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Record W4399350501 · doi:10.1136/bmjopen-2023-080746

European Autism GEnomics Registry (EAGER): protocol for a multicentre cohort study and registry

2024· article· en· W4399350501 on OpenAlexaff
Madeleine Bloomfield, Alexandra Lautarescu, Síofra Heraty, Sarah N. Douglas, Pierre Violland, Roderik Plas, Anjuli Ghosh, Katrien Van den Bosch, Eliza Eaton, Michael Absoud, Roberta Battini, Nadia Bolshakova, Sven Bölte, Paolo Bonanni, Jacqueline Borg, Sara Calderoni, Rosa Calvo, Miguel Castelo‐Branco, Josefina Castro‐Fornieles, Pilar Caro, Freddy Cliquet, Alberto Danieli, Richard Delorme, Maurizio Elia, Maja Hempel, Claire S. Leblond, Nuno Madeira, Gráinne McAlonan, Roberta Milone, Ciara J. Molloy, Susana Mouga, Virginia Montiel, Ana Pina Rodrigues, Christian P. Schaaf, Mercedes Serrano, Kristiina Tammimies, Charlotte Tye, Federico Vigevano, Guiomar Oliveira, Benedetta Mazzone, Cara O’Neill, J. W. PENDER, Verena Romero, Julian Tillmann, Bethany Oakley, Declan Murphy, Louise Gallagher, Thomas Bourgeron, Chris Chatham, Tony Charman

Bibliographic record

VenueBMJ Open · 2024
Typearticle
Languageen
FieldNeuroscience
TopicAutism Spectrum Disorder Research
Canadian institutionsUniversity of TorontoSickKids FoundationCentre for Addiction and Mental Health
FundersMedical Research CouncilNIHR Maudsley Biomedical Research CentreInnovative Medicines InitiativeEuropean CommissionVetenskapsrådetMinistero della SaluteTuberous Sclerosis AssociationEpilepsy Research UKNational Institute for Health and Care ResearchKing's College LondonUK Research and InnovationGovernment of the United KingdomHORIZON EUROPE Framework ProgrammeAgence Nationale de la RechercheEuropean Federation of Pharmaceutical Industries and AssociationsAutism SpeaksSimons Foundation Autism Research Initiative
KeywordsMedicineProtocol (science)Patient registryCohort studyFamily medicineEpidemiologyAutismCohortGenomicsMEDLINEDisease registryPediatricsAlternative medicinePathologyPsychiatryGenomeGenetics

Abstract

fetched live from OpenAlex

INTRODUCTION: Autism is a common neurodevelopmental condition with a complex genetic aetiology that includes contributions from monogenic and polygenic factors. Many autistic people have unmet healthcare needs that could be served by genomics-informed research and clinical trials. The primary aim of the European Autism GEnomics Registry (EAGER) is to establish a registry of participants with a diagnosis of autism or an associated rare genetic condition who have undergone whole-genome sequencing. The registry can facilitate recruitment for future clinical trials and research studies, based on genetic, clinical and phenotypic profiles, as well as participant preferences. The secondary aim of EAGER is to investigate the association between mental and physical health characteristics and participants' genetic profiles. METHODS AND ANALYSIS: EAGER is a European multisite cohort study and registry and is part of the AIMS-2-TRIALS consortium. EAGER was developed with input from the AIMS-2-TRIALS Autism Representatives and representatives from the rare genetic conditions community. 1500 participants with a diagnosis of autism or an associated rare genetic condition will be recruited at 13 sites across 8 countries. Participants will be given a blood or saliva sample for whole-genome sequencing and answer a series of online questionnaires. Participants may also consent to the study to access pre-existing clinical data. Participants will be added to the EAGER registry and data will be shared externally through established AIMS-2-TRIALS mechanisms. ETHICS AND DISSEMINATION: To date, EAGER has received full ethical approval for 11 out of the 13 sites in the UK (REC 23/SC/0022), Germany (S-375/2023), Portugal (CE-085/2023), Spain (HCB/2023/0038, PIC-164-22), Sweden (Dnr 2023-06737-01), Ireland (230907) and Italy (CET_62/2023, CEL-IRCCS OASI/24-01-2024/EM01, EM 2024-13/1032 EAGER). Findings will be disseminated via scientific publications and conferences but also beyond to participants and the wider community (eg, the AIMS-2-TRIALS website, stakeholder meetings, newsletters).

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 machine prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.

metaresearch head score (Codex)0.067
metaresearch head score (Gemma)0.070
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: none
GenreCandidate signal: Protocol · Consensus signal: Protocol
Teacher disagreement score0.113
Threshold uncertainty score0.378

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0670.070
Meta-epidemiology (narrow)0.0030.003
Meta-epidemiology (broad)0.0030.002
Bibliometrics0.0040.005
Science and technology studies0.0040.002
Scholarly communication0.0040.004
Open science0.0040.004
Research integrity0.0040.004
Insufficient payload (model declined to judge)0.1130.035

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 head0.127
GPT teacher head0.457
Teacher spread0.330 · 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 source (direct Gemma or distilled Codex), not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designObservational
Domainnot available
GenreProtocol

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

Citations4
Published2024
Admission routes1
Has abstractyes

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