Telomere Replication Stress Induced by POT1 Inactivation Accelerates Tumorigenesis

Cell Rep. 2016 Jun 7;15(10):2170-2184. doi: 10.1016/j.celrep.2016.05.008. Epub 2016 May 26.

Abstract

Genome sequencing studies have revealed a number of cancer-associated mutations in the telomere-binding factor POT1. Here, we show that when combined with p53 deficiency, depletion of murine POT1a in common lymphoid progenitor cells fosters genetic instability, accelerates the onset, and increases the severity of T cell lymphomas. In parallel, we examined human and mouse cells carrying POT1 mutations found in cutaneous T cell lymphoma (CTCL) patients. Inhibition of POT1 activates ATR-dependent DNA damage signaling and induces telomere fragility, replication fork stalling, and telomere elongation. Our data suggest that these phenotypes are linked to impaired CST (CTC1-STN1-TEN1) function at telomeres. Lastly, we show that proliferation of cancer cells lacking POT1 is enabled by the attenuation of the ATR kinase pathway. These results uncover a role for defective telomere replication during tumorigenesis.

Keywords: ATR; POT1; cutaneous T cell lymphoma; replication stress; telomere; thymic lymphoma.

MeSH terms

  • Amino Acid Sequence
  • Animals
  • Ataxia Telangiectasia Mutated Proteins / metabolism
  • Carcinogenesis / metabolism*
  • Carcinogenesis / pathology*
  • DNA Damage / genetics
  • DNA Repair / genetics
  • DNA Replication*
  • DNA-Binding Proteins / chemistry
  • DNA-Binding Proteins / genetics
  • DNA-Binding Proteins / metabolism*
  • Genetic Predisposition to Disease
  • Genomic Instability
  • Lymphoid Progenitor Cells / metabolism
  • Lymphoma, T-Cell, Cutaneous / genetics
  • Lymphoma, T-Cell, Cutaneous / immunology
  • Lymphoma, T-Cell, Cutaneous / pathology
  • Mice
  • Mutant Proteins / metabolism
  • Mutation / genetics
  • Protein Binding
  • Shelterin Complex
  • Stress, Physiological*
  • Telomere / metabolism*
  • Telomere-Binding Proteins
  • Thymus Gland / pathology
  • Tumor Suppressor Protein p53 / metabolism

Substances

  • DNA-Binding Proteins
  • Mutant Proteins
  • POT1 protein, mouse
  • Shelterin Complex
  • Telomere-Binding Proteins
  • Tumor Suppressor Protein p53
  • Atr protein, mouse
  • Ataxia Telangiectasia Mutated Proteins