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Record W4405155079 · doi:10.1080/13816810.2024.2427879

Highly asymmetric early presentation of FEVR requiring enucleation

2024· article· en· W4405155079 on OpenAlex

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.
fundA Canadian funder is recorded on the work.

Bibliographic record

VenueOphthalmic Genetics · 2024
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicUbiquitin and proteasome pathways
Canadian institutionsUniversity of CalgaryHospital for Sick ChildrenUniversity of Toronto
FundersAssociation canadienne du médicament génériqueFoundation Fighting Blindness
KeywordsEnucleationRetinoblastomaMedicinePresentation (obstetrics)OphthalmologySurgeryBiologyGenetics

Abstract

fetched live from OpenAlex

Introduction Familial exudative vitreoretinopathy (FEVR) is a rare inherited disorder characterized by abnormal retinal vascular development. While it typically presents in childhood, distinguishing it from retinoblastoma in young infants can be challenging, especially in cases with asymmetric and advanced manifestations.Methods Case report.Results A 2-month-old female with microcephaly and intrauterine growth restriction (IUGR) presented with a left eye intraocular mass involving the entire globe and a flat anterior chamber. MRI showed no calcifications or contrast enhancement typical of retinoblastoma. Intravenous fluorescein angiography showed incomplete vascularization in the contralateral eye with compensatory neovascularization. The left eye was enucleated, and histology demonstrated a dysplastic retina with a retrolental membrane and abnormal vascular proliferations, confirming a diagnosis of FEVR. Genetic testing identified a novel pathogenic CTNNB1 p.Gly635* variant, inherited from the mother in whom it was present at 10–20% mosaicism.Discussion Variants in CTNNB1 cause of CTNNB1-neurodevelopmental disorder, characterized by microcephaly, IUGR, autism spectrum disorder, intellectual disability, and FEVR in 20–40% of cases. Affected children present at an early age and advanced stages of disease. This case highlights that FEVR can have a highly asymmetric and advanced presentation at an early age and must be distinguished from retinoblastoma in the differential diagnosis of leukocoria.

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)0.000
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
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 score0.020
Threshold uncertainty score0.472

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
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 head0.020
GPT teacher head0.278
Teacher spread0.258 · 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