Combined ALK and MDM2 inhibition increases antitumor activity and overcomes resistance in human ALK mutant neuroblastoma cell lines and xenograft models

Elife. 2017 Apr 20:6:e17137. doi: 10.7554/eLife.17137.

Abstract

The efficacy of ALK inhibitors in patients with ALK-mutant neuroblastoma is limited, highlighting the need to improve their effectiveness in these patients. To this end, we sought to develop a combination strategy to enhance the antitumor activity of ALK inhibitor monotherapy in human neuroblastoma cell lines and xenograft models expressing activated ALK. Herein, we report that combined inhibition of ALK and MDM2 induced a complementary set of anti-proliferative and pro-apoptotic proteins. Consequently, this combination treatment synergistically inhibited proliferation of TP53 wild-type neuroblastoma cells harboring ALK amplification or mutations in vitro, and resulted in complete and durable responses in neuroblastoma xenografts derived from these cells. We further demonstrate that concurrent inhibition of MDM2 and ALK was able to overcome ceritinib resistance conferred by MYCN upregulation in vitro and in vivo. Together, combined inhibition of ALK and MDM2 may provide an effective treatment for TP53 wild-type neuroblastoma with ALK aberrations.

Keywords: ALK; MDM2; Neuroblastoma; TP53; cancer biology; human.

MeSH terms

  • Anaplastic Lymphoma Kinase
  • Animals
  • Antineoplastic Agents / pharmacology
  • Antineoplastic Agents / therapeutic use*
  • Cell Line, Tumor
  • Disease Models, Animal
  • Drug Resistance, Neoplasm*
  • Heterografts
  • Humans
  • Mice, Nude
  • Neoplasm Transplantation
  • Neuroblastoma / drug therapy*
  • Neuroblastoma / pathology*
  • Proto-Oncogene Proteins c-mdm2 / antagonists & inhibitors*
  • Pyrimidines / pharmacology
  • Pyrimidines / therapeutic use
  • Receptor Protein-Tyrosine Kinases / antagonists & inhibitors*
  • Sulfones / pharmacology
  • Sulfones / therapeutic use

Substances

  • Antineoplastic Agents
  • Pyrimidines
  • Sulfones
  • MDM2 protein, human
  • Proto-Oncogene Proteins c-mdm2
  • ALK protein, human
  • Alk protein, mouse
  • Anaplastic Lymphoma Kinase
  • Receptor Protein-Tyrosine Kinases
  • ceritinib

Grants and funding

The research was funded by Novartis, Inc., where all authors were employees at the time the study was conducted. The authors declare no other competing financial interests.