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Record W4409101131 · doi:10.1016/j.jaapos.2025.104183

Optic atrophy in Lamb-Shaffer syndrome: two case presentations with ophthalmic imaging studies

2025· article· en· W4409101131 on OpenAlexafffund
Moath O Baeshen, Stuti M. Tanya, Daniela Toffoli

Bibliographic record

VenueJournal of American Association for Pediatric Ophthalmology and Strabismus · 2025
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicOcular Disorders and Treatments
Canadian institutionsMcGill University
FundersMcGill University Health Centre
KeywordsAtrophyMedicineGeographic atrophyOphthalmologyPathologyMacular degeneration

Abstract

fetched live from OpenAlex

Lamb-Shaffer syndrome is a rare neuro-developmental disorder associated with haploinsufficiency of the SOX5 gene. Clinical manifestations include speech delay, behavioral challenges, intellectual disability, and dysmorphic features. Up to 57% of patients have ophthalmic findings, including strabismus, refractive error, and optic nerve abnormalities. We present 2 siblings who are the first documented cases of Lamb-Shaffer syndrome with the p.R571W: c.1711 C>T SOX5 mutation to demonstrate optic nerve atrophy.

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)0.001
metaresearch head score (Gemma)0.005
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Case report · Consensus signal: Case report
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.005
Threshold uncertainty score0.015

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0010.005
Meta-epidemiology (narrow)0.0030.002
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0030.001
Science and technology studies0.0020.003
Scholarly communication0.0020.002
Open science0.0020.003
Research integrity0.0050.003
Insufficient payload (model declined to judge)0.0030.001

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 head0.008
GPT teacher head0.319
Teacher spread0.311 · 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 designCase report
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

Citations0
Published2025
Admission routes2
Has abstractyes

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