MétaCan
Menu
Back to cohort
Record W3185407023 · doi:10.1016/j.ajo.2021.07.013

Longitudinal Changes of Fixation Stability and Location Within 24 Months in Stargardt Disease: ProgStar Report No. 16

2021· article· en· W3185407023 on OpenAlexfundno aff
Etienne M. Schönbach, Rupert W. Strauß, Marco Cattaneo, Kaoru Fujinami, David G. Birch, Artur V. Cideciyan, Janet S. Sunness, Eberhart Zrenner, Srinivas R. Sadda, Hendrik P. N. Scholl

Bibliographic record

VenueAmerican Journal of Ophthalmology · 2021
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicRetinal Development and Disorders
Canadian institutionsnot available
FundersGenentechDeutsche Akademie der Naturforscher Leopoldina - Nationale Akademie der WissenschaftenHeidelberg EngineeringEberhard Karls Universität TübingenAstellas PharmaMinistry of Education, Culture, Sports, Science and TechnologyMinistry of Health, Labour and WelfareUniversitätsspital BaselUniversität BaselGreat Britain Sasakawa FoundationSanofi GenzymeF. Hoffmann-La RocheNovartisJapan Agency for Medical Research and DevelopmentAustrian Science FundAmgenFoundation Fighting BlindnessBedford Institute of OceanographyNightstaRxMerckU.S. Department of Defense
KeywordsStargardt diseaseFixation (population genetics)MedicineCohortProspective cohort studyOphthalmologySurgeryMacular degenerationInternal medicinePopulation

Abstract

fetched live from OpenAlex

Objective: Stargardt Disease type 1 (STGD1) is the most common macular dystrophy. The assessment of fixation describes an important dimension of visual function but there is limited data on its progression over time. We present longitudinal changes and investigate its usefulness for clinical trials.Design: International, multicenter, prospective cohort studyParticipants and Main Outcome Measures: A total of 239 individuals with genetically confirmed STGD1 (≥ 1 disease-causing ABCA4 variant). We determined the fixation stability (FS) using the bivariate contour ellipse area (1 SD-BCEA) and fixation location (FL) using the eccentricity of fixation from the fovea during 5 study visits every 6 months.Results: At baseline, 239 patients (105 males, 44 %) and 459 eyes with a median age of 32 years were included. The baseline mean log BCEA was 0.70 ± 1.41 log deg2Tanna P Strauss RW Fujinami K Michaelides M. Stargardt disease: clinical features, molecular genetics, animal models and therapeutic options.Br J Ophthalmol. 2017; 101: 25-30Crossref PubMed Scopus (138) Google Scholar and the mean FL was 6.25 ± 4.40 deg. Although the mean log BCEA did not monotonically increase from visit to visit, the overall yearly increase in log BCEA was 0.124 log deg2Tanna P Strauss RW Fujinami K Michaelides M. Stargardt disease: clinical features, molecular genetics, animal models and therapeutic options.Br J Ophthalmol. 2017; 101: 25-30Crossref PubMed Scopus (138) Google Scholar (95% CI, 0.063-0.185). The rate of change was not different between the two years but increased faster in eyes without flecks outside of the vascular arcades and depended on baseline log BCEA. FL did not change statistically significantly over time.Conclusion and relevance: Fixation parameters are unlikely to be sensitive outcome measures for clinical trials in STGD1 but they may provide useful ancillary information in selected cases to longitudinally describe and understand an eye's visual function.

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)0.001
Version: codex-gemma-dda1882f352aValidation 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 score0.010
Threshold uncertainty score0.342

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.001
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 head0.016
GPT teacher head0.273
Teacher spread0.257 · 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 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

Citations12
Published2021
Admission routes1
Has abstractyes

Explore more

Same venueAmerican Journal of OphthalmologySame topicRetinal Development and DisordersFrench-language works237,207