Laying the foundations for gene therapy in Italy for patients with haemophilia A: A Delphi consensus study

Haemophilia. 2023 Mar;29(2):435-444. doi: 10.1111/hae.14709. Epub 2022 Dec 5.

Abstract

Introduction: Current treatment for haemophilia A involves factor VIII replacement or non-replacement (emicizumab) therapies, neither of which permanently normalise factor VIII levels. Gene therapy using adeno-associated viral (AAV) vectors is an emerging long-term treatment strategy for people with severe haemophilia A (PwSHA) that is likely to be available for clinical use in the near future.

Aim: This article proposes practical guidelines for the assessment, treatment, and follow-up of potential PwSHA candidates for AAV-based gene therapy.

Method: Using the Delphi method, a working group of Italian stakeholders with expertise in and knowledge of the care of adults with haemophilia A analysed literature for AAV-based gene therapy and drafted a list of statements that were circulated to a panel of Italian peers. During two rounds of voting, panel members voted on their agreement with each statement to reach a consensus.

Results: The Delphi process yielded 40 statements regarding haemophilia A gene therapy, across five topics: (1) organisational model; (2) multidisciplinary team; (3) patient engagement; (4) laboratory surveillance; and (5) patient follow-up and gene therapy outcomes. The consensus was reached for all 40 statements, with the second round of voting needed for five statements.

Conclusion: Use of the hub-and-spoke organisational model and multidisciplinary teams are expected to optimise patient selection for gene therapy, as well as the management of dosing and patient follow-up, patient engagement, laboratory surveillance, and patient expectations regarding outcomes. This approach should allow the benefits of AAV-based gene therapy for haemophilia A to be maximised.

Keywords: Delphi technique; Italy; consensus; genetic therapy; haemophilia A; patient care team; patient selection.

Publication types

  • Review

MeSH terms

  • Delphi Technique
  • Factor VIII
  • Genetic Therapy
  • Hemophilia A* / genetics
  • Hemophilia A* / therapy
  • Humans
  • Italy

Substances

  • Factor VIII

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