Loss of MYO5B Leads to Reductions in Na+ Absorption With Maintenance of CFTR-Dependent Cl- Secretion in Enterocytes

Gastroenterology
Amy Christine EngevikJames R Goldenring

Abstract

Inactivating mutations in MYO5B cause microvillus inclusion disease (MVID), but the physiological cause of the diarrhea associated with this disease is unclear. We investigated whether loss of MYO5B results in aberrant expression of apical enterocyte transporters. We studied alterations in apical membrane transporters in MYO5B-knockout mice, as well as mice with tamoxifen-inducible, intestine-specific disruption of Myo5b (VilCreERT2;Myo5bflox/flox mice) or those not given tamoxifen (controls). Intestinal tissues were collected from mice and analyzed by immunostaining, immunoelectron microscopy, or cultured enteroids were derived. Functions of brush border transporters in intestinal mucosa were measured in Ussing chambers. We obtained duodenal biopsy specimens from individuals with MVID and individuals without MVID (controls) and compared transporter distribution by immunocytochemistry. Compared to intestinal tissues from littermate controls, intestinal tissues from MYO5B-knockout mice had decreased apical localization of SLC9A3 (also called NHE3), SLC5A1 (also called SGLT1), aquaporin (AQP) 7, and sucrase isomaltase, and subapical localization of intestinal alkaline phosphatase and CDC42. However, CFTR was present on apical mem...Continue Reading

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Citations

Sep 29, 2019·The Journal of Cell Biology·Amy C EngevikJames R Goldenring
Dec 31, 2020·Journal of Clinical Medicine·Changsen LengSven C D van IJzendoorn
Jan 2, 2021·World Journal of Gastroenterology : WJG·Sriram AmirneniRodrigo M Florentino
Feb 7, 2021·Cellular and Molecular Gastroenterology and Hepatology·Amy C EngevikJames R Goldenring
Apr 4, 2021·Journal of Animal Physiology and Animal Nutrition·Hao LvAnshan Shan
Apr 17, 2021·Mucosal Immunology·Fabienne Charbit-HenrionNadine Cerf-Bensussan
Aug 11, 2020·Gastroenterology·Shabnam Abtahi, Jerrold R Turner
Jul 2, 2021·Cellular and Molecular Gastroenterology and Hepatology·Barun DasMamata Sivagnanam
Jul 2, 2021·JCI Insight·Izumi KajiJames R Goldenring
Nov 24, 2021·Liver International : Official Journal of the International Association for the Study of the Liver·Li WangJian-She Wang

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