A new phenotype of dysferlinopathy with congenital onset


Por: Paradas, C, Gonzalez-Quereda, L, De Luna, N, Gallardo, E, Garcia-Consuegra, I, Gomez, H, Cabello, A, Illa, I, Gallano, P

Publicada: 1 ene 2009
Resumen:
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. (C) 2008 Elsevier B.V. All rights reserved.

Filiaciones:
Paradas, C:
 Hosp Valme, Dept Neurol, Seville, Spain

Gonzalez-Quereda, L:
 Univ Autonoma Barcelona, Dept Genet, Hosp Santa Creu & St Pau, Barcelona 08025, Spain

 CIBERER, Ctr Invest Red Enfermedades Raras, Barcelona, Spain

De Luna, N:
 Univ Autonoma Barcelona, Dept Neurol, Hosp Santa Creu & St Pau, Barcelona 08025, Spain

 CIBERNED, Ctr Invest Biomed Red Enfermedades Neurodegenerat, Barcelona, Spain

Gallardo, E:
 Univ Autonoma Barcelona, Dept Neurol, Hosp Santa Creu & St Pau, Barcelona 08025, Spain

 CIBERNED, Ctr Invest Biomed Red Enfermedades Neurodegenerat, Barcelona, Spain

Garcia-Consuegra, I:
 Hosp Univ 12 Octubre, Neuropathol Sect, Dept Pathol, Madrid, Spain

Gomez, H:
 Complejo Hosp Zafra Llerena, Dept Pediat, Badajoz, Spain

Cabello, A:
 Hosp Univ 12 Octubre, Neuropathol Sect, Dept Pathol, Madrid, Spain

Illa, I:
 Univ Autonoma Barcelona, Dept Neurol, Hosp Santa Creu & St Pau, Barcelona 08025, Spain

 CIBERNED, Ctr Invest Biomed Red Enfermedades Neurodegenerat, Barcelona, Spain

Gallano, P:
 Univ Autonoma Barcelona, Dept Genet, Hosp Santa Creu & St Pau, Barcelona 08025, Spain

 CIBERER, Ctr Invest Red Enfermedades Raras, Barcelona, Spain
ISSN: 09608966





NEUROMUSCULAR DISORDERS
Editorial
PERGAMON-ELSEVIER SCIENCE LTD, THE BOULEVARD, LANGFORD LANE, KIDLINGTON, OXFORD OX5 1GB, ENGLAND, Estados Unidos America
Tipo de documento: Article
Volumen: 19 Número: 1
Páginas: 21-25
WOS Id: 000263756400003
ID de PubMed: 19084402

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