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本文引用的文献

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A new mutation of the fukutin gene causing late-onset limb girdle muscular dystrophy.一种导致晚发性肢带型肌营养不良的 FKRP 基因突变新类型。
Neuromuscul Disord. 2013 Jul;23(7):562-7. doi: 10.1016/j.nmd.2013.04.006. Epub 2013 Jun 6.
2
A Portuguese case of Fukuyama congenital muscular dystrophy caused by a multi-exonic duplication in the fukutin gene.葡萄牙一例福冈型先天性肌营养不良症,由福ukin 基因的多个外显子重复引起。
Neuromuscul Disord. 2013 Jul;23(7):557-61. doi: 10.1016/j.nmd.2013.03.005. Epub 2013 Apr 11.
3
Missense mutations in β-1,3-N-acetylglucosaminyltransferase 1 (B3GNT1) cause Walker-Warburg syndrome.β-1,3-N-乙酰氨基葡萄糖基转移酶 1(B3GNT1)中的错义突变导致沃克-沃伯格综合征。
Hum Mol Genet. 2013 May 1;22(9):1746-54. doi: 10.1093/hmg/ddt021. Epub 2013 Jan 28.
4
Exome sequencing and functional validation in zebrafish identify GTDC2 mutations as a cause of Walker-Warburg syndrome.外显子组测序和功能验证在斑马鱼中发现 GTDC2 突变是 Walker-Warburg 综合征的一个原因。
Am J Hum Genet. 2012 Sep 7;91(3):541-7. doi: 10.1016/j.ajhg.2012.07.009.
5
ISPD loss-of-function mutations disrupt dystroglycan O-mannosylation and cause Walker-Warburg syndrome.ISPD 功能丧失突变会破坏 dystroglycan 的 O-甘露糖基化,并导致 Walker-Warburg 综合征。
Nat Genet. 2012 May;44(5):575-80. doi: 10.1038/ng.2252.
6
Muscular dystrophies due to glycosylation defects: diagnosis and therapeutic strategies.由于糖基化缺陷引起的肌肉萎缩症:诊断和治疗策略。
Curr Opin Neurol. 2011 Oct;24(5):437-42. doi: 10.1097/WCO.0b013e32834a95e3.
7
A framework for variation discovery and genotyping using next-generation DNA sequencing data.利用下一代 DNA 测序数据进行变异发现和基因分型的框架。
Nat Genet. 2011 May;43(5):491-8. doi: 10.1038/ng.806. Epub 2011 Apr 10.
8
Fukutin mutations in non-Japanese patients with congenital muscular dystrophy: less severe mutations predominate in patients with a non-Walker-Warburg phenotype.非日本先天性肌营养不良症患者中的 Fukutin 突变:非 Walker-Warburg 表型患者中以较轻的突变为主。
Neuromuscul Disord. 2011 Jan;21(1):20-30. doi: 10.1016/j.nmd.2010.08.007. Epub 2010 Oct 18.
9
Deficiency of Dol-P-Man synthase subunit DPM3 bridges the congenital disorders of glycosylation with the dystroglycanopathies.Dol-P-Man合成酶亚基DPM3的缺乏将糖基化先天性疾病与肌营养不良糖蛋白病联系起来。
Am J Hum Genet. 2009 Jul;85(1):76-86. doi: 10.1016/j.ajhg.2009.06.006. Epub 2009 Jul 2.
10
HomozygosityMapper--an interactive approach to homozygosity mapping.纯合性定位器——一种用于纯合性定位的交互式方法。
Nucleic Acids Res. 2009 Jul;37(Web Server issue):W593-9. doi: 10.1093/nar/gkp369. Epub 2009 May 21.

一个埃及的 Fukuyama 先天性肌营养不良症伴小头畸形家系中发现 fukutin 基因突变。

Novel mutation in the fukutin gene in an Egyptian family with Fukuyama congenital muscular dystrophy and microcephaly.

机构信息

Clinical Genetics Department, Human Genetics and Genome Research Division, National Research Centre, Cairo 12311, Egypt.

Howard Hughes Medical Institute, University of California, San Diego, La Jolla, CA 92093, USA.

出版信息

Gene. 2014 Apr 15;539(2):279-82. doi: 10.1016/j.gene.2014.01.070. Epub 2014 Feb 13.

DOI:10.1016/j.gene.2014.01.070
PMID:24530477
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC5226855/
Abstract

Fukuyama-type congenital muscular dystrophy (FCMD, MIM#253800) is an autosomal recessive disorder characterized by severe muscular dystrophy associated with brain malformations. FCMD is the second most common form of muscular dystrophy after Duchenne muscular dystrophy and one of the most common autosomal recessive diseases among the Japanese population, and yet few patients outside of Japan had been reported with this disorder. We report the first known Egyptian patient with FCMD, established by clinical features of generalized weakness, pseudohypertrophy of calf muscles, progressive joint contractures, severe scoliosis, elevated serum creatine kinase level, myopathic electrodiagnostic changes, brain MRI with cobblestone complex, and mutation in the fukutin gene. In addition, our patient displayed primary microcephaly, not previously reported associated with fukutin mutations. Our results expand the geographic and clinical spectrum of fukutin mutations.

摘要

福山型先天性肌肉萎缩症 (FCMD,MIM#253800) 是一种常染色体隐性疾病,其特征为严重的肌肉萎缩症伴有脑部畸形。FCMD 是继杜兴氏肌肉营养不良症之后第二常见的肌肉萎缩症类型,也是日本人中最常见的常染色体隐性疾病之一,但在日本以外的地区,很少有报道过这种疾病。我们报告了首例已知的埃及 FCMD 患者,该患者具有全身性肌无力、小腿假性肥大、进行性关节挛缩、严重脊柱侧凸、血清肌酸激酶水平升高、肌病性电诊断改变、鹅卵石样脑 MRI 以及 fukutin 基因突变等临床表现。此外,我们的患者还表现出原发性小头畸形,这与 fukutin 基因突变以前未被报道过。我们的研究结果扩展了 fukutin 基因突变的地理和临床谱。