Literature DB >> 23479141

Muscle phenotypic variability in limb girdle muscular dystrophy 2 G.

Julia F Paim1, Ana Cotta, Antonio P Vargas, Monica M Navarro, Jaquelin Valicek, Elmano Carvalho, Antonio L da-Cunha, Estevão Plentz, Shelida V Braz, Reinaldo I Takata, Camila F Almeida, Mariz Vainzof.   

Abstract

Limb girdle muscular dystrophy type 2 G (LGMD2G) is caused by mutations in the telethonin gene. Only few families were described presenting this disease, and they are mainly Brazilians. Here, we identified one additional case carrying the same common c.157C > T mutation in the telethonin gene but with an atypical histopathological muscle pattern. In a female patient with a long duration of symptoms (46 years), muscle biopsy showed, in addition to telethonin deficiency, the presence of nemaline rods, type 1 fiber predominance, nuclear internalization, lobulated fibers, and mitochondrial paracrystalline inclusions. Her first clinical signs were identified at 8 years old, which include tiptoe walking, left lower limb deformity, and frequent falls. Ambulation loss occurred at 41 years old, and now, at 54 years old, she presented pelvic girdle atrophy, winging scapula, foot deformity with incapacity to perform ankle dorsiflexion, and absent tendon reflexes. The presence of nemaline bodies could be a secondary phenomenon, possibly associated with focal Z-line abnormalities of a long-standing disease. However, these new histopathological findings, characteristic of congenital myopathies, expand muscle phenotypic variability of telethoninopathy.

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Year:  2013        PMID: 23479141     DOI: 10.1007/s12031-013-9987-6

Source DB:  PubMed          Journal:  J Mol Neurosci        ISSN: 0895-8696            Impact factor:   3.444


  15 in total

1.  Autosomal dominant limb-girdle muscular dystrophy: a large kindred with evidence for anticipation.

Authors:  J Gamez; C Navarro; A L Andreu; J M Fernandez; L Palenzuela; S Tejeira; R Fernandez-Hojas; S Schwartz; C Karadimas; S DiMauro; M Hirano; C Cervera
Journal:  Neurology       Date:  2001-02-27       Impact factor: 9.910

2.  Adult nemaline myopathy with trabecular muscle fibers.

Authors:  Viktoriya S Irodenko; Han S Lee; Stephen J de Armond; Robert B Layzer
Journal:  Muscle Nerve       Date:  2009-06       Impact factor: 3.217

3.  Calpain 3 gene mutations: genetic and clinico-pathologic findings in limb-girdle muscular dystrophy.

Authors:  J Chae; N Minami; Y Jin; M Nakagawa; K Murayama; F Igarashi; I Nonaka
Journal:  Neuromuscul Disord       Date:  2001-09       Impact factor: 4.296

Review 4.  Telethonin-deficiency initially presenting as a congenital muscular dystrophy.

Authors:  Ana Ferreiro; Monica Mezmezian; Montse Olivé; Danielle Herlicoviez; Michel Fardeau; Pascale Richard; Norma Beatriz Romero
Journal:  Neuromuscul Disord       Date:  2011-05-06       Impact factor: 4.296

5.  Limb-girdle muscular dystrophy in a Portuguese patient caused by a mutation in the telethonin gene.

Authors:  L Negrão; A Matos; A Geraldo; O Rebelo
Journal:  Acta Myol       Date:  2010-07

6.  Transcriptional analysis of the titin cap gene.

Authors:  Shuang Zhang; Priya Londhe; Meiling Zhang; Judith K Davie
Journal:  Mol Genet Genomics       Date:  2011-02-09       Impact factor: 3.291

7.  Limb-girdle muscular dystrophy type 2G is caused by mutations in the gene encoding the sarcomeric protein telethonin.

Authors:  E S Moreira; T J Wiltshire; G Faulkner; A Nilforoushan; M Vainzof; O T Suzuki; G Valle; R Reeves; M Zatz; M R Passos-Bueno; D E Jenne
Journal:  Nat Genet       Date:  2000-02       Impact factor: 38.330

8.  Telethonin protein expression in neuromuscular disorders.

Authors:  Mariz Vainzof; Eloisa S Moreira; Oscar T Suzuki; Georgine Faulkner; Georgio Valle; Alan H Beggs; Olli Carpen; Alberto F Ribeiro; Edmar Zanoteli; Juliana Gurgel-Gianneti; Ana Maria Tsanaclis; Helga C A Silva; Maria Rita Passos-Bueno; Mayana Zatz
Journal:  Biochim Biophys Acta       Date:  2002-10-09

9.  The NH2 terminus of titin spans the Z-disc: its interaction with a novel 19-kD ligand (T-cap) is required for sarcomeric integrity.

Authors:  C C Gregorio; K Trombitás; T Centner; B Kolmerer; G Stier; K Kunke; K Suzuki; F Obermayr; B Herrmann; H Granzier; H Sorimachi; S Labeit
Journal:  J Cell Biol       Date:  1998-11-16       Impact factor: 10.539

10.  Telethonin, a novel sarcomeric protein of heart and skeletal muscle.

Authors:  G Valle; G Faulkner; A De Antoni; B Pacchioni; A Pallavicini; D Pandolfo; N Tiso; S Toppo; S Trevisan; G Lanfranchi
Journal:  FEBS Lett       Date:  1997-09-29       Impact factor: 4.124

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  3 in total

1.  Distal myopathy due to TCAP variants in four unrelated Chinese patients.

Authors:  Xiaoqing Lv; Fei Gao; Tingjun Dai; Dandan Zhao; Wei Jiang; Hongzhi Geng; Fuchen Liu; Pengfei Lin; Chuanzhu Yan
Journal:  Neurogenetics       Date:  2020-08-06       Impact factor: 2.660

2.  Limb girdle muscular dystrophy type 2G with myopathic-neurogenic motor unit potentials and a novel muscle image pattern.

Authors:  Ana Cotta; Julia Filardi Paim; Antonio Lopes da-Cunha-Junior; Rafael Xavier Neto; Simone Vilela Nunes; Monica Magalhaes Navarro; Jaquelin Valicek; Elmano Carvalho; Lydia U Yamamoto; Camila F Almeida; Shelida Vasconcelos Braz; Reinaldo Issao Takata; Mariz Vainzof
Journal:  BMC Clin Pathol       Date:  2014-10-04

3.  Tcap Deficiency in Zebrafish Leads to ROS Production and Mitophagy, and Idebenone Improves its Phenotypes.

Authors:  Xiaoqing Lv; Rui Zhang; Ling Xu; Guangyu Wang; Chuanzhu Yan; Pengfei Lin
Journal:  Front Cell Dev Biol       Date:  2022-03-15
  3 in total

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