The NO Answer for Autism Spectrum Disorder.

S-nitrosylation Shank3 autism spectrum disorder behavior contactin-associated protein-like2 nitric oxide

Journal

Advanced science (Weinheim, Baden-Wurttemberg, Germany)
ISSN: 2198-3844
Titre abrégé: Adv Sci (Weinh)
Pays: Germany
ID NLM: 101664569

Informations de publication

Date de publication:
08 2023
Historique:
revised: 19 04 2023
received: 07 10 2022
medline: 7 8 2023
pubmed: 22 5 2023
entrez: 22 5 2023
Statut: ppublish

Résumé

Autism spectrum disorders (ASDs) include a wide range of neurodevelopmental disorders. Several reports showed that mutations in different high-risk ASD genes lead to ASD. However, the underlying molecular mechanisms have not been deciphered. Recently, they reported a dramatic increase in nitric oxide (NO) levels in ASD mouse models. Here, they conducted a multidisciplinary study to investigate the role of NO in ASD. High levels of nitrosative stress biomarkers are found in both the Shank3 and Cntnap2 ASD mouse models. Pharmacological intervention with a neuronal NO synthase (nNOS) inhibitor in both models led to a reversal of the molecular, synaptic, and behavioral ASD-associated phenotypes. Importantly, treating iPSC-derived cortical neurons from patients with SHANK3 mutation with the nNOS inhibitor showed similar therapeutic effects. Clinically, they found a significant increase in nitrosative stress biomarkers in the plasma of low-functioning ASD patients. Bioinformatics of the SNO-proteome revealed that the complement system is enriched in ASD. This novel work reveals, for the first time, that NO plays a significant role in ASD. Their important findings will open novel directions to examine NO in diverse mutations on the spectrum as well as in other neurodevelopmental disorders. Finally, it suggests a novel strategy for effectively treating ASD.

Identifiants

pubmed: 37212048
doi: 10.1002/advs.202205783
pmc: PMC10401098
doi:

Substances chimiques

Nitric Oxide 31C4KY9ESH
Biomarkers 0
Shank3 protein, mouse 0
Microfilament Proteins 0
Nerve Tissue Proteins 0

Types de publication

Journal Article Research Support, U.S. Gov't, Non-P.H.S.

Langues

eng

Sous-ensembles de citation

IM

Pagination

e2205783

Subventions

Organisme : US Department of Defense (DoD)
Organisme : Israeli Science Foundation
Organisme : Eagles Autism Foundation
Organisme : National Institute of Psychobiology in Israel
Organisme : Israeli Council for Higher Education Maof Grant
Organisme : Berettler Centre for Research in Molecular Pharmacology and Therapeutics Grant
Organisme : Satell Family Foundation and the Neubauer Family Foundation

Informations de copyright

© 2023 The Authors. Advanced Science published by Wiley-VCH GmbH.

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Auteurs

Manish Kumar Tripathi (MK)

Institute for Drug Research, School of Pharmacy, Faculty of Medicine, The Hebrew University of Jerusalem, Jerusalem, 91120, Israel.

Shashank Kumar Ojha (SK)

Institute for Drug Research, School of Pharmacy, Faculty of Medicine, The Hebrew University of Jerusalem, Jerusalem, 91120, Israel.

Maryam Kartawy (M)

Institute for Drug Research, School of Pharmacy, Faculty of Medicine, The Hebrew University of Jerusalem, Jerusalem, 91120, Israel.

Wajeha Hamoudi (W)

Institute for Drug Research, School of Pharmacy, Faculty of Medicine, The Hebrew University of Jerusalem, Jerusalem, 91120, Israel.

Ashwani Choudhary (A)

Sagol Department of Neurobiology, Faculty of Natural Sciences, University of Haifa, Haifa, 31905, Israel.

Shani Stern (S)

Sagol Department of Neurobiology, Faculty of Natural Sciences, University of Haifa, Haifa, 31905, Israel.

Adi Aran (A)

Neuropediatric Unit, Shaare Zedek Medical Center, Jerusalem, 91031, Israel.
Faculty of Medicine, The Hebrew University of Jerusalem, Jerusalem, 91120, Israel.

Haitham Amal (H)

Institute for Drug Research, School of Pharmacy, Faculty of Medicine, The Hebrew University of Jerusalem, Jerusalem, 91120, Israel.

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