| Literature DB >> 28629962 |
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.Entities:
Keywords: Alk inhibitor; NF1; Neurofibromatosis; Spatial memory; Swim speed; Water maze
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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