Inactivation of c-Cbl reverses neonatal lethality and T cell developmental arrest of SLP-76-deficient mice

J Exp Med. 2004 Jul 5;200(1):25-34. doi: 10.1084/jem.20040262.

Abstract

c-Cbl is an adaptor protein that negatively regulates signal transduction events involved in thymic-positive selection. To further characterize the function of c-Cbl in T cell development, we analyzed the effect of c-Cbl inactivation in mice deficient in the scaffolding molecule SLP-76. SLP-76-deficient mice show a high frequency of neonatal lethality; and in surviving mice, T cell development is blocked at the DN3 stage. Inactivation of c-cbl completely reversed the neonatal lethality seen in SLP-76-deficient mice and partially reversed the T cell development arrest in these mice. SLP-76(-/-) Cbl(-/-) mice exhibited marked expansion of polarized T helper type (Th)1 and Th2 cell peripheral CD4(+) T cells, lymphoid infiltrates of parenchymal organs, and premature death. This rescue of T cell development is T cell receptor dependent because it does not occur in recombination activating gene 2(-/-) SLP-76(-/-) Cbl(-/-) triple knockout mice. Analysis of the signal transduction properties of SLP-76(-/-) Cbl(-/-) T cells reveals a novel SLP-76- and linker for activation of T cells-independent pathway of extracellular signal-regulated kinase activation, which is normally down-regulated by c-Cbl.

MeSH terms

  • Adaptor Proteins, Signal Transducing*
  • Animals
  • Animals, Newborn
  • Carrier Proteins / genetics
  • Carrier Proteins / metabolism
  • Cell Separation
  • DNA-Binding Proteins / genetics
  • DNA-Binding Proteins / metabolism
  • Flow Cytometry
  • Interferon-gamma / metabolism
  • Interleukin-2 / metabolism
  • Interleukin-4 / metabolism
  • Liver / cytology
  • Liver / metabolism
  • Lung / cytology
  • Lung / metabolism
  • Membrane Proteins / genetics
  • Membrane Proteins / metabolism
  • Mice
  • Mice, Knockout
  • Phosphoproteins / genetics
  • Phosphoproteins / metabolism*
  • Proto-Oncogene Proteins / genetics
  • Proto-Oncogene Proteins / metabolism*
  • Proto-Oncogene Proteins c-cbl
  • Receptors, Antigen, T-Cell / metabolism
  • Signal Transduction / physiology
  • Spleen / cytology
  • Survival Rate
  • T-Lymphocyte Subsets / physiology
  • T-Lymphocytes / physiology*
  • Ubiquitin-Protein Ligases / genetics
  • Ubiquitin-Protein Ligases / metabolism*

Substances

  • Adaptor Proteins, Signal Transducing
  • Carrier Proteins
  • DNA-Binding Proteins
  • Interleukin-2
  • Lat protein, mouse
  • Membrane Proteins
  • Phosphoproteins
  • Proto-Oncogene Proteins
  • Rag2 protein, mouse
  • Receptors, Antigen, T-Cell
  • SLP-76 signal Transducing adaptor proteins
  • V(D)J recombination activating protein 2
  • Interleukin-4
  • Interferon-gamma
  • Proto-Oncogene Proteins c-cbl
  • Ubiquitin-Protein Ligases
  • CBL protein, human
  • Cbl protein, mouse