Background and aims

In the United Kingdom, newborn screening for phenylketonuria (PKU) was introduced on a national level in 1969 and for congenital hypothyroidism (CHT) in 1981. Recent developments in newborn blood spot screening in the UK, however, have highlighted the need to re-examine information and communication needs for parents and health professionals in this area.  These developments include: policy decisions in 2000 and 2001 to introduce screening for cystic fibrosis and sickle cell disorders respectively; innovations in screening technologies that have increased the number of disorders that can be detected from dried blood spot specimens; and raised expectations for informed consent.

A recent systematic review of existing reviews on newborn blood spot screening and communication about screening revealed gaps in the research literature on communication for newborn screening.1  It also highlighted the lack of primary research on the information and communication needs of parents and health professionals in the newborn period.

Research carried out by the Parent Support Research Team of the UK Newborn Screening Programme Centre has aimed to address some of the gaps in the literature.   A qualitative study was undertaken to assess parents’ and health professionals’ information needs.  In addition, the team carried out this survey of existing information resources on newborn blood spot screening for parents and health professionals, which is the focus of this report.

The aims of the survey were to:

• Identify existing information resources on newborn blood spot screening available to parents and health professionals internationally
• Develop an appraisal tool to evaluate the quality of the existing information resources, with a view to identifying strengths and weaknesses of the available information
• Draw on the survey of leaflets to inform the development of our own information resources: initially, a standardised pre-screening leaflet for parents throughout the UK.

Methods

Leaflets and other information resources on newborn blood spot screening were sourced through internet websites, health service providers and support organisations in the UK.

Information resources on newborn blood spot screening and the conditions screened for were catalogued electronically for easy retrieval and analysis.  To identify leaflets that were relevant to the task of developing a general parent leaflet, all leaflets that described the heel-prick test were included in the survey of leaflets.  Those that merely mentioned the procedure and/or contained information about the conditions only were excluded. 

An appraisal tool was developed by adapting DISCERN criteria for judging the quality of patient information on treatment choices, and National Screening Committee (NSC) criteria for making policy decisions about the implementation of screening programmes. Two researchers tested the tool by applying it to a limited number of leaflets, compared their answers and achieved agreement on what information was required to meet our criteria.  The finalised tool contained questions grouped under the following sub-headings: purpose of the leaflet; reasons for screening; process and consequences of screening; follow-up to screening; production of the leaflet.

Findings

Some of the key findings include:

• Of the 106 leaflets included in the survey, most were for parents (79/106) and this was evident in the title of about half of them (42/79) and in the text of the great majority (67/79).
• The vast majority of the leaflets (91/106) explained the aims of screening; three-quarters provided some information on the natural history of at least one of the conditions (79/106). Less than half of the leaflets (43/106), however, mentioned prevention or the difficulties of prevention.
• The great majority of leaflets described the procedures for doing the blood spot screening test particularly how the blood sample is taken (93/106) and the possibility of repeat testing (76/106)
• Two-thirds of leaflets (68/106) did not indicate whether or not screening was compulsory or mandatory, though many of these implied that screening was routine.
• Less than half of the leaflets indicated when parents would receive screening results (45/106). 
• About half the leaflets contained information about the limitations or possible harms of screening (55/106).
• The majority of leaflets indicated the need for follow-up tests to confirm diagnosis after a positive screening result (71/106).
• The vast majority of leaflets cited no source of evidence for the information provided (89/106).
• Over half the leaflets indicated the date of development and/or review (56/106), but the great majority (90/106) did not mention how the information was developed.
• Overall, the leaflets/information sheets rated better on issues rooted in DISCERN criteria for the provision of information on treatment choices, than on NSC criteria for choices regarding whether or not to introduce a screening programme. 

Conclusions

Most leaflets provided information on their purpose, the aims of screening, the conditions screened for, the screening procedure, the need for follow-up to confirm positive screening results, and treatment for the conditions.  The appraisers, however, were generous when using the appraisal tool.  In some cases the information provided in the leaflets barely met the requirement.  Whilst most leaflets mentioned the benefits of screening, few addressed the issue of the possible harms, particularly false results, indicated that screening was a choice, discussed alternatives to screening, or gave information about when and how parents would receive results.  Fewer still provided any sources of evidence for the information provided or how the information was developed.

Recommendations

We recommend:

• the use of DISCERN with topic specific guidelines as a starting point for developing patient information;
• appraising patient information with DISCERN and a topic specific tool to access good practice in information provision.

This report should be cited as: Hargreaves K, Stewart R, Oliver S (2005) Survey of information resources on newborn blood spot screening for parents and health professionals: a systematic review. London: Social Science Research Unit, Institute of Education, University of London.