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Comprehensive analysis of complement activation in a hydroxyurea-treated patient cohort with sickle cell disease

2025· article· en· W4416378440 on OpenAlexafffund
Daniel Diatlov, Arlette Bohorquez, Melanie Kirby‐Allen, Christoph Licht

Bibliographic record

VenueBlood Advances · 2025
Typearticle
Languageen
FieldMedicine
TopicHemoglobinopathies and Related Disorders
Canadian institutionsHospital for Sick ChildrenUniversity of Toronto
FundersCanadian Institutes of Health ResearchHospital for Sick ChildrenAcademy of FinlandPfizer
KeywordsComplement systemHemeComplement (music)HemoglobinDiseaseHemoglobinopathyCellSickle cell anemia

Abstract

fetched live from OpenAlex

ABSTRACT: Sickle cell disease (SCD) is an understudied, life-threatening genetic disorder affecting ∼300 000 infants yearly with limited treatment options. The complement system, a critical part of innate immunity, has emerged as a contributor to SCD pathophysiology, thus presenting a potential new treatment target. Our aim was to assess complement activity in children with SCD receiving hydroxyurea (HU) therapy during vaso-occlusive crisis (VOC) and steady state. Blood samples were collected from 46 pediatric patients with SCD during VOC (early and late) and steady state, with control samples from healthy volunteers. Clinical data were obtained from patient records, and patient heme levels were measured using colorimetric assay. Complement deposition on endothelial cells (ECs) was quantified using high-throughput automated immunofluorescence imaging. Complement protein concentration was measured using enzyme-linked immunosorbent assay and multiplex assays. We found that, in vitro, heme drove C3b and C5b-9 deposition on ECs. Patients had increased heme levels during both VOC and steady state compared with healthy controls. However, complement activation correlated with total hemoglobin (Hb) concentration in patients during steady state, but not heme levels. C5b-9 deposition on ECs was significantly higher in patients during early crisis compared with late crisis, suggesting heightened complement activity early in VOC, with C5b-9 deposition also strongly correlating with circulating soluble C5b-9 levels. A significant increase in the C3b/C3 ratio further indicated early complement activation during VOC. In conclusion, complement activity is likely highest in early VOC in patients with SCD, and presents a critical potential treatment target, but is overall attenuated by HU therapy despite elevated heme or Hb levels.

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: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.027
Threshold uncertainty score0.351

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.001
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 head0.005
GPT teacher head0.242
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 teacher head, not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
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

Citations0
Published2025
Admission routes2
Has abstractyes

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