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Record W4408531462 · doi:10.1016/j.gimo.2025.102848

P004: Variant curation guideline specifications for ABCD1 from the ClinGen Peroxisomal Variant Curation Expert Panel*

2025· article· en· W4408531462 on OpenAlex
Shruthi Mohan, Irene DeBiase, Tatiana Yuzyuk, Alexa Dickson, Tiziano Pramparo, Stephanie Francis, Pepper St. Clair, R. Logan, Randy Cockerell, Raquel Fernandez, Ashley Ratzsch, Emily Kyle, Meredith Weaver, Sharon F. Suchy, Ann Moser, Rong Mao, Nancy Braverman

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.

Bibliographic record

VenueGenetics in Medicine Open · 2025
Typearticle
Languageen
FieldMedicine
TopicBiomedical Ethics and Regulation
Canadian institutionsMcGill University Health Centre
Fundersnot available
KeywordsData curationGuidelineComputer scienceComputational biologyBiologyData scienceMedicinePathology

Abstract

fetched live from OpenAlex

Peroxisomal disorders encompass a clinically and genetically heterogeneous group of disorders caused by impaired peroxisome biogenesis or function. Among them, ABCD1 deficiency aka Adrenoleukodystrophy (ALD) is the most common, caused by pathogenic variants in ABCD1 gene (OMIM 300371) on the X chromosome. ALD manifests as early-onset cerebral adrenoleukodystrophy, late-onset adrenomyeloneuropathy, or isolated adrenal insufficiency. Disease expression varies among affected individuals, and approximately half of the female individuals with ABCD1 deficiency manifest with a late onset myeloneuropathy.

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)0.002
metaresearch head score (Gemma)0.001
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Not applicable · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: none
Teacher disagreement score0.809
Threshold uncertainty score0.509

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0020.001
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 head0.216
GPT teacher head0.436
Teacher spread0.220 · 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