Literature DB >> 30447756

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

Andrei I Molosh1, Anantha Shekhar2.   

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.
© 2018 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  Autism spectrum disorders; Behavior; Cognition; Mouse models; Neurofibromatosis type 1; PAK inhibitors

Mesh:

Substances:

Year:  2018        PMID: 30447756     DOI: 10.1016/bs.pbr.2018.09.014

Source DB:  PubMed          Journal:  Prog Brain Res        ISSN: 0079-6123            Impact factor:   2.453


  5 in total

1.  Social behaviors and contextual memory of Vcp mutant mice are sensitive to nutrition and can be ameliorated by amino acid supplementation.

Authors:  Tzyy-Nan Huang; Yu-Tzu Shih; Si-Cih Lin; Yi-Ping Hsueh
Journal:  iScience       Date:  2020-12-15

2.  Delineating the autistic phenotype in children with neurofibromatosis type 1.

Authors:  Anita K Chisholm; Kristina M Haebich; Natalie A Pride; Karin S Walsh; Francesca Lami; Alex Ure; Tiba Maloof; Amanda Brignell; Melissa Rouel; Yael Granader; Alice Maier; Belinda Barton; Hayley Darke; Gabriel Dabscheck; Vicki A Anderson; Katrina Williams; Kathryn N North; Jonathan M Payne
Journal:  Mol Autism       Date:  2022-01-04       Impact factor: 7.509

3.  RAS and beyond: the many faces of the neurofibromatosis type 1 protein.

Authors:  Corina Anastasaki; Paola Orozco; David H Gutmann
Journal:  Dis Model Mech       Date:  2022-02-21       Impact factor: 5.732

4.  Targeted Sequencing Detects Variants That May Contribute to the Risk of Neuropsychiatric Disorders.

Authors:  Jayant Mahadevan; Reeteka Sud; Ravi Kumar Nadella; Pulaparambil Vani; Anand G Subramaniam; Pradip Paul; Aparna Ganapathy; Ashraf U Mannan; Vijay Chandru; Biju Viswanath; Meera Purushottam; Sanjeev Jain
Journal:  Indian J Psychol Med       Date:  2021-03-25

5.  Guanfacine treatment improves ADHD phenotypes of impulsivity and hyperactivity in a neurofibromatosis type 1 mouse model.

Authors:  J L Lukkes; H P Drozd; S D Fitz; A I Molosh; D W Clapp; A Shekhar
Journal:  J Neurodev Disord       Date:  2020-01-15       Impact factor: 4.025

  5 in total

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