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PMID: 29667529 Published · ppublish English Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, Non-P.H.S. Review

Emerging therapeutic targets for neurofibromatosis type 1.

Expert opinion on therapeutic targets ·Vol. 22 ·No. 5 ·2018-00-00 ·页码 419-437

Walker JA, Upadhyaya M

Abstract

Neurofibromatosis type 1 (NF1) is an autosomal dominantly inherited tumor predisposition syndrome with an incidence of one in 3000-4000 individuals with no currently effective therapies. The NF1 gene encodes neurofibromin, which functions as a negative regulator of RAS. NF1 is a chronic multisystem disorder affecting many different tissues. Due to cell-specific complexities of RAS signaling, therapeutic approaches for NF1 will likely have to focus on a particular tissue and manifestation of the disease. Areas covered: We discuss the multisystem nature of NF1 and the signaling pathways affected due to neurofibromin deficiency. We explore the cell-/tissue-specific molecular and cellular consequences of aberrant RAS signaling in NF1 and speculate on their potential as therapeutic targets for the disease. We discuss recent genomic, transcriptomic, and proteomic studies combined with molecular, cellular, and biochemical analyses which have identified several targets for specific NF1 manifestations. We also consider the possibility of patient-specific gene therapy approaches for NF1. Expert opinion: The emergence of NF1 genotype-phenotype correlations, characterization of cell-specific signaling pathways affected in NF1, identification of novel biomarkers, and the development of sophisticated animal models accurately reflecting human pathology will continue to provide opportunities to develop therapeutic approaches to combat this multisystem disorder.

Keywords
Neurofibromas RAS-MEK-ERK signaling RASopathies neurofibromatosis type 1 peripheral nerve sheath tumors
MeSH 主题词
Animals Biomarkers/metabolism Genes, Neurofibromatosis 1 Genetic Therapy/methods Genomics/methods Humans Molecular Targeted Therapy Neurofibromatosis 1/genetics,physiopathology,therapy Neurofibromin 1/genetics Proteomics/methods
化学物质
Biomarkers Neurofibromin 1
作者与单位
共 2 位作者,点击展开单位 / ORCID
Walker James A
a Center for Genomic Medicine , Massachusetts General Hospital, Harvard Medical School , Boston , MA , USA.
Upadhyaya Meena
b Division of Cancer and Genetics , Cardiff University , Cardiff , UK.
Article Info
Journal
Expert opinion on therapeutic targets
Abbr.
Expert Opin Ther Targets
ISSN
1744-7631
Published
2018-00-00
电子出版
2018-00-07
页码
419-437
Language
English
Country/Region
England
NLM ID
101127833
基金资助
NINDS NIH HHS · R21 NS096402 · United States
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