An inducible intestinal epithelial cell-specific NHE3 knockout mouse model mimicking congenital sodium diarrhea

Clin Sci (Lond). 2020 Apr 30;134(8):941-953. doi: 10.1042/CS20200065.

Abstract

The sodium-hydrogen exchanger isoform 3 (NHE3, SLC9A3) is abundantly expressed in the gastrointestinal tract and is proposed to play essential roles in Na+ and fluid absorption as well as acid-base homeostasis. Mutations in the SLC9A3 gene can cause congenital sodium diarrhea (CSD). However, understanding the precise role of intestinal NHE3 has been severely hampered due to the lack of a suitable animal model. To navigate this problem and better understand the role of intestinal NHE3, we generated a tamoxifen-inducible intestinal epithelial cell-specific NHE3 knockout mouse model (NHE3IEC-KO). Before tamoxifen administration, the phenotype and blood parameters of NHE3IEC-KO were unremarkable compared with control mice. After tamoxifen administration, NHE3IEC-KO mice have undetectable levels of NHE3 in the intestine. NHE3IEC-KO mice develop watery, alkaline diarrhea in combination with a swollen small intestine, cecum and colon. The persistent diarrhea results in higher fluid intake. After 3 weeks, NHE3IEC-KO mice show a ∼25% mortality rate. The contribution of intestinal NHE3 to acid-base and Na+ homeostasis under normal conditions becomes evident in NHE3IEC-KO mice that have metabolic acidosis, lower blood bicarbonate levels, hyponatremia and hyperkalemia associated with drastically elevated plasma aldosterone levels. These results demonstrate that intestinal NHE3 has a significant contribution to acid-base, Na+ and volume homeostasis, and lack of intestinal NHE3 has consequences on intestinal structural integrity. This mouse model mimics and explains the phenotype of individuals with CSD carrying SLC9A3 mutations.

Keywords: congenital sodium diarrhea; gastrointestinal physiology; homeostasis; renal physiology; sodium hydrogen exchange.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Abnormalities, Multiple / genetics*
  • Abnormalities, Multiple / metabolism
  • Abnormalities, Multiple / mortality
  • Abnormalities, Multiple / pathology
  • Animals
  • Diarrhea / congenital*
  • Diarrhea / genetics
  • Diarrhea / metabolism
  • Diarrhea / mortality
  • Diarrhea / pathology
  • Disease Models, Animal
  • Epithelial Cells / metabolism*
  • Female
  • Humans
  • Intestinal Mucosa / metabolism
  • Intestinal Mucosa / pathology
  • Male
  • Metabolism, Inborn Errors / genetics*
  • Metabolism, Inborn Errors / metabolism
  • Metabolism, Inborn Errors / mortality
  • Metabolism, Inborn Errors / pathology
  • Mice
  • Mice, Knockout
  • Mutation
  • Sodium-Hydrogen Exchanger 3 / genetics*
  • Sodium-Hydrogen Exchanger 3 / metabolism

Substances

  • Slc9a3 protein, mouse
  • Sodium-Hydrogen Exchanger 3

Supplementary concepts

  • Diarrhea 3, Secretory Sodium, Congenital