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Therapy Development for Microvillus Inclusion Disease using Patient-derived Enteroids.

Meri Kalashyan1, Krishnan Raghunathan1, Haley Oller1

  • 1Division of Gastroenterology, Hepatology and Nutrition, Boston Children's Hospital; Harvard Medical School, Boston, MA.

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Summary

Microvillus Inclusion Disease (MVID) patient enteroids show structural and functional defects. Notch inhibition with DAPT restored brush border structure and Na+/H+ exchange, offering therapeutic potential.

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Area of Science:

  • Gastroenterology and Hepatology
  • Cell Biology
  • Genetics

Background:

  • Microvillus Inclusion Disease (MVID) is a severe infantile enteropathy caused by MYO5B mutations, leading to malabsorption and requiring intensive care.
  • Patient-derived enteroids are valuable for studying monogenic epithelial disorders, but are scarce for MVID.

Approach:

  • Developed human enteroids from MVID patients with MYO5B loss-of-function variants to model the disease.
  • Utilized multiplex immunofluorescence, electrolyte transport assays, and transcriptomic analysis to investigate MVID pathology and therapeutic targets.
  • Investigated the effects of Notch signaling inhibition using DAPT on MVID enteroid structure and function.

Key Points:

  • MVID enteroids recapitulated in vivo structural changes and showed impaired Na+/H+ exchange but near-normal chloride secretion.
  • The anti-diarrheal drug Crofelemer inhibited agonist-mediated fluid secretion in MVID enteroids.
  • Notch inhibition with DAPT restored brush border structure and Na+/H+ exchange activity in MVID enteroids.

Conclusions:

  • Patient-derived enteroids are a powerful tool for understanding MVID pathogenesis and developing therapeutic strategies.
  • Notch signaling inhibition presents a promising therapeutic avenue for MVID by restoring enterocyte structure and function.
  • Identified SGK2 and NHERF3 as potential pathways involved in rescuing MVID cells.