Primary immunodeficiencies caused by mutations in actin regulatory proteins

Immunol Rev. 2019 Jan;287(1):121-134. doi: 10.1111/imr.12716.

Abstract

The identification of patients with monogenic gene defects have illuminated the function of different proteins in the immune system, including proteins that regulate the actin cytoskeleton. Many of these actin regulatory proteins are exclusively expressed in leukocytes and regulate the formation and branching of actin filaments. Their absence or abnormal function leads to defects in immune cell shape, cellular projections, migration, and signaling. Through the study of patients' mutations and generation of mouse models that recapitulate the patients' phenotypes, our laboratory and others have gained a better understanding of the role these proteins play in cell biology and the underlying pathogenesis of immunodeficiencies and immune dysregulatory syndromes.

Keywords: WASp; actin; primary immunodeficiencies.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, N.I.H., Intramural
  • Review

MeSH terms

  • Actin Cytoskeleton / genetics
  • Actin Cytoskeleton / metabolism*
  • Animals
  • Cell Movement / genetics
  • Cell Surface Extensions / genetics
  • Disease Models, Animal
  • Humans
  • Immunologic Deficiency Syndromes / genetics*
  • Immunologic Deficiency Syndromes / immunology
  • Mice
  • Microfilament Proteins / genetics*
  • Mutation / genetics*
  • Phenotype
  • Signal Transduction / genetics

Substances

  • Microfilament Proteins