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PMID: 19084402 Published · ppublish English Case Reports Journal Article Research Support, Non-U.S. Gov't

A new phenotype of dysferlinopathy with congenital onset.

Neuromuscular disorders : NMD ·Vol. 19 ·No. 1 ·2009-01-00 ·Pages 21-5

Paradas C, González-Quereda L, De Luna N, Gallardo E, García-Consuegra I, Gómez H, Cabello A, Illa I, Gallano P

Abstract

We report two patients with a new phenotype of dysferlinopathy presenting as congenital muscular disease. Both patients showed weakness in proximal lower limbs and neck flexor muscles at birth. The presence of normal CK levels during the first years should be noted. Initial MRI showed no abnormalities but short-time-inversion-recovery (STIR) sequences revealed a striking myoedema in gastrocnemius and hamstring muscles at the age of 5. Muscle biopsy showed mild dystrophic features and the absence of dysferlin. Dysferlin gene (DYSF) analysis revealed a p.Ala927LeufsX21 mutation in a homozygous state in both siblings. This new phenotype widens the clinical spectrum of dysferlin myopathies.

MeSH Terms
Amino Acid Substitution/genetics Biopsy Child, Preschool Creatine Kinase/analysis,metabolism DNA Mutational Analysis Dysferlin Edema/genetics,pathology,physiopathology Female Genetic Markers/genetics Genetic Predisposition to Disease/genetics Genetic Testing Genotype Humans Magnetic Resonance Imaging Male Membrane Proteins/genetics Muscle Proteins/genetics Muscle Weakness/genetics,pathology,physiopathology Muscle, Skeletal/metabolism,pathology,physiopathology Muscular Diseases/congenital,genetics,physiopathology Mutation/genetics Phenotype
Chemicals
DYSF protein, human Dysferlin Genetic Markers Membrane Proteins Muscle Proteins Creatine Kinase
Authors & Affiliations
9 authors, click to expand affiliations / ORCID
Paradas C
Department of Neurology, Hospital de Valme, Sevilla, Spain.
González-Quereda L
De Luna N
Gallardo E
García-Consuegra I
Gómez H
Cabello A
Illa I
Gallano P
Article Info
Journal
Neuromuscular disorders : NMD
Abbr.
Neuromuscul Disord
ISSN
0960-8966
Published
2009-01-00
Epub
2008-00-11
Pages
21-5
Language
English
Region
England
NLM ID
9111470
Subset
IM
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