WT1 loss attenuates the TP53-induced DNA damage response in T-cell acute lymphoblastic leukemia

Haematologica. 2018 Feb;103(2):266-277. doi: 10.3324/haematol.2017.170431. Epub 2017 Nov 23.

Abstract

Loss-of-function mutations and deletions in Wilms tumor 1 (WT1) gene are present in approximately 10% of T-cell acute lymphoblastic leukemia. Clinically, WT1 mutations are enriched in relapsed series and are associated to inferior relapse-free survival in thymic T-cell acute lymphoblastic leukemia cases. Here we demonstrate that WT1 plays a critical role in the response to DNA damage in T-cell leukemia. WT1 loss conferred resistance to DNA damaging agents and attenuated the transcriptional activation of important apoptotic regulators downstream of TP53 in TP53-competent MOLT4 T-leukemia cells but not in TP53-mutant T-cell acute lymphoblastic leukemia cell lines. Notably, WT1 loss positively affected the expression of the X-linked inhibitor of apoptosis protein, XIAP, and genetic or chemical inhibition with embelin (a XIAP inhibitor) significantly restored sensitivity to γ-radiation in both T-cell acute lymphoblastic leukemia cell lines and patient-derived xenografts. These results reveal an important role for the WT1 tumor suppressor gene in the response to DNA damage, and support the view that anti-XIAP targeted therapies could have a role in the treatment of WT1-mutant T-cell leukemia.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Cell Line, Tumor
  • DNA Damage / drug effects*
  • Dose-Response Relationship, Radiation
  • Gamma Rays
  • Heterografts
  • Humans
  • Inhibitor of Apoptosis Proteins / antagonists & inhibitors
  • Inhibitor of Apoptosis Proteins / physiology
  • Mice, SCID
  • Precursor T-Cell Lymphoblastic Leukemia-Lymphoma / genetics*
  • Precursor T-Cell Lymphoblastic Leukemia-Lymphoma / radiotherapy
  • Tumor Suppressor Protein p53 / drug effects*
  • Tumor Suppressor Protein p53 / physiology
  • WT1 Proteins / deficiency*
  • WT1 Proteins / physiology

Substances

  • Birc4 protein, mouse
  • Inhibitor of Apoptosis Proteins
  • TP53 protein, human
  • Tumor Suppressor Protein p53
  • WT1 Proteins