Non-invasive optoacoustic imaging of glycogen-storage and muscle degeneration in late-onset Pompe disease

Nat Commun. 2024 Sep 8;15(1):7843. doi: 10.1038/s41467-024-52143-6.

Abstract

Pompe disease (PD) is a rare autosomal recessive glycogen storage disorder that causes proximal muscle weakness and loss of respiratory function. While enzyme replacement therapy (ERT) is the only effective treatment, biomarkers for disease monitoring are scarce. Following ex vivo biomarker validation in phantom studies, we apply multispectral optoacoustic tomography (MSOT), a laser- and ultrasound-based non-invasive imaging approach, in a clinical trial (NCT05083806) to image the biceps muscles of 10 late-onset PD (LOPD) patients and 10 matched healthy controls. MSOT is compared with muscle magnetic resonance imaging (MRI), ultrasound, spirometry, muscle testing and quality of life scores. Next, results are validated in an independent LOPD patient cohort from a second clinical site. Our study demonstrates that MSOT enables imaging of subcellular disease pathology with increases in glycogen/water, collagen and lipid signals, providing higher sensitivity in detecting muscle degeneration than current methods. This translational approach suggests implementation in the complex care of these rare disease patients.

Publication types

  • Clinical Study

MeSH terms

  • Adult
  • Case-Control Studies
  • Female
  • Glycogen Storage Disease Type II* / diagnostic imaging
  • Glycogen Storage Disease Type II* / metabolism
  • Glycogen Storage Disease Type II* / pathology
  • Glycogen* / metabolism
  • Humans
  • Magnetic Resonance Imaging* / methods
  • Male
  • Middle Aged
  • Muscle, Skeletal / diagnostic imaging
  • Muscle, Skeletal / metabolism
  • Muscle, Skeletal / pathology
  • Phantoms, Imaging
  • Photoacoustic Techniques* / methods
  • Ultrasonography / methods

Substances

  • Glycogen