Plain English Summary
Background and study aims
Each year about 800,000 babies in the UK have a blood test taken (called newborn bloodspot screening (NBS)) to screen for specific conditions, which if treated early will improve the child’s health and well-being. In 2015-16, over 10,000 babies were identified as being affected or healthy carriers of a gene for one of the conditions screened for, which include sickle cell disease, cystic fibrosis, metabolic conditions and hypothyroidism. When a positive result occurs, a variety of ways are used to deliver the result but many parents complain about the approaches used. The aim of this study is for parents and health professionals to work together to design interventions to facilitate effective communication of positive NBS results to parents by health professionals. This study includes four phases.
Who can participate?
Health care professors in NBS and parents of children who have received a NBS+ result in the previous 3-12 months.
What does the study involve?
In the first phase of the study, staff are invited to participate in telephone interview lasting 30-45 minutes. In the second phase of the study, staff are observed for 60 minutes on up to 5 occasions and are also invited to take part in semi-structured interviews lasting 60 minutes, participant in a staff feedback event lasting 120 minutes, and work in co-design working groups each lasting 180 minutes. Parents and carers are invited to participate in one hour filmed narrative interviews, feedback events (120 minutes), a staff and parent even (180 minutes) and in the co-designed working groups. In the third phase of the study, staff receive training and are invited for semi-structure interviews. They are also observed for one hour up to five occasions. Parents and carers are invited to one hour semi-structured interviews. In the fourth phase of the study includes stakeholder meeting lasting 180 minutes.
What are the possible benefits and risks of participating?
There will be no direct benefits or anticipated risks for those taking part in the study.
Where is the study run from?
This study is being run by City, University of London (UK) and takes place in hospitals in the UK.
When is the study starting and how long is it expected to run for?
March 2015 to March 2020
Who is funding the study?
National Institute for Health Research (UK)
Who is the main contact?
Dr Jane Chudleigh (Public)
j.chudleigh@city.ac.uk
Trial website
https://www.city.ac.uk/news/2017/september/rethinking-strategies-for-pos
Contact information
Type
Public
Primary contact
Dr Jane Chudleigh
ORCID ID
http://orcid.org/0000-0002-7334-8708
Contact details
City
University of London
Northampton Square
London
EC1V 0HB
United Kingdom
+44 20 7040 0484
j.chudleigh@city.ac.uk
Additional identifiers
EudraCT number
ClinicalTrials.gov number
Protocol/serial number
36339
Study information
Scientific title
Rethinking Strategies for Positive Newborn Screening Result (NBS+) Delivery (ReSPoND): A process evaluation of co-designing interventions to minimise impact on parental emotional well-being and stress
Acronym
ReSPoND
Study hypothesis
Can parents and staff co-design interventions to improve delivery of positive Newborn Screening Result (NBS) results to parents that can be successfully implemented into routine practice in a cost effective manner?
Ethics approval
London-Stanmore Ethics Committee, 01/01/2018, 17/LO/2102
Study design
Non-randomised; Both; Design type: Process of Care, Complex Intervention, Other, Qualitative
Primary study design
Interventional
Secondary study design
Randomised controlled trial
Trial setting
Hospitals
Trial type
Treatment
Patient information sheet
Not available in web format, please use the contact details below to request a patient information sheet
Condition
Genetic screening in newborn
Intervention
This study contains four phases. Phase one is planned for six months. The second phase is conducted after six to 12 months.
Phase 1 (0-6 months):
This involves a conducting a national survey using semi-structured telephone interviews with all 13 NBS-laboratories (NBSL) in England and representatives of clinical teams(n=40) for each of the four condition specific group (CSGs). These will determine current approaches used for communication of NBS+ results from laboratory to parents for each CSG and inform selection of case study sites using a predefined sampling framework. Quantitative data is analysed using descriptive statistics, qualitative data are analysed using content analysis.
PHASE 2: uses Experience-based Co-design in two selected study sites (from Phase 1)
1. Non-participant observation of 20 staff delivering NBS+ results to parents. 15 semi-structured interviews with staff (NBSL staff/Nurses/Consultants/Health Visitors/Midwives/Genetic Counsellors) involved in communicating NBS+ results.
Staff(n=15) meets to identify priorities for improving delivery of NBS+ results. Data will be analysed for themes to inform co-design working groups co-designed working groups (CDWGs) (stage 3) (6-12 months).
