Literature DB >> 12403561

Molecular and cellular mechanisms underlying the cognitive deficits associated with neurofibromatosis 1.

Rui M Costa1, Alcino J Silva.   

Abstract

Neurofibromatosis 1 is one of the most common single-gene disorders affecting neurologic function in humans. Mutations in the NF1 gene cause abnormalities in cell growth and differentiation and lead to a variety of learning disabilities. Neurofibromin has several biochemical functions, such as Ras-guanosine triphosphatase activity, adenylate cyclase modulation, and microtubule binding, all of which could be critical for brain function. We review how studies in mouse models are helping to unravel the molecular and cellular mechanisms underlying cognitive deficits in neurofibromatosis 1. These studies suggest that the learning disabilities associated with neurofibromatosis 1 are caused by excessive Ras activity that leads to increased gamma-aminobutyric acid (GABA(A)) inhibition and to decreased long-term potentiation. These findings have brought us closer than ever to the development of possible treatments for the learning disabilities associated with neurofibromatosis 1.

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Year:  2002        PMID: 12403561     DOI: 10.1177/088307380201700813

Source DB:  PubMed          Journal:  J Child Neurol        ISSN: 0883-0738            Impact factor:   1.987


  26 in total

1.  ALTERED CALCIUM CURRENTS AND AXONAL GROWTH IN Nf1 HAPLOINSUFFICIENT MICE.

Authors:  Yuying Wang; Joel M Brittain; Sarah M Wilson; Cynthia M Hingtgen; Rajesh Khanna
Journal:  Transl Neurosci       Date:  2010-06       Impact factor: 1.757

Review 2.  Sensitization of Ion Channels Contributes to Central and Peripheral Dysfunction in Neurofibromatosis Type 1.

Authors:  Aubin Moutal; Erik T Dustrude; Rajesh Khanna
Journal:  Mol Neurobiol       Date:  2016-05-11       Impact factor: 5.590

3.  Perfusion single photon emission computed tomography in a mouse model of neurofibromatosis type 1: towards a biomarker of neurologic deficits.

Authors:  Ivayla Apostolova; Dagmara Niedzielska; Thorsten Derlin; Eva J Koziolek; Holger Amthauer; Benedikt Salmen; Jens Pahnke; Winfried Brenner; Victor F Mautner; Ralph Buchert
Journal:  J Cereb Blood Flow Metab       Date:  2015-03-18       Impact factor: 6.200

Review 4.  Alternative splicing of the neurofibromatosis type I pre-mRNA.

Authors:  Victoria A Barron; Hua Lou
Journal:  Biosci Rep       Date:  2012-04-01       Impact factor: 3.840

5.  The Fourth International Symposium on Genetic Disorders of the Ras/MAPK pathway.

Authors:  David A Stevenson; Lisa Schill; Lisa Schoyer; Brage S Andresen; Annette Bakker; Pinar Bayrak-Toydemir; Emma Burkitt-Wright; Kathryn Chatfield; Florent Elefteriou; Ype Elgersma; Michael J Fisher; David Franz; Bruce D Gelb; Anne Goriely; Karen W Gripp; Antonio Y Hardan; Kim M Keppler-Noreuil; Bronwyn Kerr; Bruce Korf; Chiara Leoni; Frank McCormick; Scott R Plotkin; Katherine A Rauen; Karlyne Reilly; Amy Roberts; Abby Sandler; Dawn Siegel; Karin Walsh; Brigitte C Widemann
Journal:  Am J Med Genet A       Date:  2016-05-07       Impact factor: 2.802

6.  Educational delay and attainment in persons with neurofibromatosis 1 in Denmark.

Authors:  Karoline Doser; Line Kenborg; Elisabeth Wreford Andersen; Pernille Envold Bidstrup; Anja Kroyer; Hanne Hove; John Østergaard; Sven Asger Sørensen; Christoffer Johansen; John Mulvihill; Jeanette Falck Winther; Susanne Oksbjerg Dalton
Journal:  Eur J Hum Genet       Date:  2019-02-28       Impact factor: 4.246

Review 7.  Impaired synaptic plasticity in RASopathies: a mini-review.

Authors:  Florian Mainberger; Susanne Langer; Volker Mall; Nikolai H Jung
Journal:  J Neural Transm (Vienna)       Date:  2016-08-26       Impact factor: 3.575

Review 8.  Neurofibromatosis type 1: new insights into neurocognitive issues.

Authors:  Maria T Acosta; Gerard A Gioia; Alcino J Silva
Journal:  Curr Neurol Neurosci Rep       Date:  2006-03       Impact factor: 5.081

Review 9.  The molecular and cellular biology of enhanced cognition.

Authors:  Yong-Seok Lee; Alcino J Silva
Journal:  Nat Rev Neurosci       Date:  2009-02       Impact factor: 34.870

10.  Cognitive and psychosocial phenotype of young children with neurofibromatosis-1.

Authors:  Bonita P Klein-Tasman; Kelly M Janke; Wen Luo; Christy L Casnar; Scott J Hunter; James Tonsgard; Pamela Trapane; Faye van der Fluit; Lorri A Kais
Journal:  J Int Neuropsychol Soc       Date:  2013-11-15       Impact factor: 2.892

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