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严重生长发育迟缓、小头畸形、智力残疾和特征性面部特征是由纯合子QARS突变所致。

Severe growth deficiency, microcephaly, intellectual disability, and characteristic facial features are due to a homozygous QARS mutation.

作者信息

Leshinsky-Silver Esther, Ling Jiqiang, Wu Jiang, Vinkler Chana, Yosovich Keren, Bahar Sarit, Yanoov-Sharav Miri, Lerman-Sagie Tally, Lev Dorit

机构信息

Molecular Genetics Laboratory, Wolfson Medical Center, Holon, Israel.

Metabolic-Neurogenetic Clinic, Wolfson Medical Center, Holon, Israel.

出版信息

Neurogenetics. 2017 Jul;18(3):141-146. doi: 10.1007/s10048-017-0516-6. Epub 2017 Jun 15.

DOI:10.1007/s10048-017-0516-6
PMID:28620870
Abstract

Glutaminyl tRNA synthase is highly expressed in the developing fetal human brain. Mutations in the glutaminyl-tRNA synthetase (QARS) gene have been reported in patients with progressive microcephaly, cerebral-cerebellar atrophy, and intractable seizures. We have previously reported a new recessive syndrome of severe linear growth retardation, poor weight gain, microcephaly, characteristic facial features, cutaneous syndactyly of the toes, high myopia, and intellectual disability in two sisters of Ashkenazi-Jewish origin (Eur J Med Genet 2014;57(6):288-92). Homozygosity mapping and whole exome sequencing revealed a homozygous missense (V476I) mutation in the QARS gene, located in the catalytic domain. The patient's fibroblasts demonstrated markedly reduced QARS amino acylation activity in vitro. Furthermore, the same homozygous mutation was found in an unrelated girl of Ashkenazi origin with the same phenotype. The clinical presentation of our patients differs from the original QARS-associated syndrome in the severe postnatal growth failure, absence of epilepsy, and minor MRI findings, thus further expanding the phenotypic spectrum of the glutaminyl-tRNA synthetase deficiency syndromes.

摘要

谷氨酰胺基tRNA合成酶在发育中的人类胎儿大脑中高度表达。据报道,谷氨酰胺基tRNA合成酶(QARS)基因突变见于患有进行性小头畸形、大脑小脑萎缩和顽固性癫痫的患者。我们之前报道过一种新的隐性综合征,两名阿什肯纳兹犹太裔姐妹患有严重的线性生长迟缓、体重增加不佳、小头畸形、特征性面部特征、脚趾皮肤并指、高度近视和智力残疾(《欧洲医学遗传学杂志》2014年;57(6):288 - 92)。纯合子定位和全外显子组测序显示,位于催化结构域的QARS基因存在纯合错义(V476I)突变。患者的成纤维细胞在体外显示出明显降低的QARS氨基酰化活性。此外,在一名具有相同表型的阿什肯纳兹裔无关女孩中也发现了相同的纯合突变。我们患者的临床表现与最初的QARS相关综合征不同,表现为严重的出生后生长发育不良、无癫痫以及轻微的MRI检查结果,从而进一步扩展了谷氨酰胺基tRNA合成酶缺乏综合征的表型谱。

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本文引用的文献

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Mutations in the glutaminyl-tRNA synthetase gene cause early-onset epileptic encephalopathy.谷氨酰胺-tRNA合成酶基因突变会导致早发性癫痫性脑病。
J Hum Genet. 2015 Feb;60(2):97-101. doi: 10.1038/jhg.2014.103. Epub 2014 Dec 4.
2
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Neurogenetics. 2015 Apr;16(2):145-9. doi: 10.1007/s10048-014-0432-y. Epub 2014 Nov 30.
3
Congenital Visual Impairment and Progressive Microcephaly Due to Lysyl-Transfer Ribonucleic Acid (RNA) Synthetase (KARS) Mutations: The Expanding Phenotype of Aminoacyl-Transfer RNA Synthetase Mutations in Human Disease.
为氨酰-tRNA合成酶(ARS)1缺乏症的治疗奠定基础:表型特征及治疗效果综述
J Inherit Metab Dis. 2025 Mar;48(2):e70017. doi: 10.1002/jimd.70017.
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Childhood-onset focal epilepsy and acute para-infectious encephalopathy in a patient with biallelic QARS1 variants.一名携带双等位基因QARS1变异的患者出现儿童期起病的局灶性癫痫和急性感染后相关性脑病。
Neurol Sci. 2025 Mar;46(3):1395-1398. doi: 10.1007/s10072-024-07957-z. Epub 2024 Dec 24.
5
Aminoacyl-tRNA synthetase defects in neurological diseases.神经疾病中的氨酰-tRNA合成酶缺陷
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6
A Novel Homozygous Missense Mutation in the Gene: Expanding the Phenotype of Multisystem Disease.该基因中的一种新型纯合错义突变:扩展多系统疾病的表型
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4
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Clin Genet. 2014 Dec;86(6):508-9. doi: 10.1111/cge.12457. Epub 2014 Aug 15.
5
A newly recognized syndrome of severe growth deficiency, microcephaly, intellectual disability, and characteristic facial features.一种新发现的严重生长发育迟缓、小头畸形、智力残疾及特征性面部特征的综合征。
Eur J Med Genet. 2014 May-Jun;57(6):288-92. doi: 10.1016/j.ejmg.2014.03.010. Epub 2014 Apr 5.
6
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7
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8
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