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Record W4405445932 · doi:10.1136/bmjoo-2024-ukegg.10

#CC-Paper-01 Novel ocular imaging findings in Mucopolysaccharidosis II

2024· article· en· W4405445932 on OpenAlexaff
Haseeb Akhtar, Isabelle Chow, Moin Mohamed, Omar A. Mahroo

Bibliographic record

Venuenot available
Typearticle
Languageen
FieldMedicine
TopicLysosomal Storage Disorders Research
Canadian institutionsSt. Thomas Hospital
Fundersnot available
KeywordsMucopolysaccharidosisComputer scienceOphthalmologyMedicineInternal medicine

Abstract

fetched live from OpenAlex

Background Ocular involvement occurs in Mucopolysaccharidosis Type II (MPS II; Hunter syndrome). We report findings from imaging and electrophysiology, including novel imaging findings from optical coherence tomography (OCT).Methods A 22 year old male underwent detailed ocular phenotyping, including ultra-widefield pseudocolour and autofluorescence imaging, spectral domain OCT and electroretinogram (ERG) recording. He had been diagnosed with MPS II aged 5 and reported intermittent blurring of vision over the last 2 years.Results Fundal examination showed discrete pale lesions extending vertically, above and below the optic disc. Autofluorescence revealed concentric hyperautofluorescent rings. OCT showed macular oedema and paracentral outer retinal loss. OCT over the pale lesions showed these were inward scleral protrusions or thickening. ERGs showed generalised rod more than cone system dysfunction with a negative waveform to the dark-adapted bright flash.Conclusions Several findings are consistent with prior reports. Here, we were able to confirm, using OCT, that the pale lesions were scleral. The negative ERG waveform may reflect dark-adapted cone system responses.

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)0.002
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: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.007
Threshold uncertainty score0.023

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0010.002
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0020.001
Science and technology studies0.0000.000
Scholarly communication0.0010.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0070.001

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 head0.019
GPT teacher head0.309
Teacher spread0.290 · 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".

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Citations0
Published2024
Admission routes1
Has abstractyes

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