Literature DB >> 15725589

Oculopharyngeal muscular dystrophy with PABPN1 mutation in a Chinese Malaysian woman.

Khean Jin Goh1, Kum Thong Wong, Ichizo Nishino, Narihiro Minami, Ikuya Nonaka.   

Abstract

Oculopharyngeal muscular dystrophy (OPMD) is an autosomal dominant disorder of middle age presenting as progressive dysphagia and eyelid ptosis, due to short expansions of the GCG trinucleotide repeat (from GCG6 to GCG8-13) in the polyadenylate binding-protein nuclear 1 (PABPN1) gene. OPMD is rarely seen in Asians and morphologically and/or genetically confirmed cases have been reported in Japanese kindreds only. We report a 64 year old Chinese-Malaysian woman who presented with progressive dysphagia and bilateral ptosis for about 6 years. Her mother and elder brother (both deceased) were believed to be affected. Muscle histopathology revealed angulated fibres with rimmed vacuoles. Genetic analysis showed repeat expansion in one allele to (GCG)9 while normal in the other (GCG)6. This is the first non-Japanese Asian family with genetically confirmed OPMD.

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Year:  2005        PMID: 15725589     DOI: 10.1016/j.nmd.2004.10.016

Source DB:  PubMed          Journal:  Neuromuscul Disord        ISSN: 0960-8966            Impact factor:   4.296


  4 in total

1.  Gene diagnosis of oculopharyngeal muscular dystrophy in a Chinese family by a GeneScan method.

Authors:  Pan You; Qilin Ma; Tao Tao
Journal:  J Clin Lab Anal       Date:  2010       Impact factor: 2.352

2.  Oculopharyngeal muscular dystrophy: phenotypic and genotypic studies in a Chinese population.

Authors:  Jingli Shan; Bin Chen; Pengfei Lin; Duoling Li; Yuebei Luo; Kunqian Ji; Jinfan Zheng; Yun Yuan; Chuanzhu Yan
Journal:  Neuromolecular Med       Date:  2014-10-05       Impact factor: 3.843

3.  A GCG expansion (GCG)₁₁ in polyadenylate-binding protein nuclear 1 gene caused oculopharyngeal muscular dystrophy in a Chinese family.

Authors:  Juan Ye; Huina Zhang; Yandan Zhou; Han Wu; Changjun Wang; Xin Shi
Journal:  Mol Vis       Date:  2011-05-25       Impact factor: 2.367

Review 4.  Recent Progress in Oculopharyngeal Muscular Dystrophy.

Authors:  Satoshi Yamashita
Journal:  J Clin Med       Date:  2021-03-29       Impact factor: 4.241

  4 in total

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