The Neurobeachin-like 2 Protein Regulates Mast Cell Homeostasis

J Immunol. 2017 Oct 15;199(8):2948-2957. doi: 10.4049/jimmunol.1700556. Epub 2017 Sep 8.

Abstract

The neurobeachin-like 2 protein (Nbeal2) belongs to the family of beige and Chediak-Higashi (BEACH) domain proteins. Loss-of-function mutations in the human NBEAL2 gene or Nbeal2 deficiency in mice cause gray platelet syndrome, a bleeding disorder characterized by macrothrombocytopenia, splenomegaly, and paucity of α-granules in megakaryocytes and platelets. We found that in mast cells, Nbeal2 regulates the activation of the Shp1-STAT5 signaling axis and the composition of the c-Kit/STAT signalosome. Furthermore, Nbeal2 mediates granule formation and restricts the expression of the transcription factors, IRF8, GATA2, and MITF as well as of the cell-cycle inhibitor p27, which are essential for mast cell differentiation, proliferation, and cytokine production. These data demonstrate the relevance of Nbeal2 in mast cells above and beyond granule biosynthesis.

MeSH terms

  • Animals
  • Blood Proteins / genetics
  • Blood Proteins / metabolism*
  • Cell Cycle
  • Cells, Cultured
  • Cytoplasmic Granules / metabolism*
  • GATA2 Transcription Factor / genetics
  • GATA2 Transcription Factor / metabolism
  • Gray Platelet Syndrome / genetics*
  • Hemorrhage
  • Interferon Regulatory Factors / genetics
  • Interferon Regulatory Factors / metabolism
  • Mast Cells / physiology*
  • Megakaryocytes / physiology*
  • Mice
  • Mice, Knockout
  • Mutation / genetics
  • Protein Tyrosine Phosphatase, Non-Receptor Type 6 / metabolism
  • Proto-Oncogene Proteins c-kit / metabolism
  • STAT5 Transcription Factor / metabolism
  • Signal Transduction
  • Splenomegaly
  • Thrombocytopenia

Substances

  • Blood Proteins
  • GATA2 Transcription Factor
  • Gata2 protein, mouse
  • Interferon Regulatory Factors
  • Nbeal2 protein, mouse
  • STAT5 Transcription Factor
  • interferon regulatory factor-8
  • Proto-Oncogene Proteins c-kit
  • Protein Tyrosine Phosphatase, Non-Receptor Type 6