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Record W4286592276 · doi:10.1097/ico.0000000000003078

Keratopathy in Noonan Syndrome

2022· article· en· W4286592276 on OpenAlexaff
Yelin Yang, Jyh Haur Woo, Asim Ali

Bibliographic record

VenueCornea · 2022
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicProtein Tyrosine Phosphatases
Canadian institutionsHospital for Sick ChildrenUniversity of Toronto
Fundersnot available
KeywordsMedicineNoonan syndromePtosisCorneaOphthalmologySurgeryInternal medicine

Abstract

fetched live from OpenAlex

PURPOSE: Anterior segment abnormalities associated with Noonan syndrome are rare. We report our experience with 2 patients who developed keratopathy with significant visual sequelae. METHODS: case series. RESULTS: The first patient is a 9-year-old boy with genetically confirmed Noonan syndrome. At presentation, he was noted to have diffuse inferior epitheliopathy with vascularization and bilateral mild ptosis. Over 1 year, he developed focal areas of scarring with deterioration of vision and underwent superficial keratectomy in the left eye. However, over the following 2 years, he experienced recurrent corneal scarring and vascularization. The second patient is a 7-year-old boy with phenotypic Noonan syndrome. At presentation, he had an anterior subepithelial corneal scar inferiorly with epithelial defects in both eyes. He also had bilateral moderate ptosis and lagophthalmos. Despite lubrication, he developed recurrent bilateral corneal erosions with focal areas of scarring associated with vascularization and underwent superficial keratectomy for both eyes. Despite this, there was worsening corneal scarring and vascularization over time, eventually requiring deep anterior lamellar keratoplasty in the left eye. The host cornea showed a disturbed Bowman layer and an acellular mass of fibrous collagenous tissue between epithelium and stroma. CONCLUSIONS: Noonan syndrome may be associated with visually significant keratopathy, manifesting as focal corneal scarring with vascularization. These changes may due to an excessive fibrotic response in Noonan syndrome. Early recognition and treatment can help to delay the progression of keratopathy and need for surgical intervention.

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 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 score0.290
Threshold uncertainty score0.329

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 head0.006
GPT teacher head0.207
Teacher spread0.201 · 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.

The models applied no category: nothing in the taxonomy fit this work.
Study designBench or experimental
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
Published2022
Admission routes1
Has abstractyes

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