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PMID: 10739764 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't

Different mutations in the LMNA gene cause autosomal dominant and autosomal recessive Emery-Dreifuss muscular dystrophy.

American journal of human genetics ·Vol. 66 ·No. 4 ·2000-04-00 ·Pages 1407-12

Raffaele Di Barletta M, Ricci E, Galluzzi G, Tonali P, Mora M, Morandi L, Romorini A, Voit T, Orstavik KH, Merlini L, Trevisan C, Biancalana V, Housmanowa-Petrusewicz I, Bione S, Ricotti R, Schwartz K, Bonne G, Toniolo D

Abstract

Emery-Dreifuss muscular dystrophy (EMD) is a condition characterized by the clinical triad of early-onset contractures, progressive weakness in humeroperoneal muscles, and cardiomyopathy with conduction block. The disease was described for the first time as an X-linked muscular dystrophy, but autosomal dominant and autosomal recessive forms were reported. The genes for X-linked EMD and autosomal dominant EMD (AD-EMD) were identified. We report here that heterozygote mutations in LMNA, the gene for AD-EMD, may cause diverse phenotypes ranging from typical EMD to no phenotypic effect. Our results show that LMNA mutations are also responsible for the recessive form of the disease. Our results give further support to the notion that different genetic forms of EMD have a common pathophysiological background. The distribution of the mutations in AD-EMD patients (in the tail and in the 2A rod domain) suggests that unique interactions between lamin A/C and other nuclear components exist that have an important role in cardiac and skeletal muscle function.

MeSH Terms
Adolescent Adult Aged Aged, 80 and over Amino Acid Substitution/genetics Base Sequence Child, Preschool DNA Mutational Analysis Female Genes, Dominant/genetics Genes, Recessive/genetics Genetic Predisposition to Disease/genetics Heterozygote Humans Infant Laminin/chemistry,genetics,metabolism Male Middle Aged Muscular Dystrophy, Emery-Dreifuss/genetics,metabolism,physiopathology Mutation/genetics Pedigree Penetrance Polymorphism, Single-Stranded Conformational Protein Structure, Tertiary
Chemicals
Laminin laminin A
Authors & Affiliations
18 authors, click to expand affiliations / ORCID
Raffaele Di Barletta M
Institute of Genetics, Biochemistry and Evolution-Consiglio Nazionale delle Ricerche, 27100 Pavia, Italy.
Ricci E
Galluzzi G
Tonali P
Mora M
Morandi L
Romorini A
Voit T
Orstavik K H
Merlini L
Trevisan C
Biancalana V
Housmanowa-Petrusewicz I
Bione S
Ricotti R
Schwartz K
Bonne G
Toniolo D
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30 references, click to expand
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Article Info
Journal
American journal of human genetics
Abbr.
Am J Hum Genet
ISSN
0002-9297
Published
2000-04-00
Epub
2000-00-16
Pages
1407-12
Language
English
Region
United States
NLM ID
0370475
PMCID
PMC1288205
Subset
IM
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