The Experience of Screening for SCID

NCT ID: NCT05651113

Last Updated: 2022-12-14

Study Results

Results pending

The study team has not published outcome measurements, participant flow, or safety data for this trial yet. Check back later for updates.

Basic Information

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Recruitment Status

RECRUITING

Total Enrollment

90 participants

Study Classification

OBSERVATIONAL

Study Start Date

2022-11-09

Study Completion Date

2029-03-31

Brief Summary

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This project will evaluate the impact of including Severe Combined Immunodeficiency into the newborn bloodspot screening panel. It will recruit parents and health professionals primarily from the sites where this new form of screening is being trialled well as additional sites where clinicians will be involved in the care of these babies and comparator groups are needed.

The proposed work will consist of two work packages. The first, a mixed-methods study conducted with families from the point of screening information being returned through to the child's fifth birthday. The second, a qualitative interview study conducted with health professionals during the clinical evaluation phase of the national pilot programme.

Detailed Description

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BACKGROUND

Some countries have introduced newborn screening for Severe Combined Immunodeficiency (SCID) and modelling suggests it is probably cost effective in a UK setting. However, there is some uncertainty about this, and questions remain unanswered relating to its potential introduction in UK. The UK National Screening Committee (UKNSC) recommended that an evaluation should take place 'in practice' to try to answer some of these questions. The evaluation will take place in England and will involve about two thirds of babies born from September 2021 to August 2023. This amounts to approximately 800,000 newborns.

Screening for SCID within the NHS Newborn Blood Spot Screening Programme will make use of the blood spot already taken, usually by midwives, on babies at 5 days old. If, as a result of screening of this sample, babies are found to be at higher risk of SCID, parents will be notified, and the result explained. An appointment will be made for them to be seen the next day by an immunologist. At this appointment, the immunologist/clinical nurse specialist will explain the implications again and another blood test (flow cytometry) will be performed that day. The results are usually available the same day. These will be explained to the parent and depending on the result, they may be discharged ('false positive screening result') or appropriate management arranged ('true positive'). Amongst the true positives will be children with SCID, children who have other disorders (temporary or permanent) resulting in severe immune dysfunction and some children who have a problem with a particular part of their immune system (T cells) for whom the outcome is unknown on an individual basis. Although the target of the programme is SCID, children with the disorder are likely to represent only a minority of the true positives.

RATIONALE Previous research in other screening programmes has shown that delivering a false positive result to a participant, or asking them to attend hospital for further tests, following screening can have both short- and long-term deleterious effects. The size and nature of this in screening for SCID was one of the issues that the UKNSC wished to be explored. Preliminary survey and interview studies have started to explore the inherent communication dilemmas, the experiences of parents and health professionals. This project will seek to extend this work by exploring the views and experiences of parents and health professionals who participate in the clinical evaluation.

RESEARCH QUESTION/ AIMS

1. Family Study

1. To explore the effects on families whose babies had a positive SCID screening test (low TRECs). This will include babies with normal results on confirmatory testing, as well as those with a result suggesting they have SCID or another disorder affecting their immune system.
2. To compare views of the above families with families where the initial screening result did not put the child in a higher risk category and families who received a false positive result from another newborn bloodspot screening programme.
3. To compare quality of life of children with SCID whose diagnosis was reached by screening, family history or symptomatic presentation.
4. To measure the quality of life of families who have a child with secondary or syndromic T cell lymphopenia
5. To measure the quality of life and screening experience of children with idiopathic T cell lymphopenia compared to experiences of families who have a child diagnosed with CFSPID.
2. Health Professional Study A) Explore the views and experiences of a sample of midwives who gained consent and took the initial blood sample.

B) To explore the views and experiences of the immunologists/clinical nurse specialists who saw the families at the time of flow cytometry.

N.B. Throughout this work we refer to 'parents' for simplicity. However, we recognise that the family situations of the infants may be more complex, with the existence of legal guardians and non-resident parents. The baseline position is for resident guardians to be included, but if specific circumstances suggest a wider parental group, additional participants may be included.

Conditions

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Severe Combined Immunodeficiency

Study Design

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Observational Model Type

COHORT

Study Time Perspective

OTHER

Study Groups

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Positive NBS for SCID

Parents whose baby was referred to an immunologist due their SCID screening result (minimum n= 10-25)

SCID Screening

Intervention Type OTHER

The addition of newborn bloodspot screening for severe combined immunodeficiency

Normal NBS result

Parents who received a normal screening result (minimum n=10-25)

SCID Screening

Intervention Type OTHER

The addition of newborn bloodspot screening for severe combined immunodeficiency

False positive NBS result

Parents who have received a false positive result elsewhere in screening (minimum n=2-10)

No interventions assigned to this group

CFSPID Designation

Parents who received a CFSPID result (minimum n=\~10)

No interventions assigned to this group

SCID via family history or clinical presentation

Parents whose baby was identified with SCID due to family history or clinical presentation (minimum n=10)

No interventions assigned to this group

Parents whose babies have died

Parents who have received an abnormal screening result (T cell receptor excision circles (TRECs) or who were identified with SCID due to family history or clinical presentation and whose baby has subsequently died (n=\~10)

SCID Screening

Intervention Type OTHER

The addition of newborn bloodspot screening for severe combined immunodeficiency

Interventions

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SCID Screening

The addition of newborn bloodspot screening for severe combined immunodeficiency

Intervention Type OTHER

Eligibility Criteria

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Inclusion Criteria

Participants must be at least 18 years of age. Participants must be able to understand the purpose and implications of the research study.

Where participants are speakers of languages other than English, the research team will arrange a translator.

Exclusion Criteria

Where a child dies after recruitment to the project the team will sensitively check whether the parents wish to continue participation to enable their views to be included.
Minimum Eligible Age

18 Years

Eligible Sex

ALL

Accepts Healthy Volunteers

Yes

Sponsors

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University of Manchester

OTHER

Sponsor Role collaborator

University of Sheffield

OTHER

Sponsor Role collaborator

Coventry University

OTHER

Sponsor Role collaborator

National Health Service, United Kingdom

OTHER_GOV

Sponsor Role collaborator

King's College London

OTHER

Sponsor Role lead

Responsible Party

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Jane Chudleigh

Principal Investigator

Responsibility Role PRINCIPAL_INVESTIGATOR

Locations

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King's College London

London, , United Kingdom

Site Status RECRUITING

Countries

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United Kingdom

Facility Contacts

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Jane Chudleigh, PhD

Role: primary

Pru Holder, MSc

Role: backup

Other Identifiers

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SCID Protocol 5Aug2022 V4

Identifier Type: -

Identifier Source: org_study_id