Hypersensitivity to Distractors in Fragile X Syndrome from Loss of Modulation of Cortical VIP Interneurons.

Humans Male Female Animals Mice Vasoactive Intestinal Peptide Fragile X Syndrome Fragile X Mental Retardation Protein / genetics Activities of Daily Living Interneurons Mice, Knockout Disease Models, Animal
Fmr1 knockout VIP attention-deficit disorder autism spectrum disorders calcium imaging inhibition

Journal

The Journal of neuroscience : the official journal of the Society for Neuroscience
ISSN: 1529-2401
Titre abrégé: J Neurosci
Pays: United States
ID NLM: 8102140

Informations de publication

Date de publication:
29 Nov 2023
Historique:
received: 29 03 2023
revised: 11 08 2023
accepted: 14 09 2023
medline: 1 12 2023
pubmed: 11 10 2023
entrez: 10 10 2023
Statut: epublish

Résumé

Attention deficit is one of the most prominent and disabling symptoms in Fragile X syndrome (FXS). Hypersensitivity to sensory stimuli contributes to attention difficulties by overwhelming and/or distracting affected individuals, which disrupts activities of daily living at home and learning at school. We find that auditory or visual distractors selectively impair visual discrimination performance in humans and mice with FXS but not in typically developing controls. In both species, males and females were examined. Vasoactive intestinal polypeptide (VIP) neurons were significantly modulated by incorrect responses in the poststimulus period during early distractor trials in WT mice, consistent with their known role as error signals. Strikingly, however, VIP cells from

Identifiants

DOI: 10.1523/JNEUROSCI.0571-23.2023 PMID: 37816596 PMC: PMC10697397
pubmed: 37816596
pii: JNEUROSCI.0571-23.2023
doi: 10.1523/JNEUROSCI.0571-23.2023
pmc: PMC10697397
doi:

Substances chimiques

Vasoactive Intestinal Peptide 37221-79-7
Fragile X Mental Retardation Protein 139135-51-6
Fmr1 protein, mouse 0

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

8172-8188

Subventions

Organisme : NICHD NIH HHS
ID : R01 HD054453
Pays : United States
Organisme : NINDS NIH HHS
ID : R01 NS117597
Pays : United States

Commentaires et corrections

Type : UpdateOf

Informations de copyright

Copyright © 2023 Rahmatullah, Schmitt et al.

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Auteurs

Noorhan Rahmatullah (N)

Neuroscience Graduate Program, University of California, Riverside, Riverside, California 92521.
Department of Psychology, University of California, Riverside, Riverside, California 92521.

Lauren M Schmitt (LM)

Division of Behavioral Medicine and Clinical Psychology, Cincinnati Children's Hospital Medical Center, Cincinnati, OH 45229.
Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati OH 45267.

Lisa De Stefano (L)

Department of Psychiatry, Cincinnati Children's Hospital Medical Center, University of Cincinnati College of Medicine, Cincinnatti, Ohio 45267.

Sam Post (S)

Department of Psychology, University of California, Riverside, Riverside, California 92521.

Jessica Robledo (J)

Department of Psychology, University of California, Riverside, Riverside, California 92521.

Gunvant Chaudhari (G)

Department of Neurology, David Geffen School of Medicine, University of California, Los Angeles, Los Angeles, California 90095.

Ernest Pedapati (E)

Department of Psychiatry, Cincinnati Children's Hospital Medical Center, University of Cincinnati College of Medicine, Cincinnatti, Ohio 45267.
Department of Neurology, Cincinnati Children's Hospital Medical Center, University of Cincinnati College of Medicine, Cincinnatti, Ohio 45267.

Craig Erickson (C)

Department of Psychiatry, Cincinnati Children's Hospital Medical Center, University of Cincinnati College of Medicine, Cincinnatti, Ohio 45267.

Carlos Portera-Cailliau (C)

Department of Neurology, David Geffen School of Medicine, University of California, Los Angeles, Los Angeles, California 90095 anubhuti.goel@ucr.edu cpcailliau@mednet.ucla.edu.
Department of Neurobiology, David Geffen School of Medicine, University of California, Los Angeles, Los Angeles, California 90095.
ORCID: 0000-0001-5735-6380

Anubhuti Goel (A)

Neuroscience Graduate Program, University of California, Riverside, Riverside, California 92521 anubhuti.goel@ucr.edu cpcailliau@mednet.ucla.edu.
Department of Psychology, University of California, Riverside, Riverside, California 92521.
ORCID: 0000-0002-9514-0406

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Classifications MeSH

questionsmedicales.fr › Eucaryotes › Animaux › Chordés › Vertébrés › Mammifères › Eutheria › Primates › Haplorhini › Catarrhini › Hominidae › Humains › Hypersensitivity to Distractors in Fragile X...
questionsmedicales.fr › Eucaryotes › Animaux › Hypersensitivity to Distractors in Fragile X...
questionsmedicales.fr › Eucaryotes › Animaux › Chordés › Vertébrés › Mammifères › Eutheria › Rodentia › Muridae › Murinae › Souris › Hypersensitivity to Distractors in Fragile X...
questionsmedicales.fr › Acides aminés, peptides et protéines › Protéines › Protéines de tissu nerveux › Neuropeptides › Peptide vasoactif intestinal › Hypersensitivity to Distractors in Fragile X...
questionsmedicales.fr › Malformations et maladies congénitales, héréditaires et néonatales › Maladies génétiques congénitales › Maladies neurodégénératives héréditaires › Retard mental lié à l'X › Syndrome du chromosome X fragile › Hypersensitivity to Distractors in Fragile X...
questionsmedicales.fr › Acides aminés, peptides et protéines › Protéines › Nucléoprotéines › Protéines de liaison à l'ARN › Protéine du syndrome X fragile › Hypersensitivity to Distractors in Fragile X...
questionsmedicales.fr › Établissements, main d'oeuvre et services de soins de santé › Services de santé › Réadaptation › Activités de la vie quotidienne › Hypersensitivity to Distractors in Fragile X...
questionsmedicales.fr › Cellules › Neurones › Interneurones › Hypersensitivity to Distractors in Fragile X...
questionsmedicales.fr › Eucaryotes › Animaux › Chordés › Vertébrés › Mammifères › Eutheria › Rodentia › Muridae › Murinae › Souris › Souris transgéniques › Souris knockout › Hypersensitivity to Distractors in Fragile X...
questionsmedicales.fr › Techniques d'investigation › Modèles théoriques › Modèles biologiques › Modèles animaux de maladie humaine › Hypersensitivity to Distractors in Fragile X...