Literature DB >> 30987788

ACTA1-myopathy with prominent finger flexor weakness and rimmed vacuoles.

Teerin Liewluck1, Zhiyv Niu2, Steven A Moore3, Mohammad Alsharabati4, Margherita Milone5.   

Abstract

Actinopathy is a group of clinically and pathologically heterogeneous myopathies due to mutations in the skeletal muscle sarcomeric α-actin 1-encoding gene (ACTA1). Disease-onset spans from prenatal life to adulthood and weakness can preferentially affect proximal or distal muscles. Myopathological findings include a spectrum of structural abnormalities with nemaline rods being the most common. We report a daughter and father with prominent finger flexors and/or quadriceps involvement. Muscle biopsies revealed rimmed vacuoles in both patients, associated with type 1 fiber atrophy in the daughter, and nemaline rods in the father. Next generation sequencing identified a novel dominant ACTA1 variant, c.149G>A (p.Gly50Asp) in both individuals and no abnormal variants in vacuolar myopathy-associated genes. Our findings expand the clinico-pathological spectrum of actinopathy.
Copyright © 2019 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  ACTA1; Congenital fiber type disproportion; Finger flexor weakness; IBM; Inclusion body myositis; Nemaline myopathy; Rimmed vacuoles

Mesh:

Substances:

Year:  2019        PMID: 30987788      PMCID: PMC6812538          DOI: 10.1016/j.nmd.2019.02.012

Source DB:  PubMed          Journal:  Neuromuscul Disord        ISSN: 0960-8966            Impact factor:   4.296


  12 in total

1.  Association of a Novel ACTA1 Mutation With a Dominant Progressive Scapuloperoneal Myopathy in an Extended Family.

Authors:  Kristen Zukosky; Katherine Meilleur; Bryan J Traynor; Jahannaz Dastgir; Livija Medne; Marcella Devoto; James Collins; Jachinta Rooney; Yaqun Zou; Michele L Yang; J Raphael Gibbs; Markus Meier; Joerg Stetefeld; Richard S Finkel; Joachim Schessl; Lauren Elman; Kevin Felice; Toby A Ferguson; Ozge Ceyhan-Birsoy; Alan H Beggs; Gihan Tennekoon; Janel O Johnson; Carsten G Bönnemann
Journal:  JAMA Neurol       Date:  2015-06       Impact factor: 18.302

2.  Mutations and polymorphisms of the skeletal muscle alpha-actin gene (ACTA1).

Authors:  Nigel G Laing; Danielle E Dye; Carina Wallgren-Pettersson; Gabriele Richard; Nicole Monnier; Suzanne Lillis; Thomas L Winder; Hanns Lochmüller; Claudio Graziano; Stella Mitrani-Rosenbaum; Darren Twomey; John C Sparrow; Alan H Beggs; Kristen J Nowak
Journal:  Hum Mutat       Date:  2009-09       Impact factor: 4.878

3.  Novel presentation of Pompe disease: Inclusion-body myositis-like clinical phenotype.

Authors:  Sankar Bandyopadhyay; Matthew Wicklund; Charles S Specht
Journal:  Muscle Nerve       Date:  2015-06-03       Impact factor: 3.217

Review 4.  Skeletal muscle α-actin diseases (actinopathies): pathology and mechanisms.

Authors:  Kristen J Nowak; Gianina Ravenscroft; Nigel G Laing
Journal:  Acta Neuropathol       Date:  2012-07-24       Impact factor: 17.088

5.  Atypical presentation of GNE myopathy with asymmetric hand weakness.

Authors:  John Karl L de Dios; Joseph A Shrader; Galen O Joe; Jeffrey C McClean; Kayla Williams; Robert Evers; May Christine V Malicdan; Carla Ciccone; Ami Mankodi; Marjan Huizing; John C McKew; David A Bluemke; William A Gahl; Nuria Carrillo-Carrasco
Journal:  Neuromuscul Disord       Date:  2014-08-07       Impact factor: 4.296

Review 6.  Diagnosis and Management of Immune-Mediated Myopathies.

Authors:  Margherita Milone
Journal:  Mayo Clin Proc       Date:  2017-05       Impact factor: 7.616

7.  Autosomal dominant distal myopathy due to a novel ACTA1 mutation.

Authors:  Teerin Liewluck; Eric J Sorenson; Magdalena A Walkiewicz; Kandelaria M Rumilla; Margherita Milone
Journal:  Neuromuscul Disord       Date:  2017-05-05       Impact factor: 4.296

8.  Targeted sequencing and identification of genetic variants in sporadic inclusion body myositis.

Authors:  Conrad C Weihl; Robert H Baloh; Youjin Lee; Tsui-Fen Chou; Sara K Pittman; Glenn Lopate; Peggy Allred; Jennifer Jockel-Balsarotti; Alan Pestronk; Matthew B Harms
Journal:  Neuromuscul Disord       Date:  2015-01-06       Impact factor: 4.296

9.  Myotonic dystrophy type 2: molecular, diagnostic and clinical spectrum.

Authors:  J W Day; K Ricker; J F Jacobsen; L J Rasmussen; K A Dick; W Kress; C Schneider; M C Koch; G J Beilman; A R Harrison; J C Dalton; L P W Ranum
Journal:  Neurology       Date:  2003-02-25       Impact factor: 9.910

10.  188th ENMC International Workshop: Inclusion Body Myositis, 2-4 December 2011, Naarden, The Netherlands.

Authors:  M R Rose
Journal:  Neuromuscul Disord       Date:  2013-08-30       Impact factor: 4.296

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.