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Whole-exome sequencing revealed a novel homozygous missense variant in OSGEP gene: a case report of Galloway-Mowat syndrome in Iran.全外显子组测序揭示了 OSGEP 基因中的一个新型纯合错义变异:伊朗一例 Galloway-Mowat 综合征。
CEN Case Rep. 2023 Nov;12(4):374-377. doi: 10.1007/s13730-023-00775-w. Epub 2023 Mar 1.
2
Novel homozygous OSGEP gene pathogenic variants in two unrelated patients with Galloway-Mowat syndrome: case report and review of the literature.两例无亲缘关系的 Galloway-Mowat 综合征患者中新型纯合 OSGEP 基因致病性变异:病例报告及文献复习。
BMC Nephrol. 2019 Apr 11;20(1):126. doi: 10.1186/s12882-019-1317-y.
3
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Am J Med Genet A. 2016 Apr;170A(4):992-8. doi: 10.1002/ajmg.a.37533. Epub 2016 Jan 5.
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Novel variants in OSGEP leading to Galloway-Mowat syndrome by altering its subcellular localization.导致 Galloway-Mowat 综合征的 OSGEP 新型变异,通过改变其亚细胞定位。
Clin Chim Acta. 2021 Dec;523:297-303. doi: 10.1016/j.cca.2021.10.012. Epub 2021 Oct 16.
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Case Report: Novel compound heterozygous variants cause Galloway-Mowat syndrome.病例报告:新型复合杂合变异导致加洛韦-莫瓦特综合征。
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Galloway-mowat syndrome 3 (GAMOS3): a novel disease-causing variant in OSGEP gene and expansion of the clinical spectrum.加洛韦-莫瓦特综合征3型(GAMOS3):OSGEP基因中的一种新型致病变异及临床谱的扩展
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本文引用的文献

1
Novel variants in OSGEP leading to Galloway-Mowat syndrome by altering its subcellular localization.导致 Galloway-Mowat 综合征的 OSGEP 新型变异,通过改变其亚细胞定位。
Clin Chim Acta. 2021 Dec;523:297-303. doi: 10.1016/j.cca.2021.10.012. Epub 2021 Oct 16.
2
Novel homozygous OSGEP gene pathogenic variants in two unrelated patients with Galloway-Mowat syndrome: case report and review of the literature.两例无亲缘关系的 Galloway-Mowat 综合征患者中新型纯合 OSGEP 基因致病性变异:病例报告及文献复习。
BMC Nephrol. 2019 Apr 11;20(1):126. doi: 10.1186/s12882-019-1317-y.
3
Galloway-Mowat syndrome in Taiwan: OSGEP mutation and unique clinical phenotype.台湾的 Galloway-Mowat 综合征:OSGEP 突变与独特的临床表型。
Orphanet J Rare Dis. 2018 Dec 17;13(1):226. doi: 10.1186/s13023-018-0961-9.
4
A familial case of Galloway-Mowat syndrome due to a novel TP53RK mutation: a case report.一例因新型TP53RK突变导致的加洛韦-莫瓦特综合征家族病例:病例报告
BMC Med Genet. 2018 Jul 27;19(1):131. doi: 10.1186/s12881-018-0649-y.
5
Mutations in KEOPS-complex genes cause nephrotic syndrome with primary microcephaly.KEOPS复合体基因的突变会导致伴有原发性小头畸形的肾病综合征。
Nat Genet. 2017 Oct;49(10):1529-1538. doi: 10.1038/ng.3933. Epub 2017 Aug 14.
6
Extending the mutation spectrum for Galloway-Mowat syndrome to include homozygous missense mutations in the WDR73 gene.将加洛韦-莫瓦特综合征的突变谱扩展至包括WDR73基因中的纯合错义突变。
Am J Med Genet A. 2016 Apr;170A(4):992-8. doi: 10.1002/ajmg.a.37533. Epub 2016 Jan 5.
7
Infant Boy with Microcephaly Gastroesophageal Refl ux and Nephrotic Syndrome (Galloway-Mowat Syndrome): A Case Report.患有小头畸形、胃食管反流和肾病综合征(加洛韦 - 莫瓦特综合征)的男婴:一例报告
Middle East J Dig Dis. 2012 Jan;4(1):51-4.
8
Galloway-Mowat syndrome: an early-onset progressive encephalopathy with intractable epilepsy associated to renal impairment. Two novel cases and review of literature.高-莫瓦特综合征:一种与肾功能损害相关的早发性进行性脑病,伴有难治性癫痫。两例新病例及文献复习。
Seizure. 2010 Mar;19(2):132-5. doi: 10.1016/j.seizure.2009.12.002. Epub 2010 Jan 18.
9
Diagnostic dilemmas in four infants with nephrotic syndrome, microcephaly and severe developmental delay.四名患有肾病综合征、小头畸形和严重发育迟缓婴儿的诊断困境。
Clin Dysmorphol. 2001 Apr;10(2):115-21. doi: 10.1097/00019605-200104000-00008.
10
Congenital microcephaly with hiatus hernia and nephrotic syndrome in two sibs.两例同胞患先天性小头畸形伴食管裂孔疝和肾病综合征
J Med Genet. 1968 Dec;5(4):319-21. doi: 10.1136/jmg.5.4.319.

