共 16 条
Novel duplication mutation in the patatin domain of adipose triglyceride lipase (PNPLA2) in neutral lipid storage disease with severe myopathy
被引:71
作者:

Akiyama, Masashi
论文数: 0 引用数: 0
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机构: Hokkaido Univ, Grad Sch Med, Dept Dermatol, Sapporo, Hokkaido 060, Japan

Sakai, Kaori
论文数: 0 引用数: 0
h-index: 0
机构: Hokkaido Univ, Grad Sch Med, Dept Dermatol, Sapporo, Hokkaido 060, Japan

Ogawa, Masaya
论文数: 0 引用数: 0
h-index: 0
机构: Hokkaido Univ, Grad Sch Med, Dept Dermatol, Sapporo, Hokkaido 060, Japan

McMillan, James R.
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h-index: 0
机构: Hokkaido Univ, Grad Sch Med, Dept Dermatol, Sapporo, Hokkaido 060, Japan

Sawamura, Daisuke
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机构: Hokkaido Univ, Grad Sch Med, Dept Dermatol, Sapporo, Hokkaido 060, Japan

Shimizu, Hiroshi
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h-index: 0
机构: Hokkaido Univ, Grad Sch Med, Dept Dermatol, Sapporo, Hokkaido 060, Japan
机构:
[1] Hokkaido Univ, Grad Sch Med, Dept Dermatol, Sapporo, Hokkaido 060, Japan
[2] Aomori Prefect Cent Hosp, Dept Neurol, Hirosaki, Aomori, Japan
[3] Hirosaki Univ, Grad Sch Med, Dept Dermatol, Hirosaki, Aomori, Japan
关键词:
ATGL;
myopathy;
patatin;
PNPLA2;
D O I:
10.1002/mus.20869
中图分类号:
R74 [神经病学与精神病学];
学科分类号:
摘要:
Recently, mutations in PNPLA2 encoding adipose triglyceride lipase (ATGL) were reported to underlie a neutral lipid storage disease (NLSD) subgroup characterized by mild myopathy and the absence of ichthyosis. In the present study a novel homozygous PNPLA2 mutation c.475_478dupCTCC (p.Gln160ProfsX19) in the patatin domain, the ATGL active site, was detected in a woman with NLSD and severe myopathy. The present results suggest that a premature truncation mutation in the patatin domain causes NLSD with severe myopathy.
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页码:856 / 859
页数:4
相关论文
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