Neurofibromatosis type 1 and optic pathway glioma: Molecular interplay and therapeutic insights

Pediatr Blood Cancer. 2018 Mar;65(3). doi: 10.1002/pbc.26838. Epub 2017 Oct 19.

Abstract

Children with neurofibromatosis type 1 (NF1) are predisposed to develop central nervous system neoplasms, the most common of which are low-grade gliomas (LGGs). The absence of human NF1 associated LGG-derived cell lines, coupled with an inability to generate patient-derived xenograft models, represents barriers to profile molecularly targeted therapies for these tumors. Thus, genetically engineered mouse models have been identified to evaluate the interplay between Nf1-deficient tumor cells and nonneoplastic stromal cells to evaluate potential therapies for these neoplasms. Future treatments might also consider targeting the nonneoplastic cells in NF1-LGGs to reduce tumor growth and neurologic morbidity in affected children.

Keywords: genetically engineered mouse models; neoplastic cells; neurofibromatosis 1; nonneoplastic stromal cells; optic pathway glioma; therapeutic insights.

Publication types

  • Research Support, N.I.H., Extramural
  • Review

MeSH terms

  • Adolescent
  • Animals
  • Cell Line, Tumor
  • Child
  • Child, Preschool
  • Female
  • Glioma* / genetics
  • Glioma* / metabolism
  • Glioma* / pathology
  • Glioma* / therapy
  • Humans
  • Infant
  • Infant, Newborn
  • Male
  • Mice
  • Neoplasms, Experimental* / genetics
  • Neoplasms, Experimental* / metabolism
  • Neoplasms, Experimental* / pathology
  • Neoplasms, Experimental* / therapy
  • Neurofibromatosis 1* / genetics
  • Neurofibromatosis 1* / metabolism
  • Neurofibromatosis 1* / pathology
  • Neurofibromatosis 1* / therapy
  • Neurofibromin 1 / deficiency*
  • Optic Nerve Neoplasms* / genetics
  • Optic Nerve Neoplasms* / metabolism
  • Optic Nerve Neoplasms* / pathology
  • Optic Nerve Neoplasms* / therapy
  • Xenograft Model Antitumor Assays / methods

Substances

  • NF1 protein, human
  • Neurofibromin 1