Knowledge about newborn screening (NBS) is an important factor for parents to make an informed decision about participation. In Europe, countries inform parents differently about their NBS program, potentially including different knowledge aspects in their information. The aim of this study was to assess twenty-six European parental information products and to analyze their knowledge aspects through a content analysis. The analyzed aspects were compared to a list of eight knowledge aspects from scientific literature. The list includes aspects important for parents' decision-making, such as the purpose of screening. The study showed that most of the eight knowledge aspects are included in NBS information products of the majority of countries. However, there were differences between countries, for example in the amount of detail and phrasing of the information. Additional relevant knowledge aspects have also been identified and are recommended to optimize information products, such as the handling of residual bloodspot samples. This study only assessed knowledge aspects in information products meant for printing, but many countries also use other communication methods, and the impact on knowledge of the delivery of the information needs further study. Preferences of parents on alternative communication methods need to be considered and evaluated on their effectiveness.

Centre for Health Protection National Institue for Public Health and the Environment 3720 BA Bilthoven The Netherlands

Department of Clinical Genetics Section Community Genetics Amsterdam University Medical Center Vrije Universiteit Amsterdam 1007 MB Amsterdam The Netherlands

Department of Pediatrics and Inherited Metabolic Disorders Charles University 1st Faculty of Medicine and General University Hospital Praque 128 08 Prague 2 Czech Republic

Institute for Medical Humanities 1st Faculty of Medicine Charles University Praque 128 08 Praque 2 Czech Republic

International Society for Neonatal Screening 3721CK Bilthoven The Netherlands

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Korenev S., Lemonde H., Cleary M., Chakrapani A. Newborn Screening for Inborn Errors of Metabolism. Paediatrics and Child Health; London, UK: 2019.

El-Hattab A.W., Almannai M., Sutton V.R. Newborn Screening: History, Current Status, and Future Directions. Pediatr. Clin. N. Am. 2018;65:389–405. doi: 10.1016/j.pcl.2017.11.013. PubMed DOI

Therrell B.L., Padilla C.D., Loeber J.G., Kneisser I., Saadallah A., Borrajo G.J., Adams J. Current status of newborn screening worldwide. Semin. Perinatol. 2015;39:171–187. doi: 10.1053/j.semperi.2015.03.002. PubMed DOI

Bhattacharya K., Wotton T., Wiley V. The evolution of blood-spot newborn screening. Transl. Pediatr. 2014;3:63–70. PubMed PMC

E Jansen M., Metternick-Jones S.C., Lister K.J. International differences in the evaluation of conditions for newborn bloodspot screening: A review of scientific literature and policy documents. Eur. J. Hum. Genet. 2016;25:10–16. doi: 10.1038/ejhg.2016.126. PubMed DOI PMC

Loeber J.G., Burgard P., Cornel M.C., Rigter T., Weinreich S.S., Rupp K., Hoffmann G.F., Vittozzi L. Newborn screening programmes in Europe; arguments and efforts regarding harmonization. Part 1—From blood spot to screening result. J. Inherit. Metab. Dis. 2012;35:603–611. doi: 10.1007/s10545-012-9483-0. PubMed DOI

Pollitt R.J. Introducing new screens: Why are we all doing different things? J. Inherit. Metab. Dis. 2007;30:423–429. doi: 10.1007/s10545-007-0647-2. PubMed DOI

Franková V., Members of the European Society of Human Genetics (ESHG)-EuroGentest Quality Sub-Committee. Driscoll R.O., Jansen M.E., Loeber J.G., Kožich V., Bonham J., Borde P., Brincat I., Cheillan D., et al. Regulatory landscape of providing information on newborn screening to parents across Europe. Eur. J. Hum. Genet. 2021;29:67–78. doi: 10.1038/s41431-020-00716-6. PubMed DOI PMC

Botkin J.R., Rothwell E., Anderson R.A., Rose N.C., Dolan S.M., Kuppermann M. Prenatal education of parents about newborn screening and residual dried blood spots a randomized clinical trial. JAMA Pediatr. 2016;170:543–549. doi: 10.1001/jamapediatrics.2015.4850. PubMed DOI PMC

Ulph F., Dharni N., Bennett R., Lavender T. Consent for newborn screening: Screening professionals’ and parents’ views. Public Health. 2020;178:151–158. doi: 10.1016/j.puhe.2019.08.009. PubMed DOI

Etchegary H., Nicholls S.G., Tessier L., Simmonds C., Potter B.K., Brehaut J.C., Pullman D., Hayeems R., Zelenietz S., Lamoureux M., et al. Consent for newborn screening: Parents’ and health-care professionals’ experiences of consent in practice. Eur. J. Hum. Genet. 2016;24:1530–1534. doi: 10.1038/ejhg.2016.55. PubMed DOI PMC

Ulph F., Wright S., Dharni N., Payne K., Bennett R., A Roberts S., Walshe K., Lavender T. Provision of information about newborn screening antenatally: A sequential exploratory mixed-methods project. Health Technol. Assess. 2017;21:1–240. doi: 10.3310/hta21550. PubMed DOI PMC

Huang M.-C., Lee C.-K., Lin S.-J., Lu I.-C. Parental consent for newborn screening in southern Taiwan. J. Med. Ethics. 2005;31:621–624. doi: 10.1136/jme.2004.010074. PubMed DOI PMC

Charles T., Pitt J., Halliday J., Amor D.J. Implementation of written consent for newborn screening in Victoria, Australia. J. Paediatr. Child Health. 2013;50:399–404. doi: 10.1111/jpc.12484. PubMed DOI

Detmar S., Hosli E., Dijkstra N., Nijsingh N., Rijnders M., Verweij M. Information and Informed Consent for Neonatal Screening: Opinions and Preferences of Parents. Birth. 2007;34:238–244. doi: 10.1111/j.1523-536X.2007.00176.x. PubMed DOI

