Exploring parents’ experiences of their infant's 'cystic fibrosis screen positive, inconclusive diagnosis' (CFSPID) designation through interviews

ISRCTN ISRCTN29644140
DOI https://doi.org/10.1186/ISRCTN29644140
IRAS number 278871
Secondary identifying numbers IRAS 278871
Submission date
18/03/2021
Registration date
19/04/2021
Last edited
09/06/2023
Recruitment status
No longer recruiting
Overall study status
Completed
Condition category
Neonatal Diseases
Prospectively registered
Protocol
Statistical analysis plan
Results
Individual participant data
Record updated in last year

Plain English summary of protocol

Background and study aims:
Within the first few days of a newborn baby's life, a small blood sample is taken to test them for genetic health conditions. This is called newborn bloodpsot screening or NBS. The aim of NBS is to identify conditions early, so that prevention or treatment can start as soon as possible. However, new NBS technology can pick up cases where doctors aren't sure if the baby will develop a condition or not. One example of this is 'cystic fibrosis screen positive, inconclusive diagnosis' or CFSPID. Babies with CFSPID have a positive NBS result for cystic fibrosis, but usually do not have any symptoms and usually remain well. Currently, we do not know very much about how CFSPID affects families emotionally, although some research suggests that families do not cope well with the uncertainty of CFSPID. The aim of this study is to interview parents of children with CFSPID, to find out about their experiences. It is hoped that having more knowledge about this experience will help us to improve things for other families in the future.

Who can participate?
Parent(s) of children with CFSPID will be told about the study by their own clinic and can contact the researcher if they want to take part. Parents must be able to understand the study and be willing to talk about their experiences. Parents will not be interviewed if their child has passed away or is seriously ill.

What does the study involve?
The study will involve interviews with parents about their experiences. The interviewer will have a set of questions about their experiences, but the parents are in control of how the interview goes and what they choose to discuss. The interviews will be around 1-2 hours long depending on parents' responses. The interviews will be recorded (just the voices) and typed up, changing names and removing any information that could identify individuals. The study team will then permanently delete the voice recording and analyse the typed interviews for anything significant about CFSPID. A method called 'grounded theory' will be used with the aim to bring together the content from all the interviews to create a theory about how people experience CFSPID. To test the theory as it develops, parents may be asked to take part in a second interview to provide their feedback on the theory so far. This interview will be run and analysed in the same way as the first interview. Parents do not have to take part in a second interview if they do not want to.

What are the possible benefits and risks of participating?
There are no specific benefits to participating although in our experience the study team have found that many people value the opportunity to talk about their experiences.

This study involves people talking about events and topics that they may find upsetting, and so there is a risk that they will become distressed. The interviews have been designed to minimise this risk and the interviewer is trained in how to interview people sensitively. The participants have full control over the interview and may choose to pause or end the interview at any time. The participants will be signposted to sources of support if they are distressed.

Where is the study run from?
The University of Manchester (UK)

When is the study starting and how long is it expected to run for?
September 2019 to May 2023

Who is funding the study?
The study is being carried out as part of the researcher's PhD, which is funded by a scholarship from The University of Manchester (UK)

Who is the main contact?
Faye Johnson, faye.johnson@manchester.ac.uk

Contact information

Ms Faye Johnson
Scientific

Division of Psychology and Mental Health
School of Health Sciences
The University of Manchester
Oxford Road
Manchester
M13 9PL
United Kingdom

ORCiD logoORCID ID 0000-0001-6937-6872
Phone +44 (0)7455935274
Email faye.johnson@manchester.ac.uk

