The Dysferlinopathies Conundrum: Clinical Spectra, Disease Mechanism and Genetic Approaches for Treatments

Biomolecules. 2024 Feb 21;14(3):256. doi: 10.3390/biom14030256.

Abstract

Dysferlinopathies refer to a spectrum of muscular dystrophies that cause progressive muscle weakness and degeneration. They are caused by mutations in the DYSF gene, which encodes the dysferlin protein that is crucial for repairing muscle membranes. This review delves into the clinical spectra of dysferlinopathies, their molecular mechanisms, and the spectrum of emerging therapeutic strategies. We examine the phenotypic heterogeneity of dysferlinopathies, highlighting the incomplete understanding of genotype-phenotype correlations and discussing the implications of various DYSF mutations. In addition, we explore the potential of symptomatic, pharmacological, molecular, and genetic therapies in mitigating the disease's progression. We also consider the roles of diet and metabolism in managing dysferlinopathies, as well as the impact of clinical trials on treatment paradigms. Furthermore, we examine the utility of animal models in elucidating disease mechanisms. By culminating the complexities inherent in dysferlinopathies, this write up emphasizes the need for multidisciplinary approaches, precision medicine, and extensive collaboration in research and clinical trial design to advance our understanding and treatment of these challenging disorders.

Keywords: Miyoshi myopathy; distal myopathy with anterior tibial onset (DMAT); dysferlin; dysferlinopathy; exon skipping; genetic therapy; limb-girdle muscular dystrophy recessive type 2 (LGMDR2); membrane resealing; mini-dysferlin.

Publication types

  • Review

MeSH terms

  • Animals
  • Membrane Proteins / genetics
  • Muscle Proteins / genetics
  • Muscular Dystrophies* / genetics
  • Muscular Dystrophies, Limb-Girdle* / genetics
  • Muscular Dystrophies, Limb-Girdle* / metabolism
  • Muscular Dystrophies, Limb-Girdle* / therapy
  • Mutation

Substances

  • Muscle Proteins
  • Membrane Proteins

Supplementary concepts

  • Dysferlinopathy

Grants and funding

No specific grant support was received for this study. T.Y. is supported by the Muscular Dystrophy Canada, the Friends of Garrett Cumming Research Fund, the HM Toupin Neurological Science Research Fund, Canadian Institutes of Health Research (CIHR), the Canada Foundation for Innovation, Alberta Advanced Education and Technology, Alberta Innovates: Health Solutions (AIHS), Jesse’s Journey, and the Women and Children’s Health Research Institute (WCHRI), The Rare Disease Foundation, and the BC Children’s Hospital Foundation. S.A. is supported by scholarships from the Maternal and Child Health (MatCH) Program, the Alberta Innovates, the Women and Children’s Health Research Institute (WCHRI), the Andrew Stewart Memorial Graduate Prizes, and the Alberta Graduate Excellence Scholarships (AGES).