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Record W2465376623 · doi:10.3389/fnbeh.2016.00136

Multiple Drug Treatments That Increase cAMP Signaling Restore Long-Term Memory and Aberrant Signaling in Fragile X Syndrome Models

2016· article· en· W2465376623 on OpenAlex
Catherine H. Choi, Brian P. Schoenfeld, Aaron Bell, Joseph Hinchey, Cory Rosenfelt, Michael Gertner, Sean Campbell, Danielle Emerson, Paul Hinchey, Maria Kollaros, Neal Ferrick, Daniel B. Chambers, Steven Langer, Steven Sust, Aatika Malik, Allison M. Terlizzi, David A. Liebelt, David Ferreiro, Ali Sharma, Eric Koenigsberg, Richard J. Choi, Natalia Louneva, Steven E. Arnold, Robert E. Featherstone, Steven J. Siegel, R. Suzanne Zukin, Thomas V. McDonald, François V. Bolduc, Thomas A. Jongens, Sean McBride

Why this work is in the frame

A frame that forgets how it found something cannot be audited. These are the routes that admitted this work.

affAt least one author lists a Canadian institution in the pinned OpenAlex snapshot.
fundA Canadian funder is recorded on the work.

Bibliographic record

VenueFrontiers in Behavioral Neuroscience · 2016
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicGenetics and Neurodevelopmental Disorders
Canadian institutionsUniversity of Alberta
FundersNational Institute of Neurological Disorders and StrokeNational Institute of General Medical SciencesAlbert Einstein College of Medicine, Yeshiva UniversityNational Institutes of HealthNational Institute of Mental HealthCanadian Child Health Clinician Scientist ProgramU.S. Department of DefenseNational Fragile X FoundationYeshiva UniversityUniversity of PennsylvaniaFRAXA Research FoundationDrexel UniversityAutism Speaks
KeywordsFragile X syndromeFMR1Autism spectrum disorderNeuroscienceHippocampusShort-term memoryAutismPsychologyCognitionBiologyFragile xWorking memoryGeneticsPsychiatryGene

Abstract

fetched live from OpenAlex

Fragile X is the most common monogenic disorder associated with intellectual disability (ID) and autism spectrum disorders (ASD). Additionally, many patients are afflicted with executive dysfunction, ADHD, seizure disorder and sleep disturbances. Fragile X is caused by loss of FMRP expression, which is encoded by the FMR1 gene. Both the fly and mouse models of fragile X are also based on having no functional protein expression of their respective FMR1 homologs. The fly model displays well defined cognitive impairments and structural brain defects and the mouse model, although having subtle behavioral defects, has robust electrophysiological phenotypes and provides a tool to do extensive biochemical analysis of select brain regions. Decreased cAMP signaling has been observed in samples from the fly and mouse models of fragile X as well as in samples derived from human patients. Indeed, we have previously demonstrated that strategies that increase cAMP signaling can rescue short term memory in the fly model and restore DHPG induced mGluR mediated long term depression (LTD) in the hippocampus to proper levels in the mouse model (McBride et al., 2005; Choi et al., 2011, 2015). Here, we demonstrate that the same three strategies used previously with the potential to be used clinically, lithium treatment, PDE-4 inhibitor treatment or mGluR antagonist treatment can rescue long term memory in the fly model and alter the cAMP signaling pathway in the hippocampus of the mouse model.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

Full frame distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.362
Threshold uncertainty score0.825

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0000.000

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.026
GPT teacher head0.251
Teacher spread0.226 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it