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Record W4366525787 · doi:10.1136/gutjnl-2023-329689

Genetic coding variant in complement factor B (CFB) is associated with increased risk for perianal Crohn’s disease and leads to impaired CFB cleavage and phagocytosis

2023· review· en· W4366525787 on OpenAlexaff
Marzieh Akhlaghpour, Talin Haritunians, Shyam K. More, Lisa Thomas, Dalton T. Stamps, Shishir Dube, Dalin Li, Shaohong Yang, Carol J. Landers, Emebet Mengesha, Hussein Hamade, Ramachandran Murali, Alka A. Potdar, Andrea J. Wolf, Gregory J. Botwin, Michelle Khrom, Ashwin N. Ananthakrishnan, William A. Faubion, Bana Jabrì, Sérgio A. Lira, Rodney D. Newberry, Robert S. Sandler, R. Balfour Sartor, Ramnik J. Xavier, Steven R. Brant, Judy H. Cho, Richard H. Duerr, Mark Lazarev, John D. Rioux, L. Philip Schumm, Mark S. Silverberg, Karen Zaghiyan, Phillip Fleshner, Gil Melmed, Eric A. Vasiliauskas, Christina Ha, Shervin Rabizadeh, Gaurav Syal, Nirupama Bonthala, David Ziring, Stephan R. Targan, Millie D. Long, Dermot McGovern, Kathrin S. Michelsen

Bibliographic record

VenueGut · 2023
Typereview
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicInflammatory Bowel Disease
Canadian institutionsUniversity of TorontoMount Sinai HospitalUniversité de Montréal
FundersNational Institute of Diabetes and Digestive and Kidney DiseasesInflammatory Bowel and Immunobiology Research InstituteCedars-Sinai Medical CenterFrancis Crick InstituteLeona M. and Harry B. Helmsley Charitable Trust
KeywordsCrohn's diseaseComplement factor BComplement systemImmunologyFactor HCoding (social sciences)MedicineBiologyDiseaseVirologyInternal medicineAntibodyMathematicsStatistics

Abstract

fetched live from OpenAlex

Objective Perianal Crohn’s disease (pCD) occurs in up to 40% of patients with CD and is associated with poor quality of life, limited treatment responses and poorly understood aetiology. We performed a genetic association study comparing CD subjects with and without perianal disease and subsequently performed functional follow-up studies for a pCD associated SNP in Complement Factor B ( CFB ). Design Immunochip-based meta-analysis on 4056 pCD and 11 088 patients with CD from three independent cohorts was performed. Serological and clinical variables were analysed by regression analyses. Risk allele of rs4151651 was introduced into human CFB plasmid by site-directed mutagenesis. Binding of recombinant G252 or S252 CFB to C3b and its cleavage was determined in cell-free assays. Macrophage phagocytosis in presence of recombinant CFB or serum from CFB risk, or protective CD or healthy subjects was assessed by flow cytometry. Results Perianal complications were associated with colonic involvement, OmpC and ASCA serology, and serology quartile sum score. We identified a genetic association for pCD (rs4151651), a non-synonymous SNP (G252S) in CFB , in all three cohorts. Recombinant S252 CFB had reduced binding to C3b, its cleavage was impaired, and complement-driven phagocytosis and cytokine secretion were reduced compared with G252 CFB. Serine 252 generates a de novo glycosylation site in CFB. Serum from homozygous risk patients displayed significantly decreased macrophage phagocytosis compared with non-risk serum. Conclusion pCD-associated rs4151651 in CFB is a loss-of-function mutation that impairs its cleavage, activation of alternative complement pathway, and pathogen phagocytosis thus implicating the alternative complement pathway and CFB in pCD aetiology.

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 categoriesMeta-epidemiology (narrow)
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: none
GenreCandidate signal: Review · Consensus signal: none
Teacher disagreement score0.653
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.000
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 head0.037
GPT teacher head0.303
Teacher spread0.266 · 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
GenreReview

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

Citations22
Published2023
Admission routes1
Has abstractyes

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