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Record W4414591535 · doi:10.1002/ana.78056

Primary Lateral Sclerosis Natural History Study: <scp>Primary Lateral Sclerosis Functional Rating Scale</scp> and Other Outcomes Assessment

2025· article· en· W4414591535 on OpenAlexaff
Ikjae Lee, Grace E. Jang, Ying Kuen Cheung, Alexander Sherman, Wendy Johnston, Frank Diaz, Joseph Americo Fernandes, Ali A. Habib, Nicholas J. Maragakis, Sabrina Paganoni, Katherine M. Burke, Björn Oskarsson, Jaimin Shah, Lorne Zinman, Mary Kay Floeter, Senda Ajroud‐Driss, Kelly Gwathmey, Terry Heiman‐Patterson, Omar Jawdat, Edward J. Kasarskis, Yaz Y. Kisanuki, James Wymer, Christina Fournier, Ghazala Hayat, Daragh Heitzman, Catherine Lomen‐Hoerth, Michael Pulley, Stephen N. Scelsa, David Walk, Stephen A. Goutman, Christen Shoesmith, Zachary Simmons, Eric J. Sorenson, Lauren Elman, Matthew Harms, Benjamin N. Hoover, RN ByoungSook Yun, Regina M. Santella, Hiroshi Mitsumoto

Bibliographic record

VenueAnnals of Neurology · 2025
Typearticle
Languageen
FieldMedicine
TopicAmyotrophic Lateral Sclerosis Research
Canadian institutionsHealth Sciences CentreWestern UniversitySunnybrook Health Science CentreUniversity of Alberta
Fundersnot available
KeywordsNatural historyMultiple sclerosisNatural history studyBiomarkerClinical neurology

Abstract

fetched live from OpenAlex

OBJECTIVE: The primary lateral sclerosis (PLS) consensus diagnostic criteria and functional rating scale (PLSFRS) were recently established to facilitate and optimize future PLS clinical trials. We examined the trajectory of the PLSFRS and other functional outcome measures and biomarkers in the PLS Natural History Study (PLS NHS) to understand their performance in this prospective cohort. METHODS: The PLS NHS is a prospective, longitudinal, multicenter study of people living with PLS in different diagnostic categories: early (disease duration <2 years); probable (2-4 years); and definite PLS (4-15 years). PLSFRS scores and other functional outcome measures were collected at baseline, 3-, 6-, 9-, and 12-month follow-up visits. Baseline characteristics were compared between the groups. The slopes of the PLSFRS and other functional outcome measures over 12 months were examined in the overall cohort and subgroups using linear mixed-effect models. The associations between baseline characteristics and the rate of PLSFRS decline were analyzed with linear regression models. RESULTS: A total of 76 participants were included: early (n = 6); probable (n = 26); and definite (n = 44) PLS. Baseline PLSFRS total scores were highest in the early PLS group, followed by the probable and definite PLS groups. In the overall cohort, the PLSFRS total score declined by 0.33 points/month (95% confidence interval [0.27-0.39], adjusted p < 0.05). The rate of decline was steepest in the early PLS group, followed by the probable and definite PLS groups. Baseline neurofilament light chain level was associated with the rate of PLSFRS decline over 1 year (p = 0.001). INTERPRETATION: In PLS, the rate of functional decline, as measured by the PLSFRS total score, is faster during the early phase of the disease. Neurofilament light might serve as a prognostic biomarker in PLS. ANN NEUROL 2026;99:418-428.

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)0.002
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: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.003
Threshold uncertainty score0.009

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0020.002
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0010.001
Science and technology studies0.0010.000
Scholarly communication0.0000.001
Open science0.0000.001
Research integrity0.0010.001
Insufficient payload (model declined to judge)0.0030.001

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 head0.091
GPT teacher head0.333
Teacher spread0.242 · 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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