Abstract Number: PB0618
Meeting: ISTH 2021 Congress
Background: Immune tolerance induction (ITI) is the only therapy to eradicate anti-factor VIII antibodies (inhibitors) in patients with hemophilia A. However, this burdensome and costly treatment fails in 10-40%. In order to determine whether to attempt ITI, it is important to identify determinants for ITI success.
Aims: This systematic review aims to assess:
(i) the genetic determinants for ITI success and
(ii) the non-genetic determinants for ITI success in patients with hemophilia A.
Methods: A comprehensive literature search was conducted on February 17 2021, in the MEDLINE and Embase databases using the following search terms; hemophilia and immune tolerance. Cross-sectional, longitudinal and cohort studies and registries reporting on determinants for ITI outcome in patients with congenital hemophilia A who underwent ITI therapy were eligible for inclusion. Data were extracted on study, patient and treatment characteristics, ITI outcome definitions and genetic and non-genetic determinants associated with ITI success or failure. Two independent reviewers performed the study selection, data extraction and methodological quality assignment using an adapted checklist of the Joanna Briggs Institute for cross-sectional and cohort studies. Studies were classified as high-quality when ≥5/9 criteria were met. Incidence of ITI success were reported for each study.
In case of limited heterogeneity, pooled effect estimates of individual determinants will be assessed.
Results: The literature search yielded 1021 unique papers. After title and abstract screening, 54 papers were selected. Full-text screening is currently ongoing. Final results of this systematic review are expected in July.
Conclusions: This systematic review will summarize the evidence on genetic and non-genetic determinants for ITI outcome in patients with congenital hemophilia A. We expect that these results will provide more insight into factors influencing the outcome of ITI.
To cite this abstract in AMA style:Oomen I, Mesquita Camelo R, Meireles Rezende S, Voorberg J, Mancuso ME, Oldenburg J, Carcao M, Matino D, Lillicrap D, Fischer K, Fijnvandraat K, Gouw SC, International GO-ITI study group . Genetic and Non-genetic Determinants for the Outcome of Immune Tolerance Induction in Patients with Hemophilia A and Inhibitors – A Systematic Review [abstract]. Res Pract Thromb Haemost. 2021; 5 (Suppl 2). https://abstracts.isth.org/abstract/genetic-and-non-genetic-determinants-for-the-outcome-of-immune-tolerance-induction-in-patients-with-hemophilia-a-and-inhibitors-a-systematic-review/. Accessed February 28, 2024.
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