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Record W2997770921 · doi:10.1101/19010991

<i>GBA</i> variants in REM sleep behavior disorder: a multicenter study

2019· preprint· en· W2997770921 on OpenAlexafffund
Lynne Krohn, Jennifer A. Ruskey, Uladzislau Rudakou, Etienne Léveillé, Farnaz Asayesh, Isabelle Arnulf, Yves Dauvilliers, Birgit Högl, Ambra Stefani, Christelle Monaca, Beatriz Abril, Giuseppe Plazzi, Elena Antelmi, Luigi Ferini‐Strambi, Anna Heidbreder, Bradley F. Boeve, Alberto J. Espay, Valérie Cochen De Cock, Brit Mollenhauer, Friederike Sixel‐Döring, Claudia Trenkwalder, Karel Šonka, David Kemlink, Michela Figorilli, Monica Puligheddu, Femke Dijkstra, Mineke Viaene, Wolfgang H. Oertel, Marco Toffoli, Gian Luigi Gigli, Mariarosaria Valente, Jean‐François Gagnon, Alex Désautels, Jacques Montplaisir, Ronald B. Postuma, Guy A. Rouleau, Ziv Gan‐Or

Bibliographic record

VenuemedRxiv · 2019
Typepreprint
Languageen
FieldMedicine
TopicLysosomal Storage Disorders Research
Canadian institutionsUniversité de MontréalHôpital du Sacré-Cœur de MontréalUniversité du Québec à MontréalMcGill UniversityCanadian Sleep & Circadian NetworkMontreal Neurological Institute and Hospital
FundersCanadian Glycomics NetworkAcorda TherapeuticsFonds de Recherche du Québec - SantéBritannia PharmaceuticalsDeutsche Parkinson VereinigungSun PharmaServierNational Institutes of HealthTau ConsortiumConsortium canadien en neurodégénérescence associée au vieillissementCanada First Research Excellence FundCanadian Institutes of Health ResearchParkinson CanadaMcGill UniversityDeutsche ForschungsgemeinschaftUCB PharmaBoston Scientific CorporationMichael J. Fox Foundation for Parkinson's ResearchLittle Family FoundationMayo ClinicBiogenNational Institute for Health and Care ResearchTeva Pharmaceutical IndustriesUniversity of OxfordParkinson's UK
KeywordsNeurodegenerationREM sleep behavior disorderOdds ratioInternal medicineMedicinePsychologyGeneticsOncologyBiologyPolysomnographyDisease

Abstract

fetched live from OpenAlex

Abstract Objective To study the role of GBA variants in the risk for isolated rapid-eye-movement (REM)-sleep behavior disorder (iRBD) and conversion to overt neurodegeneration. Methods A total of 4,147 individuals were included: 1,061 iRBD patients and 3,086 controls. GBA was fully sequenced using molecular inversion probes and Sanger sequencing. We analyzed the effects of GBA variants on the risk for iRBD, age at onset (AAO) and conversion rates. Results GBA variants were found in 9.5% of iRBD patients compared to 4.1% in controls (odds ratio [OR]=2.45, 95% CI=1.87–3.22, p =1×10 −10 ). The estimated OR for mild p.N370S variant carriers was 3.69, 95% CI=1.90–7.14, p =3.5×10 −5 , while for severe variant carriers it was 17.55, 95% CI=2.11–145.9, p =0.0015. Carriers of severe GBA variants had an average AAO of 52.8 years, 7-8 years earlier than those with mild variants or non-carriers ( p =0.029). Of the GBA variant carriers with available data, 52.5% had converted, compared to 35.6% in non-carriers ( p =0.011), with a trend for faster conversion among severe GBA variant carriers. However, the results on AAO and conversion were based on small numbers and should be taken with caution. Conclusions GBA variants robustly and differentially increase the risk of iRBD. The rate of conversion to neurodegeneration is also increased and may be faster among severe GBA variant carriers, although confirmation will be required in larger samples. Screening for RBD in healthy carriers of GBA variants should be studied as a potential way to identify GBA variant carriers who will develop a synucleinopathy in the future.

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)0.001
metaresearch head score (Gemma)0.001
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesMeta-epidemiology (narrow)
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.008
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.001
Meta-epidemiology (narrow)0.0010.001
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0010.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0010.002
Research integrity0.0000.002
Insufficient payload (model declined to judge)0.0010.001

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 head0.031
GPT teacher head0.331
Teacher spread0.300 · 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.

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

Citations5
Published2019
Admission routes2
Has abstractyes

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