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Record W4408387603 · doi:10.1016/j.jcjo.2025.02.014

Longitudinal clinical characteristics of patients with neurofibromatosis type 1

2025· article· en· W4408387603 on OpenAlexvenueno aff
Lorenz Wassermann, Sandra Rezar‐Dreindl, Gregor S. Reiter, Thomas Neumayer, Ursula Schmidt‐Erfurth, Eva Stifter

Bibliographic record

VenueCanadian Journal of Ophthalmology · 2025
Typearticle
Languageen
FieldMedicine
TopicNeurofibromatosis and Schwannoma Cases
Canadian institutionsnot available
Fundersnot available
KeywordsMedicineNeurofibromatosisOptic nerveOphthalmologyVisual acuityRefractive errorRetrospective cohort studyMagnetic resonance imagingSurgeryRadiology

Abstract

fetched live from OpenAlex

PURPOSE: This study reports on longitudinal clinical characteristics of patients with neurofibromatosis type 1 (NF1) treated at the Department of Ophthalmology of the Medical University of Vienna. METHODS: This retrospective study included children with a genetically proven diagnosis of NF1. Clinical characteristics and outcomes, including best-corrected visual acuity (BCVA), refractive error, ocular motility, specific ophthalmological findings (e.g., Lisch nodules), and the presence of cutaneous café au lait stains were evaluated. Optical coherence tomography (OCT) and magnetic resonance imaging (MRI) were obtained to evaluate optic nerve abnormalities and early detection of optic glioma. RESULTS: One hundred and sixty-eight eyes of 85 children were included. The mean follow-up of the patients was 8 ± 3.7 years (range: 2-19 years) and mean age at baseline was 3.1 ± 2.6 years. Mean (±SD) BCVA was 0.19 (±0.20) logMAR at baseline. 16.5% of patients showed the presence of optic nerve abnormalities, 18.8% had therapy, 32.9% of the patients had cafe au lait stains, and 37.1% had Lisch nodules in the eye examination. Worse BCVA at baseline was associated with the presence of severe optic nerve abnormalities (mean 0.21 ± 0.14 logMAR; p < 0.01), compared to patients without optic nerve abnormalities (mean 0.14 ± 0.14 logMAR). Additionally, BCVA at baseline was dependent on the visual test (from mean 0.04 ± 0.19 logMAR to mean 0.3 ± 0.16 logMAR; p < 0.01). There was also a significant difference in BCVA change to follow-up in patients who received therapy compared to patients who did not (p = 0.033). The age showed no significant influence on the BCVA at baseline (p = 0.92). CONCLUSIONS: Optic nerve glioma poses a significant threat to vision in NF1 patients, necessitating biannual follow-up until age 6, including MRI, fundoscopy, and OCT imaging.

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)0.003
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.002
Threshold uncertainty score0.005

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0010.003
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0010.001
Science and technology studies0.0010.000
Scholarly communication0.0010.001
Open science0.0000.000
Research integrity0.0000.001
Insufficient payload (model declined to judge)0.0010.000

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 head0.034
GPT teacher head0.306
Teacher spread0.272 · 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 designObservational
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

Citations2
Published2025
Admission routes1
Has abstractyes

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Same venueCanadian Journal of OphthalmologySame topicNeurofibromatosis and Schwannoma CasesFrench-language works237,207