Literature DB >> 34982307

Findings of limb-girdle muscular dystrophy R7 telethonin-related patients from a Chinese neuromuscular center.

Kun Huang1,2,3, Qiu-Xiang Li1,3, Hui-Qian Duan1,3, Yue-Bei Luo1,3, Fang-Fang Bi1,3, Huan Yang4,5.   

Abstract

Limb-girdle muscular dystrophy (LGMD) is a group of clinically and genetically heterogeneous neuromuscular disorders. LGMD-R7, which is caused by telethonin gene (TCAP) mutations, is one of the rarest forms of LGMD, and only a small number of LGMD-R7 cases have been described and mostly include patients from Brazil. A total of two LGMD-R7 patients were enrolled at a Chinese neuromuscular center. Demographic and clinical data were collected. Laboratory investigations and electromyography were performed. Routine and immunohistochemistry staining of muscle specimens was performed, and a next-generation sequencing panel array for genes associated with hereditary neuromuscular disorders was used for analysis. The patients exhibited predominant muscle weakness. Electromyography revealed myopathic changes. The muscle biopsy showed myopathic features, such as increased fiber size variation, muscle fiber atrophy and regeneration, slight hyperplasia of the connective tissue, and disarray of the myofibrillar network. Two patients were confirmed to have mutations in the open reading frame of TCAP by next-generation sequencing. One patient had compound heterozygous mutations, and the other patient harbored a novel homozygous mutation. Western blotting analysis of the skeletal muscle lysate confirmed the absence of telethonin in the patients. We described two LGMD-R7 patients presenting a classical LGMD phenotype and a novel homozygous TCAP mutation. Our research expands the spectrum of LGMD-R7 due to TCAP mutations based on patients from a Chinese neuromuscular center.
© 2021. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.

Entities:  

Keywords:  LGMD-R7; Limb-girdle muscular dystrophy (LGMD); Muscle pathology; Neuromuscular disorder; TCAP; Telethonin

Mesh:

Year:  2022        PMID: 34982307     DOI: 10.1007/s10048-021-00681-2

Source DB:  PubMed          Journal:  Neurogenetics        ISSN: 1364-6745            Impact factor:   2.660


  29 in total

1.  Rare diagnosis of telethoninopathy (LGMD2G) in a Turkish patient.

Authors:  Elena Ikenberg; Ivan Karin; Birgit Ertl-Wagner; Angela Abicht; Stefanie Bulst; Sabine Krause; Benedikt Schoser; Peter Reilich; Maggie C Walter
Journal:  Neuromuscul Disord       Date:  2017-06-01       Impact factor: 4.296

Review 2.  Untangling the complexity of limb-girdle muscular dystrophies.

Authors:  Teerin Liewluck; Margherita Milone
Journal:  Muscle Nerve       Date:  2018-02-07       Impact factor: 3.217

3.  Tcap gene mutations in hypertrophic cardiomyopathy and dilated cardiomyopathy.

Authors:  Takeharu Hayashi; Takuro Arimura; Manatsu Itoh-Satoh; Kazuo Ueda; Shigeru Hohda; Natsuko Inagaki; Megumi Takahashi; Hisae Hori; Michio Yasunami; Hirofumi Nishi; Yoshinori Koga; Hiroshi Nakamura; Masunori Matsuzaki; Bo Yoon Choi; Sung Won Bae; Cheol Woon You; Kyung Hoon Han; Jeong Euy Park; Ralph Knöll; Masahiko Hoshijima; Kenneth R Chien; Akinori Kimura
Journal:  J Am Coll Cardiol       Date:  2004-12-07       Impact factor: 24.094

4.  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

Review 5.  A new case of limb girdle muscular dystrophy 2G in a Greek patient, founder effect and review of the literature.

Authors:  Roberta Brusa; Francesca Magri; Dimitra Papadimitriou; Alessandra Govoni; Roberto Del Bo; Patrizia Ciscato; Marco Savarese; Claudia Cinnante; Maggie C Walter; Angela Abicht; Stefanie Bulst; Stefania Corti; Maurizio Moggio; Nereo Bresolin; Vincenzo Nigro; Giacomo Pietro Comi
Journal:  Neuromuscul Disord       Date:  2018-04-13       Impact factor: 4.296

Review 6.  Limb girdle muscular dystrophies: classification, clinical spectrum and emerging therapies.

Authors:  John Vissing
Journal:  Curr Opin Neurol       Date:  2016-10       Impact factor: 5.710

7.  The seventh form of autosomal recessive limb-girdle muscular dystrophy is mapped to 17q11-12.

Authors:  E S Moreira; M Vainzof; S K Marie; A L Sertié; M Zatz; M R Passos-Bueno
Journal:  Am J Hum Genet       Date:  1997-07       Impact factor: 11.025

8.  The genetics of dilated cardiomyopathy: a prioritized candidate gene study of LMNA, TNNT2, TCAP, and PLN.

Authors:  Marika Hirtle-Lewis; Katia Desbiens; Isabelle Ruel; Nicholas Rudzicz; Jacques Genest; James C Engert; Nadia Giannetti
Journal:  Clin Cardiol       Date:  2013-08-27       Impact factor: 2.882

9.  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

10.  Conserved expression of truncated telethonin in a patient with limb-girdle muscular dystrophy 2G.

Authors:  Rita Barresi; Charlotte Morris; Judith Hudson; Elizabeth Curtis; Clare Pickthall; Kate Bushby; Nicholas P Davies; Volker Straub
Journal:  Neuromuscul Disord       Date:  2014-12-24       Impact factor: 4.296

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  2 in total

1.  Clinicopathological-genetic features of congenital myasthenic syndrome from a Chinese neuromuscular centre.

Authors:  Kun Huang; Hui-Qian Duan; Qiu-Xiang Li; Yue-Bei Luo; Fang-Fang Bi; Huan Yang
Journal:  J Cell Mol Med       Date:  2022-06-06       Impact factor: 5.295

2.  Expanding the clinicopathological-genetic spectrum of glycogen storage disease type IXd by a Chinese neuromuscular center.

Authors:  Kun Huang; Hui-Qian Duan; Qiu-Xiang Li; Yue-Bei Luo; Fang-Fang Bi; Huan Yang
Journal:  Front Neurol       Date:  2022-08-11       Impact factor: 4.086

  2 in total

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