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01-01-2021 | Immunodeficiency | Original Article

Parents’ Perspectives and Societal Acceptance of Implementation of Newborn Screening for SCID in the Netherlands

Authors: Maartje Blom, Robbert G. M. Bredius, Marleen E. Jansen, Gert Weijman, Evelien A. Kemper, Clementien L. Vermont, Iris H. I. M. Hollink, Willem A. Dik, Joris M. van Montfrans, Mariëlle E. van Gijn, Stefanie S. Henriet, Koen J. van Aerde, Wouter Koole, Arjan C. Lankester, Eugènie H. B. M. Dekkers, Peter C. J. I. Schielen, Martine C. de Vries, Lidewij Henneman, Mirjam van der Burg, on behalf of the SONNET-Study Group

Published in: Journal of Clinical Immunology | Issue 1/2021

Abstract

Purpose

While neonatal bloodspot screening (NBS) for severe combined immunodeficiency (SCID) has been introduced more than a decade ago, implementation in NBS programs remains challenging in many countries. Even if high-quality test methods and follow-up care are available, public uptake and parental acceptance are not guaranteed. The aim of this study was to describe the parental perspective on NBS for SCID in the context of an implementation pilot. Psychosocial aspects have never been studied before for NBS for SCID and are important for societal acceptance, a major criterion when introducing new disorders in NBS programs.

Methods

To evaluate the perspective of parents, interviews were conducted with parents of newborns with abnormal SCID screening results (N = 17). In addition, questionnaires about NBS for SCID were sent to 2000 parents of healthy newborns who either participated or declined participation in the SONNET-study that screened 140,593 newborns for SCID.

Results

Support for NBS for SCID was expressed by the majority of parents in questionnaires from both a public health perspective and a personal perspective. Parents emphasized the emotional impact of an abnormal screening result in interviews. (Long-term) stress and anxiety can be experienced during and after referral indicating the importance of uniform follow-up protocols and adequate information provision.

Conclusion

The perspective of parents has led to several recommendations for NBS programs that are considering screening for SCID or other disorders. A close partnership of NBS programs’ stakeholders, immunologists, geneticists, and pediatricians-immunologists in different countries is required for moving towards universal SCID screening for all infants.

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Title: Parents’ Perspectives and Societal Acceptance of Implementation of Newborn Screening for SCID in the Netherlands
Authors: Maartje Blom
Robbert G. M. Bredius
Marleen E. Jansen
Gert Weijman
Evelien A. Kemper
Clementien L. Vermont
Iris H. I. M. Hollink
Willem A. Dik
Joris M. van Montfrans
Mariëlle E. van Gijn
Stefanie S. Henriet
Koen J. van Aerde
Wouter Koole
Arjan C. Lankester
Eugènie H. B. M. Dekkers
Peter C. J. I. Schielen
Martine C. de Vries
Lidewij Henneman
Mirjam van der Burg
on behalf of the SONNET-Study Group
Publication date: 01-01-2021
Publisher: Springer US
Keywords: Immunodeficiency
Neonatal Screening
Published in: Journal of Clinical Immunology / Issue 1/2021
Print ISSN: 0271-9142
Electronic ISSN: 1573-2592
DOI: https://doi.org/10.1007/s10875-020-00886-4

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