Human T-Cell Lymphotropic Virus Type-1 (HTLV-1), a rare retroviral infection, can result in neurological complications (HTLV Associated Myelopathy) or blood cancer. Key UK risk factors include African or Afro-Caribbean ethnicity, being born in or to parents from endemic countries, and having an HTLV-1-positive partner. Approximately 82.5 million people worldwide are living with HTLV-1. The incidence in England and Wales has risen sharply to around 36,000 cases in 2021, influenced by migration and a higher prevalence among those identifying as Black/Black African/Afro-Caribbean.
Current measures in HTLV clinical care and research focus on factors such as viral load. This does not accurately correlate with disease progression and lacks patient relevance.
A core outcome set(COS) is a consensus-based standardised collection of outcomes used in clinical practice and research. Establishing a COS for HTLV-1 is essential for promoting consistency and enhancing the quality of information on disease progression and intervention effects.
Aim: To develop a Core Outcome Set and identify outcome measurement instruments for the COS, to use in HTLV-1 clinical practice and research.
Research Questions
1. Which outcomes need to be measured in those living with HTLV HTLV-associated myelopathy (HAM) and HTLV-1 Asymptomatic carriers (ACs)?
2. Which existing measurement instruments are best to measure these core outcomes?
Objective 1:
A rapid review identifying outcomes used in HTLV clinical practice and research studies.
Focus groups and semi-structured interviews with stakeholders to identify their outcomes of interest. The resulting long list of outcomes will be refined and used in a multi-stakeholder, two-round modified Delphi survey, where the outcomes will be scored for importance. Consensus criteria have been set, and the core outcome set will be agreed upon in a multi-stakeholder consensus meeting.
Objective 2:
Identify and psychometrically review existing outcome measurement instruments. Finally, a
consensus meeting will determine the selection of one measure for each identified outcome domain.
Objectives:
1. Identify what to measure:
a. Identify and refine a list of outcome domains for those living with HAM and ACs.
b. Prioritise and reach a consensus regarding these domains from the perspective of those living with HAM and ACs, clinicians and researchers
2. Identify how to measure:
a. Which existing measures could be used for each domain
b. Evaluate and appraise the existing measures available for each domain
c. Reach a consensus on the most appropriate existing measure for each domain of the COS
Inclusion criteria for all work packages:
HTLV-1-Associated-Myelopathy and HTLV-1-Asymptomatic carriers; significant others of those with HAM and specialist clinicians and researchers working with people with HTLV-1.
PI: Adine Adonis PhD Student University of Manchester
Prof Jamie Kirkham University of Manchester (Primary Supervisor)
Prof Caroline Sanders University of Manchester
Prof Lucie Byrne-Davies University of Manchester
Prof Ramona Moldovan University of Manchester
Disease Category: Infectious disease
Disease Name: Human T-Cell Lymphotropic Virus Type 1 (HTLV-1)
Age Range: 18 - 120
Sex: Either
Nature of Intervention: Any
- Clinical experts
- Consumers (caregivers)
- Consumers (patients)
- Families
- Methodologists
- Patient/ support group representatives
- Researchers
- Service providers
- Service users
- COS for clinical trials or clinical research
- COS for practice
- Recommendations for outcome measures (measurement/how)
- Consensus meeting
- Delphi process
- Focus group(s)
- Interview
- Literature review
An iterative mixed-methods study employing both qualitative and quantitative methods will be used.
The Core Outcome Measures in Effectiveness Trials (COMET) framework to guide the development of this COS. The study protocol will be developed following Core-Outcome-Set-STAndardised Protocol-Items and Core-Outcome-Set-STAndards-for-Development guidelines for COS development.