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FVIII exposure, bleeding outcomes, and inhibitor development in 80 PUPs and MTPs with severe hemophilia A on emicizumab prophylaxis: real world data from the PedNet Registry.

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Inhibitors: A Need for Eradication?

Elena Santagostino1, Guy Young2, Carmen Escuriola Ettingshausen3

  • 1Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Angelo Bianchi Bonomi Hemophilia and Thrombosis Center, Milan, Italy, elena.santagostino@policlinico.mi.it.

Acta Haematologica
|February 21, 2019
PubMed
Summary

Immune tolerance induction (ITI) therapy successfully eradicates factor VIII (FVIII) inhibitors in most hemophilia patients. Early ITI is recommended for inhibitor patients, offering better treatment options and potentially lower costs.

Keywords:
Factor VIII concentrateHemophiliaImmune tolerance inductionInhibitorsVon Willebrand factor

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Area of Science:

  • Hematology
  • Immunology
  • Pharmacology

Background:

  • Factor VIII (FVIII) inhibitors are a major complication in hemophilia treatment.
  • Immune tolerance induction (ITI) therapy is effective in 60-80% of patients, restoring FVIII response.

Purpose of the Study:

  • To provide an overview of management strategies for hemophilia patients with inhibitors.
  • To discuss the efficacy and outcomes of ITI therapy.

Main Methods:

  • Review of presentations from the 6th International Coagulation Meeting (September 2017).
  • Tabulation of four pediatric and adult ITI therapy cases with successful outcomes.
  • Discussion of product switching (recombinant FVIII to plasma-derived FVIII/VWF) and prognostic indicators.

Main Results:

  • Successful outcomes were observed in ITI therapy cases.
  • Switching to plasma-derived FVIII/VWF concentrates may aid inhibitor eradication.
  • Early decline in inhibitor titer predicts ITI success, though biomarkers are needed.

Conclusions:

  • ITI therapy is a valuable treatment for hemophilia patients with inhibitors.
  • Prognostic biomarkers are required to better predict ITI success.
  • Emicizumab offers an alternative, but ITI benefits like wider product choice warrant its consideration.