期刊
NEUROSCIENCE
卷 445, 期 -, 页码 12-30出版社
PERGAMON-ELSEVIER SCIENCE LTD
DOI: 10.1016/j.neuroscience.2020.07.034
关键词
intellectual disabilities; neurodevelopmental disorders; Drosophila melanogaster; cognition; learning and memory; genetics
资金
- KU Leuven Funds Opening the Future (Belgium)
- SNSF NCCR Synapsy [51NF40-158776]
- SNSF (Switzerland) [310030182651]
- Novartis Foundation for Medical Biological Researchand Canton Etat de Vaud (Switzerland)
- Fondazione Roma Terzo Pilastro Internazionale
- Associazione Italiana Sindrome X Fragile
- Autism Speaks Meixner Translational Postdoctoral Fellowship (USA)
- Autism Research Institute (USA)
- Fondation Sophie Afenduli (Switzerland)
Neurodevelopmental disorders (NDDs) include a large number of conditions such as Fragile X syndrome, autism spectrum disorders and Down syndrome, among others. They are characterized by limitations in adaptive and social behaviors, as well as intellectual disability (ID). Whole-exome and whole-genome sequencing studies have highlighted a large number of NDD/ID risk genes. To dissect the genetic causes and underlying biological pathways, in vivo experimental validation of the effects of these mutations is needed. The fruit fly, Drosophila melanogaster, is an ideal model to study NDDs, with highly tractable genetics, combined with simple behavioral and circuit assays, permitting rapid medium-throughput screening of NDD/ID risk genes. Here, we review studies where the use of well-established assays to study mechanisms of learning and memory in Drosophila has permitted insights into molecular mechanisms underlying IDs. We discuss how technologies in the fly model, combined with a high degree of molecular and physiological conservation between flies and mammals, highlight the Drosophila system as an ideal model to study neurodevelopmental disorders, from genetics to behavior. This article is part of a Special Issue entitled: Animal Models of Neurodevelopmental Disorders. (C) 2020 The Authors. Published by Elsevier Ltd on behalf of IBRO.
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