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Record W4406113180 · doi:10.1111/ene.70035

Substantia nigra degeneration in spinocerebellar ataxia 2 and 7 using neuromelanin‐sensitive imaging

2025· article· en· W4406113180 on OpenAlexaff
Lydia Chougar, Giulia Coarelli, François‐Xavier Lejeune, Pia Ziegner, Rahul Gaurav, Emma Biondetti, Sabrina Sayah, Rania Hilab, Alain Dagher, Alexandra Dürr, Stéphane Lehéricy

Bibliographic record

VenueEuropean Journal of Neurology · 2025
Typearticle
Languageen
FieldNeuroscience
TopicGenetic Neurodegenerative Diseases
Canadian institutionsMcGill UniversityMontreal Neurological Institute and Hospital
FundersInstitut National de la Santé et de la Recherche Médicale
KeywordsSpinocerebellar ataxiaPars compactaSubstantia nigraNeuromelaninMedicinePathologyInternal medicineAtaxiaNeurosciencePsychologyDopaminergicDopamine

Abstract

fetched live from OpenAlex

OBJECTIVE: Spinocerebellar ataxias (SCA) are neurodegenerative diseases with widespread lesions across the central nervous system. Ataxia and spasticity are usually predominant, but patients may also present with parkinsonism. We aimed to characterize substantia nigra pars compacta (SNc) degeneration in SCA2 and 7 using neuromelanin-sensitive imaging. METHODS: Ataxic and preataxic expansion carriers with SCA2 (n=15) and SCA7 (n=15) and healthy controls (n=10) were prospectively recruited. Volume and signal-to-noise ratio (SNR) values of the SNc were extracted from neuromelanin-sensitive images. ROC curves were used to determine the metrics that best differentiated SCA participants. Correlations between imaging measurements, clinical variables, and plasma neurofilaments light chain (NfL) levels were investigated. RESULTS: SCA2 participants had lower SNR values in the SNc than controls (110.2 ± 1.3 versus 113.2 ± 1.4; p < 0.001) and those with SCA7 (112.5 ± 2.1; p < 0.01). SNR in SCA7 participants and controls did not differ. In ataxic patients, SNc volumes were lower in SCA2 (0.13 ± 0.04; p = 0.06) and SCA7 (0.10 ± 0.03, p = 0.02) patients compared to controls (0.17 ± 0.04). Signal decrease was detected at the preataxic stage in SCA2, but not in SCA7. SCA2 participants showed prominent involvement of the associative and limbic nigral territories. SNR discriminated ataxic and preataxic SCA2 participants from controls (AUC ≥0.94). SNc volume differentiated ataxic SCA7 participants from controls (AUC = 1), but not preataxic ones. In SCA7, correlations were observed between SNc volume and time to onset, CAG repeats, clinical severity scores, and NfL. CONCLUSIONS: Neuromelanin-sensitive imaging provides biomarkers of nigral degeneration in SCAs, detectable from the preataxic stage in SCA2, which could potentially serve as outcome measures in clinical trials.

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)0.001
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 score0.006

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0010.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0020.000
Science and technology studies0.0000.000
Scholarly communication0.0010.000
Open science0.0000.000
Research integrity0.0010.000
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 head0.023
GPT teacher head0.263
Teacher spread0.240 · 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".

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Citations2
Published2025
Admission routes1
Has abstractyes

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