Literature DB >> 28629962

Pharmacological inhibition of Anaplastic Lymphoma Kinase rescues spatial memory impairments in Neurofibromatosis 1 mutant mice.

Joseph B Weiss1, Sydney Weber2, Tessa Marzulla2, Jacob Raber3.   

Abstract

Heterozygous Neurofibromatosis 1 (NF1) loss of function mutations are found in 90% of patients with neurofibromatosis, a syndrome associated with disabling cognitive impairment. Drosophila studies have demonstrated a genetic interaction between Anaplastic Lymphoma Kinase (Alk) and NF1 in cognitive performance. In addition, pharmacologic inhibition of Alk improves cognitive performance in heterozygous NF1 mutant flies. In this study, we tested whether pharmacological inhibition of Alk in heterozygous NF1 mutant mice attenuates or rescues cognitive impairments. Cognitive impairment of spatial memory retention observed in heterozygous NF1 mutant mice was rescued by the Alk inhibitor. These data support the hypothesis that inhibition of Alk may cognitively benefit patients with Neurofibromatosis 1.
Copyright © 2017 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  Alk inhibitor; NF1; Neurofibromatosis; Spatial memory; Swim speed; Water maze

Mesh:

Substances:

Year:  2017        PMID: 28629962     DOI: 10.1016/j.bbr.2017.06.024

Source DB:  PubMed          Journal:  Behav Brain Res        ISSN: 0166-4328            Impact factor:   3.332


  7 in total

1.  Characterization of early communicative behavior in mouse models of neurofibromatosis type 1.

Authors:  Susan E Maloney; Krystal C Chandler; Corina Anastasaki; Michael A Rieger; David H Gutmann; Joseph D Dougherty
Journal:  Autism Res       Date:  2017-08-26       Impact factor: 5.216

Review 2.  Emerging therapeutic targets for neurofibromatosis type 1.

Authors:  James A Walker; Meena Upadhyaya
Journal:  Expert Opin Ther Targets       Date:  2018-05-07       Impact factor: 6.902

3.  Associative Learning Requires Neurofibromin to Modulate GABAergic Inputs to Drosophila Mushroom Bodies.

Authors:  Eirini-Maria Georganta; Anastasios Moressis; Efthimios M C Skoulakis
Journal:  J Neurosci       Date:  2021-05-10       Impact factor: 6.167

4.  Assessment of nociception and related quality-of-life measures in a porcine model of neurofibromatosis type 1.

Authors:  Rajesh Khanna; Aubin Moutal; Katherine A White; Aude Chefdeville; Pedro Negrao de Assis; Song Cai; Vicki J Swier; Shreya S Bellampalli; Marissa D Giunta; Benjamin W Darbro; Dawn E Quelle; Jessica C Sieren; Margaret R Wallace; Christopher S Rogers; David K Meyerholz; Jill M Weimer
Journal:  Pain       Date:  2019-11       Impact factor: 7.926

Review 5.  Neurofibromin and suppression of tumorigenesis: beyond the GAP.

Authors:  Juan Mo; Stefanie L Moye; Renee M McKay; Lu Q Le
Journal:  Oncogene       Date:  2022-01-23       Impact factor: 8.756

Review 6.  Mechanistic insights from animal models of neurofibromatosis type 1 cognitive impairment.

Authors:  Andrew H Miller; Mary C Halloran
Journal:  Dis Model Mech       Date:  2022-08-29       Impact factor: 5.732

7.  Patient-associated mutations in Drosophila Alk perturb neuronal differentiation and promote survival.

Authors:  Kathrin Pfeifer; Georg Wolfstetter; Vimala Anthonydhason; Tafheem Masudi; Badrul Arefin; Mats Bemark; Patricia Mendoza-Garcia; Ruth H Palmer
Journal:  Dis Model Mech       Date:  2022-08-16       Impact factor: 5.732

  7 in total

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