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Record W4416432712 · doi:10.1016/j.stemcr.2025.102718

Hypersynchronous iPSC-derived SHANK2 neuronal networks are rescued by mGluR5 agonism

2025· article· en· W4416432712 on OpenAlexafffund
Fraser P. McCready, Kartik S. Pradeepan, Milad Khaki, Wei Wei, Marta Guevara‐Ferrer, Nicole Matusiak, Feng Bi, Alina Piekna, Julio Martínez-Trujillo, James Ellis

Bibliographic record

VenueStem Cell Reports · 2025
Typearticle
Languageen
FieldNeuroscience
TopicAutism Spectrum Disorder Research
Canadian institutionsSickKids FoundationWestern UniversityHospital for Sick ChildrenUniversity of TorontoOntario Institute for Cancer Research
FundersNatural Sciences and Engineering Research Council of CanadaAutism SpeaksSimons Foundation Autism Research InitiativeCanada Research ChairsWestern UniversityCanada Foundation for InnovationOntario Research Foundation
KeywordsGlutamatergicBurstingAgonistPhenotypeNerve netNetwork topologyMetabotropic glutamate receptor

Abstract

fetched live from OpenAlex

Variants in the SHANK2 gene, linked to neurodevelopmental disorders like autism, were studied using human iPSC-derived neurons and multielectrode arrays. We compared two isogenic pairs of SHANK2 cell lines and found that SHANK2 networks exhibited a hyperconnectivity phenotype at the network level. These networks showed a significantly increased frequency and reduced duration of network burst events compared to controls. SHANK2 network activity was hypersynchronous, with stronger functional correlations between recording channels. Spikes within SHANK2 network bursts formed high-frequency trains, creating a distinctive burst shape. Calcium-dependent reverberating super bursts (RSBs) were common in control networks but rare in SHANK2 networks. Treatment with the group 1 mGluR agonist (S)-3,5-dihydroxyphenylglycine (DHPG) fully rescued SHANK2 network hypersynchrony, restored RSB detection, and improved network burst frequency and duration. The findings demonstrate that SHANK2 variants cause functional hyperconnectivity, which can be rescued by pharmacologically regulating glutamatergic neurotransmission.

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.

How this classification was reachedexpand

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 categoriesMeta-epidemiology (narrow)
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Bench or experimental · Consensus signal: Bench or experimental
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.056
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.001
Science and technology studies0.0010.000
Scholarly communication0.0000.000
Open science0.0010.000
Research integrity0.0000.001
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 head0.015
GPT teacher head0.252
Teacher spread0.237 · 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

Classification

machine, unvalidated

Machine predicted; a candidate call from one teacher head, not a consensus.

Study designBench or experimental
Domainnot available
GenreEmpirical

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

Quick stats

Citations1
Published2025
Admission routes2
Has abstractyes

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