A Comprehensive Update on Late-Onset Pompe Disease

Biomolecules. 2023 Aug 22;13(9):1279. doi: 10.3390/biom13091279.

Abstract

Pompe disease (PD) is an autosomal recessive disorder caused by mutations in the GAA gene that lead to a deficiency in the acid alpha-glucosidase enzyme. Two clinical presentations are usually considered, named infantile-onset Pompe disease (IOPD) and late-onset Pompe disease (LOPD), which differ in age of onset, organ involvement, and severity of disease. Assessment of acid alpha-glucosidase activity on a dried blood spot is the first-line screening test, which needs to be confirmed by genetic analysis in case of suspected deficiency. LOPD is a multi-system disease, thus requiring a multidisciplinary approach for efficacious management. Enzyme replacement therapy (ERT), which was introduced over 15 years ago, changes the natural progression of the disease. However, it has limitations, including a reduction in efficacy over time and heterogeneous therapeutic responses among patients. Novel therapeutic approaches, such as gene therapy, are currently under study. We provide a comprehensive review of diagnostic advances in LOPD and a critical discussion about the advantages and limitations of current and future treatments.

Keywords: chaperone; diagnosis; enzyme replacement therapy; gene therapy; late-onset Pompe disease; multi-system disease; myopathy.

Publication types

  • Review

MeSH terms

  • Enzyme Replacement Therapy
  • Genetic Therapy
  • Glycogen Storage Disease Type II* / diagnosis
  • Glycogen Storage Disease Type II* / genetics
  • Glycogen Storage Disease Type II* / therapy
  • Humans
  • Mutation
  • alpha-Glucosidases / genetics
  • alpha-Glucosidases / therapeutic use

Substances

  • alpha-Glucosidases

Grants and funding

This research received no external funding.