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PMID: 30447756 Published · ppublish English Journal Article Research Support, U.S. Gov't, Non-P.H.S. Review

Neurofibromatosis type 1 as a model system to study molecular mechanisms of autism spectrum disorder symptoms.

Progress in brain research ·Vol. 241 ·2018-00-00 ·页码 37-62

Molosh AI, Shekhar A

Abstract

Neurofibromatosis type 1 (NF1) is monogenic neurodevelopmental disorder caused by mutation of NF1 gene, which leads to increased susceptibility to various tumors formations. Additionally, majority of patients with NF1 are experience high incidence of cognitive deficits. Particularly, we review the growing number of reports demonstrated a higher incidence of autism spectrum disorder (ASD) in individuals with NF1. In this review we also discuss face validity of preclinical Nf1 mouse models. Then we describe discoveries from these animal models that have uncovered the deficiencies in the regulation of Ras and other intracellular pathways as critical mechanisms underlying the Nf1 cognitive problems. We also summarize and interpret recent preclinical and clinical studies that point toward potential pharmacological therapies for NF1 patients.

Keywords
Autism spectrum disorders Behavior Cognition Mouse models Neurofibromatosis type 1 PAK inhibitors
MeSH 主题词
Animals Autism Spectrum Disorder/drug therapy,genetics,metabolism,physiopathology Cognitive Dysfunction/drug therapy,genetics,metabolism,physiopathology Disease Models, Animal GABA Antagonists/pharmacology Humans Hydroxymethylglutaryl-CoA Reductase Inhibitors/pharmacology Mice Neurofibromatosis 1/drug therapy,genetics,metabolism,physiopathology Protein Kinase Inhibitors/pharmacology
化学物质
GABA Antagonists Hydroxymethylglutaryl-CoA Reductase Inhibitors Protein Kinase Inhibitors
作者与单位
共 2 位作者,点击展开单位 / ORCID
Molosh Andrei I
Department of Psychiatry, Institute of Psychiatric Research, IU School of Medicine, Indianapolis, IN, United States; Stark Neurosciences Research Institute, IU School of Medicine, Indianapolis, IN, United States. Electronic address: amolosh@iu.edu.
Shekhar Anantha
Department of Psychiatry, Institute of Psychiatric Research, IU School of Medicine, Indianapolis, IN, United States; Stark Neurosciences Research Institute, IU School of Medicine, Indianapolis, IN, United States; Department of Pharmacology & Toxicology, IU School of Medicine, Indianapolis, IN, United States; Indiana Clinical and Translational Institute, IU School of Medicine, Indianapolis, IN, United States.
Article Info
Journal
Progress in brain research
Abbr.
Prog Brain Res
ISSN
1875-7855
Corresponding email
Published
2018-00-00
电子出版
2018-00-25
页码
37-62
Language
English
Country/Region
Netherlands
NLM ID
0376441
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