| Literature DB >> 30987788 |
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.Entities:
Keywords: ACTA1; Congenital fiber type disproportion; Finger flexor weakness; IBM; Inclusion body myositis; Nemaline myopathy; Rimmed vacuoles
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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