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  Estrogens suppress a behavioral phenotype in zebrafish mutants of the autism risk gene, CNTNAP2

Hoffman, E. J., Turner, K. J., Fernandez, J. M., Cifuentes, D., Ghosh, M., Ijaz, S., et al. (2016). Estrogens suppress a behavioral phenotype in zebrafish mutants of the autism risk gene, CNTNAP2. Neuron, 89(4), 725-733. doi:10.1016/j.neuron.2015.12.039.

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Hoffman, Ellen J., Autor
Turner, Katherine J., Autor
Fernandez, Joseph M., Autor
Cifuentes, Daniel, Autor
Ghosh, Marcus, Autor
Ijaz, Sundas, Autor
Jain, Roshan A., Autor
Kubo, Fumi1, Autor           
Bill, Brent R., Autor
Baier, Herwig1, Autor           
Granato, Michael, Autor
Barresi, Michael J. F., Autor
Wilson, Stephen W., Autor
Rihel, Jason, Autor
State, Matthew W., Autor
Giraldez, Antonio J., Autor
Affiliations:
1Department: Genes-Circuits-Behavior / Baier, MPI of Neurobiology, Max Planck Society, ou_1128545              

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Schlagwörter: EPILEPSY; EXPRESSION; DISORDERS; PATHWAYS; NEURONS; MODEL; MICE
 Zusammenfassung: Autism spectrum disorders (ASDs) are a group of devastating neurodevelopmental syndromes that affect up to 1 in 68 children. Despite advances in the identification of ASD risk genes, the mechanisms underlying ASDs remain unknown. Homozygous loss-of-function mutations in Contactin Associated Protein-like 2 (CNTNAP2) are strongly linked to ASDs. Here we investigate the function of Cntnap2 and undertake pharmacological screens to identify phenotypic suppressors. We find that zebrafish cntnap2 mutants display GABAergic deficits, particularly in the forebrain, and sensitivity to drug-induced seizures. High-throughput behavioral profiling identifies nighttime hyperactivity in cntnap2 mutants, while pharmacological testing reveals dysregulation of GABAergic and glutamatergic systems. Finally, we find that estrogen receptor agonists elicit a behavioral fingerprint anti-correlative to that of cntnap2 mutants and show that the phytoestrogen biochanin A specifically reverses the mutant behavioral phenotype. These results identify estrogenic compounds as phenotypic suppressors and illuminate novel pharmacological pathways with relevance to autism.

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Sprache(n): eng - English
 Datum: 2016-02-17
 Publikationsstatus: Erschienen
 Seiten: 9
 Ort, Verlag, Ausgabe: -
 Inhaltsverzeichnis: -
 Art der Begutachtung: Expertenbegutachtung
 Identifikatoren: ISI: 000373565200008
DOI: 10.1016/j.neuron.2015.12.039
 Art des Abschluß: -

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Titel: Neuron
Genre der Quelle: Zeitschrift
 Urheber:
Affiliations:
Ort, Verlag, Ausgabe: Cambridge, Mass. : Cell Press
Seiten: - Band / Heft: 89 (4) Artikelnummer: - Start- / Endseite: 725 - 733 Identifikator: ISSN: 0896-6273
CoNE: https://pure.mpg.de/cone/journals/resource/954925560565