Lysophosphatidic Acid Increases Maturation of Brush Borders and SGLT1 Activity in MYO5B-deficient Mice, a Model of Microvillus Inclusion Disease.

ABSTRACT:

Background & aim

Myosin VB (MYO5B) is an essential trafficking protein for membrane recycling in gastrointestinal epithelial cells. The inactivating mutations of MYO5B cause the congenital diarrheal disease, microvillus inclusion disease (MVID). MYO5B deficiency in mice causes mislocalization of SGLT1 and NHE3, but retained apical function of CFTR, resulting in malabsorption and secretory diarrhea. Activation of lysophosphatidic acid (LPA) receptors can improve diarrhea, but the effect of LPA on MVID symptoms is unclear. We investigated whether LPA administration can reduce the epithelial deficits in MYO5B-knockout mice.

Methods

Studies were conducted with tamoxifen-induced, intestine-specific knockout of MYO5B (VilCre^ERT2;Myo5b^flox/flox) and littermate

SUBMITTER: Kaji I 

PROVIDER: S-EPMC8240502 | biostudies-literature | 2020 Oct

REPOSITORIES: biostudies-literature