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Record W4408415307 · doi:10.3324/haematol.2024.287057

Regulation of immune responses to therapeutic factor VIII by transplacental delivery of Fc-fused immunodominant factor VIII domains or peptides

2025· article· en· W4408415307 on OpenAlexaff
Alejandra Reyes‐Ruiz, Sandrine Delignat, Aurélien Azam, Victoria Daventure, Leslie Dourthe, Angélina Mimoun, Geneviève McCluskey, Maria T. Georgescu, David Lillicrap, Jordan D. Dimitrov, Sébastien Lacroix‐Desmazes

Bibliographic record

VenueHaematologica · 2025
Typearticle
Languageen
FieldMedicine
TopicHemophilia Treatment and Research
Canadian institutionsQueen's University
FundersAgence Nationale de la Recherche
KeywordsTransplacentalImmune toleranceImmune systemOffspringImmunologyAntibodyVon Willebrand factorFetusPlacentaBiologyPregnancyPlateletGenetics

Abstract

fetched live from OpenAlex

Patients with severe hemophilia A (HA) often develop undesired immune responses to therapeutic factor VIII (FVIII) that hamper replacement therapy with FVIII-derived products. The transplacental delivery of two Fc-fused FVIII domains in pregnant HA mice was shown to induce partial FVIII-specific immune tolerance in the offspring. Here, we evaluated whether the transplacental delivery of Fc-fused FVIII (rFVIIIFc) induces complete immune tolerance towards FVIII. rFVIIIFc injected to pregnant HA mice was poorly transferred to the fetal circulation and failed to confer tolerance to exogenous FVIII in the offspring. The poor transplacental delivery of rFVIIIFc was associated with the large size of the molecule and with the presence of positive patches at the surface of FVIII. It was, however, independent from the capacity of rFVIIIFc to bind Fcg receptors or von Willebrand factor in the maternal circulation. Conversely, the transplacental delivery of Fc-fused A2 and C2 immunodominant domains of FVIII, as well as of Fc-fused molecules containing seven different immunodominant FVIII-derived peptides decreased the levels of anti-FVIII antibodies following FVIII replacement therapy in the offspring. Our study paves the way towards the development of engineered Fc-fused molecules able to efficiently cross the placenta and confer potent and long-lasting immune tolerance to protein therapeutics.

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: Bench or experimental
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.280
Threshold uncertainty score0.755

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)0.0010.000

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 head0.038
GPT teacher head0.328
Teacher spread0.290 · 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

Citations1
Published2025
Admission routes1
Has abstractyes

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