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Autism sensory dysfunction in an evolutionarily conserved system

There is increasing evidence for a strong genetic basis for autism, with many genetic models being developed in an attempt to replicate autistic symptoms in animals. However, current animal behaviour paradigms rarely match the social and cognitive behaviours exhibited by autistic individuals. Here,...

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Autores principales: Vilidaite, Greta, Norcia, Anthony M., West, Ryan J. H., Elliott, Christopher J. H., Pei, Francesca, Wade, Alex R., Baker, Daniel H.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: The Royal Society 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6304042/
https://www.ncbi.nlm.nih.gov/pubmed/30963913
http://dx.doi.org/10.1098/rspb.2018.2255
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author Vilidaite, Greta
Norcia, Anthony M.
West, Ryan J. H.
Elliott, Christopher J. H.
Pei, Francesca
Wade, Alex R.
Baker, Daniel H.
author_facet Vilidaite, Greta
Norcia, Anthony M.
West, Ryan J. H.
Elliott, Christopher J. H.
Pei, Francesca
Wade, Alex R.
Baker, Daniel H.
author_sort Vilidaite, Greta
collection PubMed
description There is increasing evidence for a strong genetic basis for autism, with many genetic models being developed in an attempt to replicate autistic symptoms in animals. However, current animal behaviour paradigms rarely match the social and cognitive behaviours exhibited by autistic individuals. Here, we instead assay another functional domain—sensory processing—known to be affected in autism to test a novel genetic autism model in Drosophila melanogaster. We show similar visual response alterations and a similar development trajectory in Nhe3 mutant flies (total n = 72) and in autistic human participants (total n = 154). We report a dissociation between first- and second-order electrophysiological visual responses to steady-state stimulation in adult mutant fruit flies that is strikingly similar to the response pattern in human adults with ASD as well as that of a large sample of neurotypical individuals with high numbers of autistic traits. We explain this as a genetically driven, selective signalling alteration in transient visual dynamics. In contrast to adults, autistic children show a decrease in the first-order response that is matched by the fruit fly model, suggesting that a compensatory change in processing occurs during development. Our results provide the first animal model of autism comprising a differential developmental phenotype in visual processing.
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spelling pubmed-63040422019-01-02 Autism sensory dysfunction in an evolutionarily conserved system Vilidaite, Greta Norcia, Anthony M. West, Ryan J. H. Elliott, Christopher J. H. Pei, Francesca Wade, Alex R. Baker, Daniel H. Proc Biol Sci Neuroscience and Cognition There is increasing evidence for a strong genetic basis for autism, with many genetic models being developed in an attempt to replicate autistic symptoms in animals. However, current animal behaviour paradigms rarely match the social and cognitive behaviours exhibited by autistic individuals. Here, we instead assay another functional domain—sensory processing—known to be affected in autism to test a novel genetic autism model in Drosophila melanogaster. We show similar visual response alterations and a similar development trajectory in Nhe3 mutant flies (total n = 72) and in autistic human participants (total n = 154). We report a dissociation between first- and second-order electrophysiological visual responses to steady-state stimulation in adult mutant fruit flies that is strikingly similar to the response pattern in human adults with ASD as well as that of a large sample of neurotypical individuals with high numbers of autistic traits. We explain this as a genetically driven, selective signalling alteration in transient visual dynamics. In contrast to adults, autistic children show a decrease in the first-order response that is matched by the fruit fly model, suggesting that a compensatory change in processing occurs during development. Our results provide the first animal model of autism comprising a differential developmental phenotype in visual processing. The Royal Society 2018-12-19 2018-12-12 /pmc/articles/PMC6304042/ /pubmed/30963913 http://dx.doi.org/10.1098/rspb.2018.2255 Text en © 2018 The Authors. http://creativecommons.org/licenses/by/4.0/ Published by the Royal Society under the terms of the Creative Commons Attribution License http://creativecommons.org/licenses/by/4.0/, which permits unrestricted use, provided the original author and source are credited.
spellingShingle Neuroscience and Cognition
Vilidaite, Greta
Norcia, Anthony M.
West, Ryan J. H.
Elliott, Christopher J. H.
Pei, Francesca
Wade, Alex R.
Baker, Daniel H.
Autism sensory dysfunction in an evolutionarily conserved system
title Autism sensory dysfunction in an evolutionarily conserved system
title_full Autism sensory dysfunction in an evolutionarily conserved system
title_fullStr Autism sensory dysfunction in an evolutionarily conserved system
title_full_unstemmed Autism sensory dysfunction in an evolutionarily conserved system
title_short Autism sensory dysfunction in an evolutionarily conserved system
title_sort autism sensory dysfunction in an evolutionarily conserved system
topic Neuroscience and Cognition
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6304042/
https://www.ncbi.nlm.nih.gov/pubmed/30963913
http://dx.doi.org/10.1098/rspb.2018.2255
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