Diverse Phenotypic Presentation of the Welander Distal Myopathy Founder Mutation, With Myopathy and Amyotrophic Lateral Sclerosis in the Same Family

J Clin Neuromuscul Dis. 2024 Sep 1;26(1):42-46. doi: 10.1097/CND.0000000000000501.

Abstract

Welander distal myopathy is a rare myopathy with prominent and early involvement of distal upper extremity muscles, prevalent in individuals of Scandinavian origin, and caused by a founder mutation in the cytotoxic granule-associated RNA-binding protein (T-cell intracellular antigen-1; TIA1), E384K. Different pathogenic variants in the TIA1 gene, distinct from the founder 1, have recently been associated with frontotemporal dementia and amyotrophic lateral sclerosis (ALS), suggesting that TIA1-related disorders belong to the group of multisystem proteinopathies. We describe the first case of a two-generation family with the founder E384K TIA1 mutation demonstrating phenotypic variability; the mother manifested as Welander myopathy, whereas 2 daughters manifested as ALS. No other genetic cause of ALS was found in 1 of the affected daughters. We also discuss the possible mechanisms explaining this pleotropic presentation of the founder mutation.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Amyotrophic Lateral Sclerosis* / genetics
  • Distal Myopathies* / diagnosis
  • Distal Myopathies* / genetics
  • Female
  • Founder Effect
  • Humans
  • Male
  • Middle Aged
  • Mutation*
  • Pedigree*
  • Phenotype*
  • T-Cell Intracellular Antigen-1 / genetics

Substances

  • T-Cell Intracellular Antigen-1
  • TIA1 protein, human