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A pilot open label, single dose trial of fenobam in adults with fragile X syndrome
Journal of Medical Genetics · 2009 · Vol. 46(4) · pp. 266–271
Elizabeth Berry‐Kravis✉(Rush University Medical Center)David Hessl(University of California Davis Medical Center)Sarah M. Coffey(University of California, Davis)Crystal Hervey(Rush University)Andrea Schneider(University of California, Davis)John M. Yuhas(University of California, Davis)John A. HutchisonMike SnapeMichael R. Tranfaglia(Fraxa Research Foundation)Danh V. Nguyen(University of California, Davis)Randi J. Hagerman(Society for Developmental and Behavioral Pediatrics)
Abstract
Clinically significant adverse effects were not identified in this study of single dose fenobam across the range of dosages utilised. The positive effects seen in animal models of FXS treated with fenobam or other mGluR5 antagonists, the apparent lack of clinically significant adverse effects, and the potential beneficial clinical effects seen in this pilot trial support further study of the compound in adults with FXS.
Genetics and Neurodevelopmental DisordersAutism Spectrum Disorder ResearchEpigenetics and DNA MethylationDosingMedicineAdverse effectPharmacokineticsFragile X syndromeDoseClinical trialCeiling effectInternal medicinePharmacology
MeSH terms
Administration, OralAdolescentAdultChromatography, LiquidFemaleFragile X SyndromeHumansImidazolesInhibition, PsychologicalMaleNeural InhibitionNeuropsychological TestsPilot ProjectsMass SpectrometryReceptors, Metabotropic Glutamate
Funding
- FRAXA Research Foundation
- National Institutes of Health
- National Center for Research Resources
Citations
410
FWCI
38.00
field-weighted impact
References
29
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100%
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References
Suppression of two major Fragile X Syndrome mouse model phenotypes by the mGluR5 antagonist MPEP
Neuropharmacology · 2005 · 523 citations
The mGluR theory of fragile X mental retardation
Trends in Neurosciences · 2004 · 1,587 citations
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