Hargreaves K., Stewart R., Oliver S. Newborn screening information supports public health more than informed choice. Health Educ. J. 2005;64:110–119. doi: 10.1177/001789690506400203. DOI

Michie S., Dormandy E., Marteau T.M. Informed choice: Understanding knowledge in the context of screening uptake. Patient Educ. Couns. 2003;50:247–253. doi: 10.1016/S0738-3991(03)00044-2. PubMed DOI

Nicholls S.G., Southern K.W., Hons B., Mres M.B.C. Mrcp Informed Choice for Newborn Blood Spot Screening in the United Kingdom: A Survey of Parental Perceptions. Pediatrics. 2012;130:e1527–e1533. doi: 10.1542/peds.2012-1479. PubMed DOI

Van der Pal S., Otten W., Detmar S. Evaluatie van de voorlichting aan ouders over de hielprikEvaluation of the health information for parents about the Dutch Neonatal Screening Program. TSG J. Health Sci. 2010;88:449–453.

Hewlett J., Waisbren S.E. A review of the psychosocial effects of false-positive results on parents and current communication practices in newborn screening. J. Inherit. Metab. Dis. 2006;29:677–682. doi: 10.1007/s10545-006-0381-1. PubMed DOI

Vernooij-Van Langen A.M.M., Van Der Pal S.M., Reijntjens A.J.T., Loeber J.G., Dompeling E., Dankert-Roelse J.E. Parental knowledge reduces long term anxiety induced by false-positive test results after newborn screening for cystic fibrosis. Mol. Genet. Metab. Rep. 2014;1:334–344. doi: 10.1016/j.ymgmr.2014.07.006. PubMed DOI PMC

Hayeems R.Z., Miller F.A., Vermeulen M., Potter B.K., Chakraborty P., Davies C., Carroll J.C., Ratjen F., Guttmann A. False-Positive Newborn Screening for Cystic Fibrosis and Health Care Use. Pediatrics. 2017;140:e20170604. doi: 10.1542/peds.2017-0604. PubMed DOI

Fitzgerald C., Heery E., Conneally N., Linnane B., George S., Fitzpatrick P. An evaluation of pregnant women’s knowledge and attitudes about newborn bloodspot screening. Midwifery. 2017;45:21–27. doi: 10.1016/j.midw.2016.11.007. PubMed DOI

General Medical Council . Seeking Patients’ Consent: The Ethical Considerations. General Medical Council; London, UK: 1998.

Kemper A.R., Fant K.E., Clark S.J. Informing Parents About Newborn Screening. Public Health Nurs. 2005;22:332–338. doi: 10.1111/j.0737-1209.2005.220408.x. PubMed DOI

Clayton E.W. Talking with parents before newborn screening. J. Pediatr. 2005;147:S26–S29. doi: 10.1016/j.jpeds.2005.08.001. PubMed DOI

Jansen M.E., Bosch L.J.M.V.D., Hendriks M.J., Scheffer M.M.J., Heijnen M.-L., Douglas C.M.W., Van El C.G. Parental perspectives on retention and secondary use of neonatal dried bloodspots: A Dutch mixed methods study. BMC Pediatr. 2019;19:1–11. doi: 10.1186/s12887-019-1590-8. PubMed DOI PMC

Marteau T.M., Msc E.D., Michie S. A measure of informed choice. Health Expect. 2001;4:99–108. doi: 10.1046/j.1369-6513.2001.00140.x. PubMed DOI PMC

Michie S., Dormandy E., Marteau T.M. The multi-dimensional measure of informed choice: A validation study. Patient Educ. Couns. 2002;48:87–91. doi: 10.1016/S0738-3991(02)00089-7. PubMed DOI

Moody L., Choudhry K. Parental views on informed consent for expanded newborn screening. Health Expect. 2011;16:239–250. doi: 10.1111/j.1369-7625.2011.00710.x. PubMed DOI PMC

Botkin J.R., Lewis M.H., Watson M.S., Swoboda K.J., Anderson R., Berry S.A. Parental permission for pilot newborn screening research: Guidelines from the NBSTRN. Pediatrics. 2014;133:e410–e417. doi: 10.1542/peds.2013-2271. PubMed DOI PMC

Plass A.M.C., Pieters T., Van El C.G., Cornel M.C. Neonatal Screening for Treatable and Untreatable Disorders: Prospective Parents’ Opinions. Pediatrics. 2009;125:e99–e106. doi: 10.1542/peds.2009-0269. PubMed DOI

Franková V., Dohnalová A., Pešková K., Hermánková R., O’Driscoll R., Ješina P. Factors influencing parental awareness about newborn screening. Int. J. Neonatal. Screen. 2019;5:35. PubMed PMC

Moorhead S.A., Hazlett D.E., Harrison L., Carroll J.K., Irwin A., Hoving C. A New Dimension of Health Care: Systematic Review of the Uses, Benefits, and Limitations of Social Media for Health Communication. J. Med. Internet Res. 2013;15:e85. doi: 10.2196/jmir.1933. PubMed DOI PMC

Kessels R.P.C. Patients’ memory for medical information. J. R. Soc. Med. 2003;96:219–222. doi: 10.1258/jrsm.96.5.219. PubMed DOI PMC

Therrell B.L., Lloyd-Puryear M.A., Ohene-Frempong K., Ware R.E., Padilla C.D., Ambrose E.E., Barkat A., Ghazal H., Kiyaga C., Mvalo T., et al. Empowering newborn screening programs in African countries through establishment of an international collaborative effort. J. Community Genet. 2020;11:253–268. doi: 10.1007/s12687-020-00463-7. PubMed DOI PMC