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Record W4404587345 · doi:10.7554/elife.96085.3

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

2024· article· en· W4404587345 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
Typearticle
Languageen
FieldImmunology and Microbiology
TopicPhagocytosis and Immune Regulation
Canadian institutionsSickKids FoundationHospital for Sick Children
FundersCore Research for Evolutional Science and TechnologyNational Institute of Allergy and Infectious DiseasesJapan Society for the Promotion of ScienceFondation pour la Recherche MédicaleAssistance Publique - Hôpitaux de ParisArthritis Research UK
KeywordsEfferocytosisMERTKImmunologyTyrosine kinaseSystemic lupus erythematosusAutoantibodyAutoimmunityMedicineBiologyImmune systemCancer researchReceptor tyrosine kinaseMacrophageKinaseCell biologySignal transductionDiseaseAntibodyInternal medicineGeneticsIn vitro

Abstract

fetched live from OpenAlex

Systemic lupus erythematosus (SLE) is an autoimmune disease, the pathophysiology and genetic basis of which are incompletely understood. Using a forward genetic screen in multiplex families with SLE, we identified an association between SLE and compound heterozygous deleterious variants in the non-receptor tyrosine kinases (NRTKs) ACK1 and BRK . Experimental blockade of ACK1 or BRK increased circulating autoantibodies in vivo in mice and exacerbated glomerular IgG deposits in an SLE mouse model. Mechanistically, NRTKs regulate activation, migration, and proliferation of immune cells. We found that the patients’ ACK1 and BRK variants impair efferocytosis, the MERTK-mediated anti-inflammatory response to apoptotic cells, in human induced pluripotent stem cell (hiPSC)-derived macrophages, which may contribute to SLE pathogenesis. Overall, our data suggest that ACK1 and BRK deficiencies are associated with human SLE and impair efferocytosis in macrophages.

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)0.000
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Bench or experimental · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.616
Threshold uncertainty score0.551

Codex and Gemma teacher scores by category

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 communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0000.000

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 head0.011
GPT teacher head0.216
Teacher spread0.205 · 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.

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

Citations6
Published2024
Admission routes1
Has abstractyes

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