Study information

Study designQualitative interview study
Primary study designObservational
Secondary study designCross sectional study
Study setting(s)Other
Study typeOther
Participant information sheet Not available in web format, please use contact details to request a participant information sheet.
Scientific titleExploring parents’ experiences of CFSPID designation: interview study
Study objectivesThis is a qualitative study to explore parents' experiences of receiving a newborn genetic screening result of 'cystic fibrosis screen positive, inconclusive diagnosis' (CFSPID). Nascent work in this area suggests that uncertain screening results such as CFSPID can have a negative psychological impact on parents. The aim of this study is to explore this further, in order to improve the experience for parents of children with CFSPID, and more broadly to inform debates regarding expanded genomic screening.
Ethics approval(s)Pending, HRA and NHS REC
Health condition(s) or problem(s) studiedCystic fibrosis screen positive, inconclusive diagnosis (CFSPID)
InterventionThis qualitative study will use grounded theory methodology. Parents will be asked to take part in interviews to capture their experiences. The researcher will have a set of topics, questions, or tasks to help explore the area of interest, but the order and precise wording of questions will be determined by the interviewee’ responses. Interview questions are left as open as possible so participants may respond freely and authentically. The length of the interview will be determined by the length of the participants’ responses. As the study progresses and the grounded theory is developing, additional questions will be asked of participants to follow areas of importance and test the emerging theory. Some participants may be contacted to request follow-up interviews to capture their views. This will be determined by ‘theoretical sampling’ – sampling driven by decisions to test the emerging theory. As the researcher begins to build the grounded theory, interviews will explore emerging ideas and test the emerging theory. Interviews will be carried out by in a mutually convenient location of the participant’s choice (this is likely to be the participant’s home if possible), or over video conferencing or phone. Interviews will be audio recorded for transcription and analysed using grounded theory methodology.
Intervention typeOther
Primary outcome measureParent experiences of their infant's 'cystic fibrosis screen positive, inconclusive diagnosis' (CFSPID) designation using audio-recorded and transcribed participant interview responses which will then be analysed for themes pertaining to the research question collected at a single timepoint
Secondary outcome measuresThere are no secondary outcome measures
Overall study start date30/09/2019
Completion date31/05/2023

Eligibility

Participant type(s)Carer
Age groupAdult
Lower age limit18 Years
SexBoth
Target number of participants20
Total final enrolment15
Key inclusion criteria1. The parent or parents of an infant diagnosed with CFSPID following newborn bloodspot screening
2. Aged ≥18 years
3. Able to understand the purpose and implications of the research study
4. Willing and able to talk about their experiences with a researcher. Where participants are speakers of languages other than English, the research team will arrange a translator.
Key exclusion criteria1. Parent of a child who has died or has significant diagnosis of another disease or health issue
2. Barriers to fully understanding the research and communicating with the researcher
Date of first enrolment01/04/2021
Date of final enrolment31/05/2023

Locations

Countries of recruitment

  • England
  • United Kingdom

Study participating centre

The University of Manchester
Oxford Road
Manchester
M13 9PL
United Kingdom

Sponsor information

University of Manchester
University/education

Oxford Road
Manchester
M13 9PL
England
United Kingdom

Phone +44 (0)1612755436
Email FBMHethics@manchester.ac.uk
Website https://www.manchester.ac.uk/
ROR logo "ROR" https://ror.org/027m9bs27

Funders

Funder type

University/education

University of Manchester
Government organisation / Universities (academic only)
Alternative name(s)
The University of Manchester, University of Manchester UK, University of Manchester in United Kingdom, UoM
Location
United Kingdom

Results and Publications

Intention to publish date31/05/2024
Individual participant data (IPD) Intention to shareNo
IPD sharing plan summaryNot expected to be made available
Publication and dissemination planThe full study report will be in the form of Faye Johnson’s PhD thesis which will be accessible through The University of Manchester. We also plan to publish in a high-impact peer-reviewed journal. Due to the nature of qualitative research, raw data will not be available.
IPD sharing planThe datasets generated during and/or analysed during the current study are not expected to be made available due to the sensitive nature of the raw data - transcripts of confidential interviews, discussing sensitive topics, containing potentially identifiable information. The subsequent results publication(s) will contain suitable excerpts of data in the form of anonymised quotations.

Editorial Notes

09/06/2023: Total final enrolment was added.
19/10/2022: The following changes were made to the trial record:
1. The recruitment end date was changed from 30/09/2022 to 31/05/2023.
2. The overall end date was changed from 30/09/2022 to 31/05/2023.
3. The intention to publish date was changed from 30/09/2023 to 31/05/2024.
4. The plain English summary was updated to reflect these changes.
22/03/2021: Trial’s existence confirmed by University of Manchester Faculty of Biology, Medicine and Health: Research Governance.