Inhibitors and immune tolerance induction in hemophilia A, a balancing act

Sarah Schep

doi:10.33540/868

Inhibitors and immune tolerance induction in hemophilia A, a balancing act

Sarah Schep

Research output: Thesis › Doctoral thesis 1 (Research UU / Graduation UU)

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Abstract

Objective of this thesis
Two main questions have intrigued researchers and physicians in the field of hemophilia for years:How do inhibitors develop and which immunological mechanisms contribute to the eradication of inhibitors during ITI?
These are also the two main questions discussed in this thesis. It includes a combination of translational and clinical research, which was aimed to provide an in-depth evaluation of the mechanisms of inhibitor formation and eradication in hemophilia A patients. Elucidation of these mechanisms could contribute to better preventive measures against allo-immunization to FVIII and optimization of current ITI protocols, for example by identifying new pivotal targets for
tolerance reinstatement.
Increasing knowledge regarding both failure as well as (re)induction of immune tolerance is not only beneficial in hemophilia A, but could be valuable in other immune-related disorders as well.

Original language	English
Qualification	Doctor of Philosophy
Awarding Institution	University Medical Center (UMC) Utrecht
Supervisors/Advisors	Schutgens, Roger, Primary supervisor Voorberg, Jan, Supervisor Boes, Marianne, Co-supervisor Fischer, K, Co-supervisor
Award date	23 Dec 2021
Publisher	Utrecht University
Print ISBNs	978-94-6423-577-7
DOIs	https://doi.org/10.33540/868
Publication status	Published - 23 Dec 2021

Keywords

hemophilia A
factor VIII
anti-FVIII antibodies
inhibitors
immune tolerance induction
ITI
acquired hemophilia A

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10.33540/868

thesis 23-12-2021inhibitors and immune tolerance induction in hemophilia asara - 62066af45621f (1)Final published version, 11.9 MB

https://dspace.library.uu.nl/handle/1874/416536

Cite this

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title = "Inhibitors and immune tolerance induction in hemophilia A, a balancing act",

abstract = "Objective of this thesisTwo main questions have intrigued researchers and physicians in the field of hemophilia for years:How do inhibitors develop and which immunological mechanisms contribute to the eradication of inhibitors during ITI?These are also the two main questions discussed in this thesis. It includes a combination of translational and clinical research, which was aimed to provide an in-depth evaluation of the mechanisms of inhibitor formation and eradication in hemophilia A patients. Elucidation of these mechanisms could contribute to better preventive measures against allo-immunization to FVIII and optimization of current ITI protocols, for example by identifying new pivotal targets fortolerance reinstatement.Increasing knowledge regarding both failure as well as (re)induction of immune tolerance is not only beneficial in hemophilia A, but could be valuable in other immune-related disorders as well.",

keywords = "hemophilia A, factor VIII, anti-FVIII antibodies, inhibitors, immune tolerance induction, ITI, acquired hemophilia A",

author = "Sarah Schep",

year = "2021",

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doi = "10.33540/868",

language = "English",

isbn = "978-94-6423-577-7",

publisher = "Utrecht University",

type = "Doctoral thesis 1 (Research UU / Graduation UU)",

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TY - THES

T1 - Inhibitors and immune tolerance induction in hemophilia A, a balancing act

AU - Schep, Sarah

PY - 2021/12/23

Y1 - 2021/12/23

N2 - Objective of this thesisTwo main questions have intrigued researchers and physicians in the field of hemophilia for years:How do inhibitors develop and which immunological mechanisms contribute to the eradication of inhibitors during ITI?These are also the two main questions discussed in this thesis. It includes a combination of translational and clinical research, which was aimed to provide an in-depth evaluation of the mechanisms of inhibitor formation and eradication in hemophilia A patients. Elucidation of these mechanisms could contribute to better preventive measures against allo-immunization to FVIII and optimization of current ITI protocols, for example by identifying new pivotal targets fortolerance reinstatement.Increasing knowledge regarding both failure as well as (re)induction of immune tolerance is not only beneficial in hemophilia A, but could be valuable in other immune-related disorders as well.

AB - Objective of this thesisTwo main questions have intrigued researchers and physicians in the field of hemophilia for years:How do inhibitors develop and which immunological mechanisms contribute to the eradication of inhibitors during ITI?These are also the two main questions discussed in this thesis. It includes a combination of translational and clinical research, which was aimed to provide an in-depth evaluation of the mechanisms of inhibitor formation and eradication in hemophilia A patients. Elucidation of these mechanisms could contribute to better preventive measures against allo-immunization to FVIII and optimization of current ITI protocols, for example by identifying new pivotal targets fortolerance reinstatement.Increasing knowledge regarding both failure as well as (re)induction of immune tolerance is not only beneficial in hemophilia A, but could be valuable in other immune-related disorders as well.

KW - hemophilia A

KW - factor VIII

KW - anti-FVIII antibodies

KW - inhibitors

KW - immune tolerance induction

KW - ITI

KW - acquired hemophilia A

U2 - 10.33540/868

DO - 10.33540/868

M3 - Doctoral thesis 1 (Research UU / Graduation UU)

SN - 978-94-6423-577-7

PB - Utrecht University

ER -

Inhibitors and immune tolerance induction in hemophilia A, a balancing act

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Keywords

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