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2
Expanding Phenotype of De Novo Mutations in GNAO1: Four New Cases and Review of Literature.GNAO1基因新生突变的扩展表型:4例新病例及文献复习
Neuropediatrics. 2017 Oct;48(5):371-377. doi: 10.1055/s-0037-1603977. Epub 2017 Jun 19.
3
Clinical Phenotype of De Novo Mutation: Case Report and Review of Literature.新发突变的临床表型:病例报告及文献综述
Child Neurol Open. 2015 May 5;2(2):2329048X15583717. doi: 10.1177/2329048X15583717. eCollection 2015 Apr-Jun.
4
Unexplained Early Infantile Epileptic Encephalopathy in Han Chinese Children: Next-Generation Sequencing and Phenotype Enriching.汉族儿童不明原因早发性婴儿癫痫性脑病:下一代测序和表型富集。
Sci Rep. 2017 Apr 7;7:46227. doi: 10.1038/srep46227.
5
ILAE classification of the epilepsies: Position paper of the ILAE Commission for Classification and Terminology.国际抗癫痫联盟癫痫分类:国际抗癫痫联盟分类与术语委员会立场文件
Epilepsia. 2017 Apr;58(4):512-521. doi: 10.1111/epi.13709. Epub 2017 Mar 8.
6
GNAO1-associated epileptic encephalopathy and movement disorders: c.607G>A variant represents a probable mutation hotspot with a distinct phenotype.GNAO1相关的癫痫性脑病和运动障碍:c.607G>A变异代表具有独特表型的可能突变热点。
Epileptic Disord. 2017 Mar 1;19(1):67-75. doi: 10.1684/epd.2017.0888.
7
Go is required for the release of IL-8 and TNF-α, but not degranulation in human mast cells.Go蛋白是人类肥大细胞释放白细胞介素-8和肿瘤坏死因子-α所必需的,但与脱颗粒无关。
Eur J Pharmacol. 2016 Jun 5;780:115-21. doi: 10.1016/j.ejphar.2016.03.038. Epub 2016 Mar 26.
8
The genetic landscape of the epileptic encephalopathies of infancy and childhood.婴儿和儿童癫痫性脑病的遗传特征。
Lancet Neurol. 2016 Mar;15(3):304-16. doi: 10.1016/S1474-4422(15)00250-1. Epub 2015 Nov 17.
9
Phenotypic spectrum of GNAO1 variants: epileptic encephalopathy to involuntary movements with severe developmental delay.GNAO1基因变异的表型谱:从癫痫性脑病到伴有严重发育迟缓的不自主运动。
Eur J Hum Genet. 2016 Jan;24(1):129-34. doi: 10.1038/ejhg.2015.92. Epub 2015 May 13.
10
De Novo mutations in GNAO1, encoding a Gαo subunit of heterotrimeric G proteins, cause epileptic encephalopathy.GNAO1 基因中的新生突变,该基因编码异三聚体 G 蛋白的 Gαo 亚基,可导致癫痫性脑病。
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[一名婴儿反复惊厥、肺部感染并伴有精神运动发育迟缓]

[Recurrent convulsion and pulmonary infection complicated by psychomotor retardation in an infant].

作者信息

Xiong Juan, Peng Jing, Duan Hao-Lin, Chen Chen, Wang Xiao-Le, Chen Shi-Meng, Yin Fei

机构信息

Department of Pediatrics, Xiangya Hospital, Central South University, Changsha 410008, China.

出版信息

Zhongguo Dang Dai Er Ke Za Zhi. 2018 Feb;20(2):154-157. doi: 10.7499/j.issn.1008-8830.2018.02.014.

DOI:10.7499/j.issn.1008-8830.2018.02.014
PMID:29429466
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC7389246/
Abstract

A 4-month-old girl developed convulsion in the neonatal period, which was focal motor seizures in the initial stage and later became spasm and tonic spasm. And the girl also had psychomotor retardation and recurrent pulmonary infection. Electroencephalography showed hypsarrhythmia, normal results were obtained from cranial magnetic resonance imaging, cerebrospinal fluid examination, and urine organic acid analysis, as well as the spectral analyses of blood ammonia, blood lactic acid, blood amino acids, and acylcarnitines. Gene detection revealed a de novo heterozygous mutation, c.607G>A (p.G203R) , in GNAO1. The girl was then diagnosed with GNAO1-associated early infantile epileptic encephalopathy (EIEE type 17). The seizures were well controlled by topiramate and vigabatrin, but there was no improvement in psychomotor development. She also suffered from recurrent pulmonary infection and died at the age of 12 months due to severe pneumonia. For children with unexplained early infantile epileptic encephalopathy, GNAO1 gene mutations should be considered and genetic tests should be performed as early as possible. Recurrent pulmonary infection should also be taken seriously.

摘要

一名4个月大的女孩在新生儿期出现惊厥,初期为局灶性运动性发作,后来发展为痉挛和强直性痉挛。该女孩还存在精神运动发育迟缓及反复肺部感染。脑电图显示高度失律,头颅磁共振成像、脑脊液检查、尿有机酸分析以及血氨、血乳酸、血氨基酸和酰基肉碱的谱分析结果均正常。基因检测发现GNAO1基因存在一个新发杂合突变,即c.607G>A(p.G203R)。该女孩随后被诊断为GNAO1相关的早期婴儿型癫痫性脑病(EIEE 17型)。托吡酯和氨己烯酸能很好地控制癫痫发作,但精神运动发育无改善。她还反复发生肺部感染,最终在12个月大时因重症肺炎死亡。对于不明原因的早期婴儿型癫痫性脑病患儿,应考虑GNAO1基因突变并尽早进行基因检测。反复肺部感染也应予以重视。