Disruption of the microbiota-gut-brain axis is a defining characteristic of the α-Gal A (-/0) mouse model of Fabry disease

Gut Microbes. 2023 Dec;15(2):2256045. doi: 10.1080/19490976.2023.2256045.

Abstract

Fabry disease (FD) is an X-linked metabolic disease caused by a deficiency in α-galactosidase A (α-Gal A) activity. This causes accumulation of glycosphingolipids, especially globotriaosylceramide (Gb3), in different cells and organs. Neuropathic pain and gastrointestinal (GI) symptoms, such as abdominal pain, nausea, diarrhea, constipation, and early satiety, are the most frequent symptoms reported by FD patients and severely affect their quality of life. It is generally accepted that Gb3 and lyso-Gb3 are involved in the symptoms; nevertheless, the origin of these symptoms is complex and multifactorial, and the exact mechanisms of pathogenesis are still poorly understood. Here, we used a murine model of FD, the male α-Gal A (-/0) mouse, to characterize functionality, behavior, and microbiota in an attempt to elucidate the microbiota-gut-brain axis at three different ages. We provided evidence of a diarrhea-like phenotype and visceral hypersensitivity in our FD model together with reduced locomotor activity and anxiety-like behavior. We also showed for the first time that symptomology was associated with early compositional and functional dysbiosis of the gut microbiota, paralleled by alterations in fecal short-chain fatty acid levels, which partly persisted with advancing age. Interestingly, most of the dysbiotic features suggested a disruption of gut homeostasis, possibly contributing to accelerated intestinal transit, visceral hypersensitivity, and impaired communication along the gut-brain axis.

Keywords: Fabry disease; gastrointestinal disorders; gut microbiota; gut-brain axis; short-chain fatty acids; visceral pain; α-Gal A null mice.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Brain-Gut Axis
  • Diarrhea
  • Disease Models, Animal
  • Dysbiosis
  • Fabry Disease*
  • Gastrointestinal Microbiome*
  • Male
  • Mice
  • Quality of Life

Grants and funding

The APC Microbiome Ireland is a research institute funded by Science Foundation Ireland (SFI) 12/RC/2273_P2, through the Irish Government’s National Development Plan. F.U. is in receipt of an APEX postdoctoral fellowship through a Marie Skłodowska-Curie Action (MSCA) COFUND programme under Marie Skłodowska-Curie grant agreement No 754535.