Calcium flux control by Pacs1-Wdr37 promotes lymphocyte quiescence and lymphoproliferative diseases

EMBO J. 2021 May 3;40(9):e104888. doi: 10.15252/embj.2020104888. Epub 2021 Feb 25.

Abstract

Endoplasmic reticulum (ER) calcium (Ca2+ ) stores are critical to proteostasis, intracellular signaling, and cellular bioenergetics. Through forward genetic screening in mice, we identified two members of a new complex, Pacs1 and Wdr37, which are required for normal ER Ca2+ handling in lymphocytes. Deletion of Pacs1 or Wdr37 caused peripheral lymphopenia that was linked to blunted Ca2+ release from the ER after antigen receptor stimulation. Pacs1-deficient cells showed diminished inositol triphosphate receptor expression together with increased ER and oxidative stress. Mature Pacs1-/- B cells proliferated and died in vivo under lymphocyte replete conditions, indicating spontaneous loss of cellular quiescence. Disruption of Pacs1-Wdr37 did not diminish adaptive immune responses, but potently suppressed lymphoproliferative disease models by forcing loss of quiescence. Thus, Pacs1-Wdr37 plays a critical role in stabilizing lymphocyte populations through ER Ca2+ handling and presents a new target for lymphoproliferative disease therapy.

Keywords: Pacs1; Wdr37; calcium homeostasis; lymphocyte quiescence; lymphoproliferative disease.

Publication types

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

MeSH terms

  • Animals
  • B-Lymphocytes / metabolism
  • Calcium Signaling
  • Disease Models, Animal
  • Endoplasmic Reticulum / metabolism*
  • Female
  • Gene Deletion*
  • HEK293 Cells
  • Humans
  • Lymphopenia / genetics*
  • Lymphopenia / metabolism
  • Lymphoproliferative Disorders / genetics*
  • Lymphoproliferative Disorders / metabolism
  • Male
  • Mice
  • NIH 3T3 Cells
  • Nuclear Proteins / genetics*
  • Nuclear Proteins / metabolism
  • Vesicular Transport Proteins / genetics*
  • Vesicular Transport Proteins / metabolism

Substances

  • Nuclear Proteins
  • Pacs1 protein, mouse
  • Vesicular Transport Proteins