abstract

Submicroscopic duplications in the Miller-Dieker critical region have been recently described as new genomic disorders. To date, only a few cases have been reported with overlapping 17p13.3 duplications in this region. Also, small deletions that affect chromosome region 10p14→pter are rarely described in the literature. In this study, we describe, to our knowledge for the first time, a 5-year-old female patient with intellectual disability who has an unbalanced 10;17 translocation inherited from the father. The girl was diagnosed by subtelomeric FISH and array-CGH, showing a 4.43-Mb heterozygous deletion on chromosome 10p that involved 14 genes and a 3.22-Mb single-copy gain on chromosome 17p, which includes the critical region of the Miller-Dieker syndrome and 61 genes. The patient's karyotype was established as 46,XX.arr 10p15.3p15.1(138,206-4,574,436)x1,17p13.3(87,009-3,312,600)x3. Because our patient exhibits a combination of 2 imbalances, she has phenotypic features of both chromosome abnormalities, which have been reported separately. Interestingly, the majority of patients who carry the deletion 10p have visual and auditory deficiencies that are attributed to loss of the GATA3 gene. However, our patient also presents severe hearing and visual problems even though GATA3 is present, suggesting the involvement of different genes that affect the development of the visual and auditory systems.

摘要

米勒-迪克尔关键区域的亚微观重复最近被描述为新的基因组疾病。迄今为止,该区域17p13.3重复重叠的病例仅报道了少数几例。此外,影响染色体区域10p14→pter的小缺失在文献中很少被描述。在本研究中,据我们所知,我们首次描述了一名患有智力障碍的5岁女性患者,她有一个从父亲遗传而来的不平衡10;17易位。该女孩通过亚端粒荧光原位杂交(FISH)和比较基因组杂交芯片(array-CGH)进行诊断,结果显示10号染色体短臂上有一个4.43Mb的杂合缺失,涉及14个基因,17号染色体短臂上有一个3.22Mb的单拷贝增加,其中包括米勒-迪克尔综合征的关键区域和61个基因。患者的核型确定为46,XX。arr 10p15.3p15.1(138,206 - 4,574,436)x1,17p13.3(87,009 - 3,312,600)x3。由于我们的患者表现出两种不平衡的组合,她具有两种染色体异常的表型特征,这些特征此前已有分别报道。有趣的是,大多数携带10号染色体短臂缺失的患者有视觉和听觉缺陷,这归因于GATA3基因的缺失。然而,我们的患者尽管存在GATA3基因,但也出现了严重的听力和视觉问题,这表明有不同的基因参与影响视觉和听觉系统的发育。

相似文献

10

本文引用的文献

2
GATA3 abnormalities in six patients with HDR syndrome.GATA3 异常与六例 HDR 综合征患者相关。
Endocr J. 2011;58(2):117-21. doi: 10.1507/endocrj.k10e-234. Epub 2011 Jan 13.

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验