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DOI 10.7499/j.issn.1008-8830.2018.12.007

Медицинская библиотека · Zhongguo dang dai er ke za zhi = Chinese journal of contemporary pediatrics · 2018

[Clinical and genetic features of limb-girdle muscular dystrophy type 1B: a case report].

Yu Xin-Xiu, Zhong Jing-Zi, Guan Hong-Lin, Zhang Min, Lan Dan

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Аннотация

This article reports a case of limb-girdle muscular dystrophy type 1B (LGMD1B) caused by a novel splicing heterozygous mutation in the LMNA gene. The proband presented with progressive aggravation of weakness in walking. There was no atrophy of the scapular muscles and the lower-extremity proximal muscles, with normal muscle tension of the extremities, grade 4 muscle strength in the upper and lower extremities, and positive Gower sign. The level of creatine kinase was 779 U/L. Muscle hematoxylin-eosin staining showed muscular dystrophy, and there was no significant reduction in the expression of Lamin A protein. Second-generation sequencing revealed a novel splicing heterozygous mutation, c.810+2T>C, in the LMNA gene, while this locus was normal in his parents. GERP++RS software predicted that the mutation site was highly conservative. Human Splice Finder and Spliceman software predicted that the mutation might be a pathogenic mutation. ExPASy software predicted that the new amino acid sequence became shorter. There were two sequences of mRNA in the patient's muscle: one was the normal sequence, which accounted for 92.2%; the other was partial intron 4 retention, which was the abnormal splice variant accounting for 7.8%. LGMD1B is a type of autosomal dominant inherited myopathy caused by a mutation in the LMNA gene located on the autosomal 1q22. This study extends the mutation spectrum of the LMNA gene and provides help to the diagnosis of LGMD1B.

Для цитирования: Yu Xin-Xiu, Zhong Jing-Zi, Guan Hong-Lin, Zhang Min, Lan Dan. [Clinical and genetic features of limb-girdle muscular dystrophy type 1B: a case report].. Zhongguo dang dai er ke za zhi = Chinese journal of contemporary pediatrics. 2018. DOI: 10.7499/j.issn.1008-8830.2018.12.007.

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