2. Filmed, interviews with 20 parents (ensuring representation of CSGs) exploring experiences of receiving NBS+ results to identify key themes. Parents (n=20) view a composite film of the interviews to ensure it is representative of their experiences and to identify emerging issues. Themes identified from parent interviews are made into a 30-minute composite film (6-12 months).
3. Joint staff-parent event in each study site to share experiences and view composite film. Mixed staff-parent focus groups to identify joint priorities for improving delivery of NBS+ results. Thematic analysis is done to identify joint priorities. (12-15 months)
4. Parents and staff from both study sites come together in 4 co-designed working groups (6-8 members each) to produce co-designed interventions for improving delivery of NBS+ results to parents (15-18 months).
PHASE 3 (18-27 months) uses two selected study sites (Phase 1).
20-30 staff involved in delivery of NBS+ results in the study sites are trained to implement the co-designed interventions for the four CSGs concurrently. Success criteria is defined and monitored on a weekly basis during implementation.
A parallel process evaluation underpinned by Normalisation Process Theory is conducted. Non-participant observation of 20-30 staff delivering NBS+ results to parents and semi-structured interviews with 20-25 parents and 20-25 staff to identify healthcare resources required for delivery of the interventions, staff and parental experiences and factors that influence implementation. These qualitative data is also used to determine suitable outcome measures for a future evaluation study. Factors parents identify as influencing experiences during delivery of NBS+ results are compared with the content of measures such as GAD7, PHQ9, Parenting Stress Index, EQ5D and ICECAP-A to determine where most overlap occurs.
Observation and interview data are used to determine how the co-designed interventions impact on parents and which outcomes and healthcare resources are important to evaluate in a future evaluation study. A cost analysis using the NHS perspective, compares costs associated with current communication practices and the new co-designed interventions. For both, resources required are defined and combined with unit costs to produce a total costs.
PHASE 4 (27-30 months):
Key stakeholders(n=10) (NBS co-ordinators/NBSLs staff/health visitors/midwives/parents) meet and the nominal group technique used to reach consensus about the need for, and potential design, of an evaluation study of the co-designed interventions.
Intervention type
Other
Phase
Drug names
Primary outcome measure
Production of co-designed, evaluated interventions for the communication of initial, positive NBS results to parent measured during the process evaluation and health economic analysis during months 18-27.
Secondary outcome measures
Phase 1:
Description of current communication practice measured during interviews with staff at 4-6 months.
Phase 2:
Co-designed interventions for the four condition specific groups produced during the co-design working group at 6-18 months.
Phase 3:
1. The cost of current communication strategies and costs associated with the co-designed interventions measured during the health economic evaluation during months 18-27
2. The acceptability and feasibility of the of the co-designed interventions measured during the process evaluation during months 18-27
3. Choice of potential outcomes measures (GAD 7 PHQ 9 PSI EQ5D and ICECAP-A) for use in a future evaluation study measured during the process evaluation and economic evaluation during months 18-27
Phase 4:
Need for and design of a future evaluation study measured during the focus group during months 27-30.
Overall trial start date
01/03/2015
Overall trial end date
31/12/2020
Reason abandoned (if study stopped)
Eligibility
Participant inclusion criteria
Parents:
Parents of children who have received a NBS+ result in the previous 3-12 months including true positives, false positives and children who later have a cystic fibrosis screen positive, inconclusive diagnosis (CFSPID). This time frame has been chosen as the focus for this research based on feedback from parents of children who have previously received a NBS+ result. It has also been demonstrated that positive NBS can impact on child-parent relationships during the first year of life.
Health professionals:
