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Record W4414225018 · doi:10.1016/j.ajhg.2025.08.017

RetiGene, a comprehensive gene atlas for inherited retinal diseases

2025· review· en· W4414225018 on OpenAlexafffund
Carlo Rivolta, Elifnaz Çelik, Dhryata Kamdar, Francesca Cancellieri, Karolina Kamińska, Mukhtar Ullah, Pilar Barberán-Martínez, Manon Bouckaert, Marta Cortón, Emma Delanote, Lidia Fernández‐Caballero, Gema García‐García, Lara K. Holtes, Marianthi Karali, Irma López, Virginie G. Peter, Nina Schneider, Lieselot Vincke, Carmen Ayuso, Sandro Banfi, Béatrice Bocquet, Frauke Coppieters, Frans P.M. Cremers, Chris F. Inglehearn, Takeshi Iwata, Vasiliki Kalatzis, Robert K. Koenekoop, José M. Millán, Dror Sharon, Carmel Toomes, Mathieu Quinodoz

Bibliographic record

VenueThe American Journal of Human Genetics · 2025
Typereview
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicRetinal Development and Disorders
Canadian institutionsMcGill Genome CentreMcGill University Health Centre
FundersNational Eye InstituteEuropean Regional Development FundInstituto de Salud Carlos IIIHORIZON EUROPE Framework ProgrammeFondation de l'Hôpital de Montréal pour enfantsCentro de Investigación Biomédica en Red de CáncerGeneralitat ValencianaFonds de Recherche du Québec - SantéFoundation Fighting BlindnessVlaamse regeringCentro de Investigación Biomédica en Red en Bioingeniería, Biomateriales y NanomedicinaLeids Universitair Medisch CentrumCentro de Investigación Biomédica en Red Enfermedades CardiovascularesUniversiteit GentCanadian Institutes of Health ResearchFonds Wetenschappelijk OnderzoekFederación Española de Enfermedades RarasNational Institutes of HealthSchweizerischer Nationalfonds zur Förderung der Wissenschaftlichen ForschungBijzonder Onderzoeksfonds UGentFight for Sight UKEuropean CommissionFondazione TelethonRetina UKMinisterio de Sanidad, Consumo y Bienestar SocialNational Science FoundationChildren's Hospital Foundation
KeywordsGeneMendelian inheritanceOMIM : Online Mendelian Inheritance in ManCandidate geneGene mappingFunction (biology)Genetic dataGenetic testingInheritance (genetic algorithm)

Abstract

fetched live from OpenAlex

Inherited retinal diseases (IRDs) are rare disorders, typically presenting as Mendelian traits, that result in stationary or progressive visual impairment. They are characterized by extensive genetic heterogeneity, possibly the highest among all human genetic diseases, as well as diverse inheritance patterns. Despite advances in gene discovery, limited understanding of gene function and challenges in accurately interpreting variants continue to hinder both molecular diagnosis and genetic research in IRDs. One key problem is the absence of a comprehensive and widely accepted catalog of disease-associated genes, which would ensure consistent genetic testing and reliable molecular diagnoses. With the rapid pace of IRD gene discovery, gene catalogs require frequent validation and updates to remain clinically and scientifically useful. To address these gaps, we developed RetiGene, an expert-curated gene atlas that integrates variant data, bulk and single-cell RNA sequencing, and functional annotations. Through the integration of diverse data sources, RetiGene supports candidate gene prioritization, functional studies, and therapeutic development in IRDs.

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: Not applicable · Consensus signal: none
GenreCandidate signal: Review · Consensus signal: Review
Teacher disagreement score0.966
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0010.000
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 head0.027
GPT teacher head0.340
Teacher spread0.313 · 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 designNot applicable
Domainnot available
GenreReview

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

Citations25
Published2025
Admission routes2
Has abstractyes

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