Emery-Dreifuss muscular dystrophy

被引:44
作者
Muchir, Antoine [1 ]
Worman, Howard J. [1 ]
机构
[1] Columbia Univ, Dept Med, Coll Phys & Surg, New York, NY 10032 USA
关键词
D O I
10.1007/s11910-007-0025-3
中图分类号
R74 [神经病学与精神病学];
学科分类号
摘要
Emery-Dreifuss muscular dystrophy (EDMD) is inherited in an X-linked or autosomal manner. X-linked EDMD is caused by mutations in EMD, which encodes an integral protein of the nuclear envelope inner membrane called emerin. Autosomally inherited EDMD is caused by mutations in LMNA, which encodes A-type nuclear lamins, intermediate filament proteins associated with inner nuclear membrane. Although the causative mutations have been described and mouse models have been created, the pathogenic processes by which mutations in genes encoding nuclear envelope proteins cause striated muscle abnormalities in EDMD remain obscure. Working hypotheses include effects on nuclear structural integrity, increased cellular susceptibility to mechanical stress damage, alterations in gene expression in response to nuclear envelope changes, and effects on cell proliferation and differentiation.
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页码:78 / 83
页数:6
相关论文
共 51 条
[1]   THE NUCLEAR LAMINA IS A MESHWORK OF INTERMEDIATE-TYPE FILAMENTS [J].
AEBI, U ;
COHN, J ;
BUHLE, L ;
GERACE, L .
NATURE, 1986, 323 (6088) :560-564
[2]   Mouse model carrying H222P-Lmna mutation develops muscular dystrophy and dilated cardiomyopathy similar to human striated muscle laminopathies [J].
Arimura, T ;
Helbling-Leclerc, A ;
Varnous, S ;
Niel, F ;
Lacène, E ;
Fromes, Y ;
Toussaint, M ;
Mura, AM ;
Keller, DI ;
Amthor, H ;
Isnard, R ;
Malissen, M ;
Schwartz, K ;
Bonne, G .
HUMAN MOLECULAR GENETICS, 2005, 14 (01) :155-169
[3]   High incidence of sudden death with conduction system and myocardial disease due to lamins A and C gene mutation [J].
Bécane, HM ;
Bonne, G ;
Varnous, S ;
Muchir, A ;
Ortega, V ;
Hammouda, E ;
Urtizberea, JA ;
Lavergne, T ;
Fardeau, M ;
Eymard, B ;
Weber, S ;
Schwartz, K ;
Duboc, D .
PACE-PACING AND CLINICAL ELECTROPHYSIOLOGY, 2000, 23 (11) :1661-1666
[4]   PROGRESSION OF CARDIAC DISEASE IN EMERY-DREIFUSS MUSCULAR-DYSTROPHY [J].
BIALER, MG ;
MCDANIEL, NL ;
KELLY, TE .
CLINICAL CARDIOLOGY, 1991, 14 (05) :411-416
[5]   IDENTIFICATION OF A NOVEL X-LINKED GENE RESPONSIBLE FOR EMERY-DREIFUSS MUSCULAR-DYSTROPHY [J].
BIONE, S ;
MAESTRINI, E ;
RIVELLA, S ;
MANCINI, M ;
REGIS, S ;
ROMEO, G ;
TONIOLO, D .
NATURE GENETICS, 1994, 8 (04) :323-327
[6]  
Bonne G, 2000, ANN NEUROL, V48, P170, DOI 10.1002/1531-8249(200008)48:2<170::AID-ANA6>3.0.CO
[7]  
2-J
[8]   Mutations in the gene encoding lamin A/C cause autosomal dominant Emery-Dreifuss muscular dystrophy [J].
Bonne, G ;
Di Barletta, MR ;
Varnous, S ;
Bécane, HM ;
Hammouda, EH ;
Merlini, L ;
Muntoni, F ;
Greenberg, CR ;
Gary, F ;
Urtizberea, JA ;
Duboc, D ;
Fardeau, M ;
Toniolo, D ;
Schwartz, K .
NATURE GENETICS, 1999, 21 (03) :285-288
[9]   Decreased mechanical stiffness in LMNA-/- cells is caused by defective nucleo-cytoskeletal integrity: implications for the development of laminopathies [J].
Broers, JLV ;
Peeters, EAG ;
Kuijpers, HJH ;
Endert, J ;
Bouten, CVC ;
Oomens, CWJ ;
Baaijens, FPT ;
Ramaekers, FCS .
HUMAN MOLECULAR GENETICS, 2004, 13 (21) :2567-2580
[10]  
Cestan R, 1902, NOUVELLE ICONOGRAPHI, V15, P38