HIF-1α activation mediates resistance to anti-angiogenic therapy in neuroblastoma xenografts

J Pediatr Surg. 2013 Jan;48(1):39-46. doi: 10.1016/j.jpedsurg.2012.10.016.

Abstract

Introduction: The anti-tumor activity of angiogenesis inhibitors is often limited by the development of resistance to these drugs. Here we establish HIF-1α as a major factor in the development of this resistance in neuroblastoma xenografts.

Methods: Neuroblastoma xenografts were established by injecting unmodified SKNAS or NB-1691 cells (2 × 10(6) cells), or cells in which HIF-1α expression had been knocked down with shRNA, into the retroperitoneal space of SCID mice. Treatment of established tumors included bevacizumab (5mg/kg q2wk), sunitinib (40 mg/kg qd), or topotecan (0.5mg/kg qd) alone or in combination for a total of two weeks.

Results: NB-1691 xenografts showed no difference in relative growth in HIF-1α knockdowns compared to control tumors (73.33 ± 7.90 vs 79.94 ± 6.15, p=0.528). However, HIF-1α knockdowns demonstrated relative final volumes that were significantly lower than unmodified tumors when both were treated with bevacizumab (35.88 ± 4.24 vs 53.57 ± 6.61, p=0.0544) or sunitinib (12.46 ± 2.59 vs 36.36 ± 4.82, p=0.0024). Monotherapy of unmodified xenografts with bevacizumab, sunitinib, or topotecan was largely ineffective. Relative final volumes of NB-1691 xenografts were significantly less in cohorts treated with sunitinib+topotecan (4.78 ± 0.77 vs 39.17 ± 2.44 [sunitinib alone], p=0.011) and bevacizumab+topotecan (13.63 ± 1.55 vs 48.16 ± 9.94 [bevacizumab alone], p=0.014).

Conclusion: Upregulation of HIF-1α appears to be a significant mechanism of resistance to antiangiogenic therapies in neuroblastoma. Suppressing HIF-1α with low-dose topotecan potentiates the effects of the antiangiogenic drugs in a mouse model.

Publication types

  • Evaluation Study

MeSH terms

  • Angiogenesis Inhibitors / administration & dosage
  • Animals
  • Antibodies, Monoclonal, Humanized / administration & dosage
  • Antineoplastic Combined Chemotherapy Protocols / therapeutic use*
  • Bevacizumab
  • Biomarkers, Tumor / metabolism*
  • Cell Line, Tumor
  • Drug Administration Schedule
  • Drug Resistance, Neoplasm / physiology*
  • Humans
  • Hypoxia-Inducible Factor 1, alpha Subunit / metabolism*
  • Indoles / administration & dosage
  • Injections, Intraperitoneal
  • Mice
  • Mice, SCID
  • Neuroblastoma / drug therapy*
  • Neuroblastoma / metabolism
  • Neuroblastoma / pathology
  • Pyrroles / administration & dosage
  • Reverse Transcriptase Polymerase Chain Reaction
  • Sunitinib
  • Topoisomerase I Inhibitors / administration & dosage
  • Topotecan / administration & dosage
  • Treatment Outcome
  • Tumor Burden

Substances

  • Angiogenesis Inhibitors
  • Antibodies, Monoclonal, Humanized
  • Biomarkers, Tumor
  • HIF1A protein, human
  • Hypoxia-Inducible Factor 1, alpha Subunit
  • Indoles
  • Pyrroles
  • Topoisomerase I Inhibitors
  • Bevacizumab
  • Topotecan
  • Sunitinib