1. Staff employed in NBS laboratories and involved in the processing of NBS+ results
2. Staff who have been involved in communicating NBS+ results to parents in the last 6 months.
Participant type
Patient
Age group
Adult
Gender
Both
Target number of participants
Planned Sample Size: 160; UK Sample Size: 160
Participant exclusion criteria
Parents:
1. Parent of children who have received a negative NBS result
2. Parents of children with co-morbidities that are likely to influence their perception of receiving the positive NBS result
3. Parents whose baby has died prior to being approached to be involved in the study
4. Inability of parents to understand and give informed consent
5. Parents whose recruitment is contraindicated on psychosocial grounds (identified by their health visitor or specialist nurse)
Health professionals:
1. Staff who have not been involved in communicating positive NBS results to parents in the last 6 months
2. Staff who have personal experience of receiving a positive NBS result
Recruitment start date
01/04/2018
Recruitment end date
30/09/2020
Locations
Countries of recruitment
United Kingdom
Trial participating centre
Addenbrooke's Hospital
Hills Road
Cambridge
CB2 0QQ
United Kingdom
Trial participating centre
Birmingham Children's Hospital
Steelhouse Lane
Birmingham
B4 6NH
United Kingdom
Trial participating centre
Bristol Royal Infirmary
Upper Maudlin Street
Bristol
BS2 8HW
United Kingdom
Trial participating centre
Southmead Hospital
Southmead Road
Westbury-on-Trym
Bristol
BS10 5NB
United Kingdom
Trial participating centre
St Helier Hospital
Wrythe Lane
Carshalton
Carshalton
SM5 1AA
United Kingdom
Trial participating centre
Queen Mary's Hospital for Children
Wrythe Lane
Carlshalton Surrey
Carshalton
SM5 1AA
United Kingdom
Trial participating centre
St James' University Hospital
Beckett Street
Leeds
LS9 7TF
United Kingdom
Trial participating centre
Alder Hey Children’s Hospital
E Prescot Road
Liverpool
L12 2AP
United Kingdom
Trial participating centre
Great Ormond Street Hospital
Great Ormond Street
London
WC1N 3JH
United Kingdom
Trial participating centre
St Thomas' Hospital
Westminster Bridge Road
Lambeth
London
SE1 7EH
United Kingdom
Trial participating centre
Royal Manchester Children's Hospital
Oxford Road
Manchester
M13 9WL
United Kingdom
Trial participating centre
Royal Victoria Infirmary
Queen Victoria Road
Newcastle upon Tyne
NE1 4LP
United Kingdom
Trial participating centre
John Radcliffe Hospital
Headley Way
Headington
Oxford
OX3 9DU
United Kingdom
Trial participating centre
Queen Alexandra Hospital
Southwick Hill Road
Cosham
Portsmouth
PO6 3LY
United Kingdom
Trial participating centre
Sheffield Children's Hospital
Western Bank
Sheffield
S10 2TH
United Kingdom
Trial participating centre
Leeds Children’s Hospital
Leeds
LS2 9NS
United Kingdom
Sponsor information
Organisation
City, University of London
Sponsor details
Northampton Square
London
EC1V 0HB
United Kingdom
Sponsor type
University/education
Website
Funders
Funder type
Government
Funder name
National Institute for Health Research
Alternative name(s)
NIHR
Funding Body Type
government organisation
Funding Body Subtype
National government
Location
United Kingdom
Results and Publications
Publication and dissemination plan
Parents involved in the study and those who form the advisory group will be sent a summary of the research findings. The Programme Manager for the NHS Sickle Cell and Thalassaemia Screening Programme and the Programme Manager for the NHS Newborn Blood Spot Screening Programme have been approached and provided their support with regard to the implementation of these guidelines in the future. Therefore, our study findings will be disseminated on the national NBS websites (https://www.gov.uk/topic/population-screening-programmes/newborn-bloodspot, https://www.gov.uk/topic/population-screening-programmes/sickle-cell-thalassaemia) so that it may be available to staff such as health visitors, midwives and clinical nurse specialists who will be involved in the delivery of the initial positive NBS result.
Additionally, the findings will be disseminated via the website of the relevant charities and support groups associated with these conditions all of whom have been contacted and provided their endorsement for this study (CF Trust, Sickle Cell Society, British Thyroid Foundation, National Society for Phenylketonuria, CLIMB).
In addition, results will be disseminated by the usual academic routes at relevant national and international conferences and published in relevant international peer reviewed journals (including the NIHR HS&DR journal).
IPD sharing statement:
The data sharing plans for the current study are unknown and will be made available at a later date.
Intention to publish date
30/09/2021
Participant level data
To be made available at a later date
Basic results (scientific)
Publication list
2019 protocol in https://www.ncbi.nlm.nih.gov/pubmed/31508239 (added 12/09/2019)
2020 health professionals' experiences in https://pubmed.ncbi.nlm.nih.gov/33004391/ (added 07/10/2020)