全外显子组测序揭示了 OSGEP 基因中的一个新型纯合错义变异:伊朗一例 Galloway-Mowat 综合征。

Whole-exome sequencing revealed a novel homozygous missense variant in OSGEP gene: a case report of Galloway-Mowat syndrome in Iran.

机构信息

Fetal Health Research Center, Hope Generation Foundation, Tehran, Iran.

Personalized Medicine and Genometabolomics Research Center, Hope Generation Foundation, Tehran, Iran.

出版信息

CEN Case Rep. 2023 Nov;12(4):374-377. doi: 10.1007/s13730-023-00775-w. Epub 2023 Mar 1.

DOI:10.1007/s13730-023-00775-w
PMID:36856752
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10620368/
Abstract

Galloway-Mowat syndrome is a rare autosomal-recessive genetic disorder that is characterized by variety of complications such as neurological abnormalities and early-onset progressive kidney disease. Studies have been shown that pathogenic mutations in genes that belong to the KEOPS complex lead to Galloway-Mowat syndrome. Several pathogenic mutations in OSGEP gene, a member of the KEOPS complex, have been detected in Galloway-Mowat syndrome. Here we describe a 12-year-old male with intellectual disability, poor speech, seizures, microcephaly, and nephrotic syndrome that were in favor of Galloway-Mowat syndrome, born to a healthy Iranian consanguineous parents. Extracted genomic DNA from blood sample was used to perform whole-exome sequencing in the patient. The mutational screening revealed a novel homozygote OSGEP gene missense variant. Our finding established whole-exome sequencing as a valuable technic for the detection of rare variants.

摘要

高威-莫瓦特综合征是一种罕见的常染色体隐性遗传疾病,其特征是多种并发症,如神经发育异常和早发性进行性肾病。研究表明,属于 KEOPS 复合物的基因突变会导致高威-莫瓦特综合征。在高威-莫瓦特综合征患者中已经检测到 KEOPS 复合物的一个成员 OSGEP 基因的几个致病性突变。在这里,我们描述了一名 12 岁男性,他患有智力残疾、言语障碍、癫痫、小头畸形和肾病综合征,这些都有利于高威-莫瓦特综合征,他出生于一对健康的伊朗近亲父母。从血样中提取基因组 DNA,用于对患者进行全外显子组测序。突变筛查显示了一种新型的 OSGEP 基因纯合错义变异。我们的发现确立了全外显子组测序作为一种检测罕见变异的有价值的技术。