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Record W4393111860 · doi:10.7554/elife.96085.1

ACK1 and BRK non-receptor tyrosine kinase deficiencies are associated with familial systemic lupus and involved in efferocytosis

2024· preprint· en· W4393111860 on OpenAlexaff
S. Guillet, Tomi Lazarov, Natasha Jordan, Bertrand Boisson, María Tello‐Lafoz, Barbara P. Craddock, Ting Zhou, Chihiro Nishi, Rohan Bareja, Hairu Yang, Frédéric Rieux‐Laucat, Rosa Fregel, Sabrina D. Dyall, David Isenberg, David D’Cruz, Nico Lachmann, Olivier Elemento, Agnès Viale, Nicholas D. Socci, Laurent Abel, Shigekazu Nagata, Morgan Huse, W. Todd Miller, Jean‐Laurent Casanova, Frédéric Geissmann

Bibliographic record

VenueeLife · 2024
Typepreprint
Languageen
FieldImmunology and Microbiology
TopicPhagocytosis and Immune Regulation
Canadian institutionsSickKids FoundationHospital for Sick Children
FundersNational Institute of Allergy and Infectious DiseasesNational Cancer InstituteJapan Science and Technology AgencyNational Institutes of HealthCore Research for Evolutional Science and TechnologyInstitut ServierMarie-Josée and Henry R. Kravis Center for Molecular OncologyMemorial Sloan-Kettering Cancer CenterLudwig Institute for Cancer ResearchServierUniversiteit LeidenLeids Universitair Medisch CentrumCycle for SurvivalCambridge University HospitalsImperial College LondonImperial College Healthcare NHS TrustJapan Society for the Promotion of ScienceUniversity of Cambridge
KeywordsEfferocytosisTyrosine kinaseMedicineReceptorBiologyInternal medicineGenetics

Abstract

fetched live from OpenAlex

Abstract Systemic Lupus Erythematosus (SLE) is an autoimmune disease, the pathophysiology and genetic basis of which are incompletely understood. Non-receptor tyrosine kinases (NRTKs) regulate activation, migration, and proliferation of immune cells. We report compound heterozygous deleterious variants in the kinase domains of the non-receptor tyrosine kinases (NRTK) TNK2/ACK1 in one multiplex family and PTK6/BRK in another. Experimental blockade of mouse ACK1 or BRK increases glomerular IgG deposits and circulating autoantibodies in an in vivo SLE model. In addition, we found that the patients’ ACK and BRK variants impair efferocytosis, the MERTK-mediated anti-inflammatory response to apoptotic cells, in human induced Pluripotent Stem Cells (hiPSC)-derived macrophages. Overall, our data suggest that ACK1 and BRK deficiencies are associated with human SLE and impair efferocytosis.

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)0.001
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 score0.005
Threshold uncertainty score0.015

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0010.001
Science and technology studies0.0000.001
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0010.001
Insufficient payload (model declined to judge)0.0050.001

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 head0.014
GPT teacher head0.224
Teacher spread0.210 · 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

Citations0
Published2024
Admission routes1
Has abstractyes

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