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Record W4313214131 · doi:10.1101/2022.12.19.521070

LRRK2 Kinase Activity Regulates Parkinson’s Disease-Relevant Lipids at the Lysosome

2022· preprint· en· W4313214131 on OpenAlexaff
Michael T. Maloney, Xiang Wang, Rajarshi Ghosh, Shan V. Andrews, Romeo Maciuca, Shababa T. Masoud, Richard M. Caprioli, John Chen, Chi‐Lu Chiu, Sonnet S. Davis, Audrey Cheuk-Nga Ho, Hoang N. Nguyen, Nicholas E. Propson, Michelle L. Reyzer, Oliver B. Davis, Matthew C. Deen, Sha Zhu, Gilbert Di Paolo, David J. Vocadlo, Anthony A. Estrada, Javier De Vicente, Joseph W. Lewcock, Annie Arguello, Jung H. Suh, Sarah Huntwork‐Rodriguez, Anastasia G. Henry

Bibliographic record

VenuebioRxiv (Cold Spring Harbor Laboratory) · 2022
Typepreprint
Languageen
FieldMedicine
TopicLysosomal Storage Disorders Research
Canadian institutionsSimon Fraser University
FundersCelgeneNational Institute of Neurological Disorders and StrokeSanofiGlaxoSmithKlinePfizerVerily Life SciencesBristol-Myers SquibbNational Institutes of HealthU.S. Department of Health and Human ServicesMichael J. Fox Foundation for Parkinson's ResearchFoundation for the National Institutes of Health
KeywordsLRRK2GlucocerebrosidaseLysosomeParkinson's diseaseCatabolismKinaseCell biologySecretionBiologyDiseaseBiochemistryChemistryInternal medicineMedicineEnzyme

Abstract

fetched live from OpenAlex

Abstract Lysosomal dysfunction is a hallmark of Parkinson’s disease (PD), and variants in several PD-associated genes, including LRRK2 , perturb lysosomal homeostasis. Based on this, LRRK2 kinase inhibition is being explored as a therapeutic approach for the treatment of PD. LRRK2 inhibitors reduce levels of BMP, an endolysosomal lipid involved in glycosphingolipid (GSL) catabolism, in urine from preclinical models and clinical subjects, however, the mechanisms by which LRRK2 regulates BMP and the functional significance of this change to disease are undefined. We establish that LRRK2 regulates secretion of BMP- and GSL-containing vesicles from kidney into urine and modulates BMP and GSL levels in the brain. BMP accumulates within lysosomes as a secondary response to LRRK2’s effects on the activity of glucocerebrosidase (GCase), a PD-linked enzyme involved in GSL catabolism. Alterations in BMP and GCase substrate turnover are observed in CSF from LRRK2-PD patients, highlighting the relevance of LRRK2-dependent lysosomal dysfunction in disease.

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)0.000
metaresearch head score (Gemma)0.000
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Bench or experimental · Consensus signal: Bench or experimental
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.003
Threshold uncertainty score0.010

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0010.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0030.002

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 head0.020
GPT teacher head0.257
Teacher spread0.237 · 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 designBench or experimental
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

Citations11
Published2022
Admission routes1
Has abstractyes

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Same venuebioRxiv (Cold Spring Harbor Laboratory)Same topicLysosomal Storage Disorders ResearchFrench-language works237,207