Exercise for pain management in haemophilia

ISRCTN	ISRCTN17454597
DOI	https://doi.org/10.1186/ISRCTN17454597
ClinicalTrials.gov (NCT)	Nil known
Clinical Trials Information System (CTIS)	Nil known
Integrated Research Application System (IRAS)	294992
Protocol serial number	IRAS 294992, CPMS 49802
Sponsor	Royal Free London NHS Foundation Trust
Funder	National Institute for Health Research

Submission date: 18/05/2021
Registration date: 09/07/2021
Last edited: 10/10/2024

Recruitment status: No longer recruiting
Overall study status: Completed
Condition category: Haematological Disorders

Prospectively registered

Protocol

Statistical analysis plan

Results

Individual participant data

Plain English summary of protocol

Background and study aims
Haemophilia is a rare inherited bleeding disorder. If left untreated, those with a severe form of the condition bleed into their joints and muscles. After many repeated bleeds, joints becomes damaged by a condition known as haemophilic arthritis. For most adults over 40 years old with severe haemophilia, effective treatment was not readily available when they were growing up. As a result many have multiple joints affected by painful haemophilic arthritis. This affects quality of life and physical activity. Chronic pain because of this joint damage is a problem for many people with haemophilia, but evidence for effective pain management options is lacking. In other types of arthritis, exercise is effective for pain management, but it is unclear if it could work for people with haemophilia. This study was developed and created with people with haemophilia. The aim is to evaluate if the study can be done (feasibility), what the participants think about taking part in it, and to assess if the outcomes the researchers have chosen are useful.

Who can participate?
Adults aged 18 years and over, with a diagnosis of severe haemophilia A or B and chronic pain.

What does the study involve?
The intervention will be 12 sessions delivered over 6 weeks using telerehabilitation. Participants will do the exercises in their own home with their haemophilia physiotherapist leading the session over webcam. Exercises will be personalised to each person’s own abilities. Each week there will be one individual exercise session and one group exercise session (30-35 minutes each). Weeks 1, 3 and 5 will have a group knowledge sharing and discussion session before the group exercise (30-40 minutes). Participants will complete short questionnaires about their pain, physical function and quality of life at the start and end of the 6-week session and again 12 weeks after finishing the sessions. Participants will also be asked to participate in a short interview after they finish the exercise intervention, where they will be asked to share their experiences of taking part in the study.

What are the possible benefits and risks of participating?
It is unknown if people participating in this study will benefit from doing so. The researchers do not expect any serious side effects or serious consequences from taking part in this study. Some people may experience an increase in their pain after exercise and this may be a normal effect of doing a new activity. The researchers cannot fully eliminate the potential for the exercise to provoke a joint bleed, although they have safety measures in place to reduce this risk as much as possible. The findings from this study will help inform the design of follow up studies with larger numbers of people.

Where is the study run from?
The Royal Free London NHS Foundation Trust (UK)

When is the study starting and how long is it expected to run for?
January 2021 to May 2023

Who is funding the study?
National Institute for Health Research (NIHR) (UK)

Who is the main contact?
Paul McLaughlin
p.mclaughlin@nhs.net

Contact information

Mr Paul McLaughlin
Scientific

Katharine Dormandy Haemophilia Centre and Thrombosis Unit
Royal Free London NHS Foundation Trust
Pond St
London
NW3 2QG
United Kingdom

ORCID ID	0000-0002-5962-7647
Phone	+44 (0)20 7830 2068
Email	p.mclaughlin@nhs.net

Study information

Primary study design	Interventional
Study design	Multi-site uncontrolled before and after feasibility study with embedded qualitative study
Secondary study design	Non randomised study
Study type	Participant information sheet
Scientific title	Physiotherapist-led telerehabilitation and exercise intervention for the management of chronic pain in people with severe haemophilia: a non-randomised, mixed methods feasibility study
Study acronym	REMAP-Haemophilia
Study objectives	The primary objective of this study is to test the feasibility of a remote (telerehabilitation) physiotherapy-led exercise-based rehabilitation programme in people with severe haemophilia who have chronic joint pain. The secondary objective is to collect preliminary data (before and after) on intended patient-reported outcome measures relating to pain, quality of life, function and self-efficacy.
Ethics approval(s)	Approved 13/07/2021, East Midlands – Nottingham 2 Research Ethics Committee (The Old Chapel, Royal Standard Place, Nottingham, NG1 6FS, UK; +44 203 443 6294; nottingham2.rec@hra.nhs.uk), ref: 21/EM/0161
Health condition(s) or problem(s) studied	Chronic pain in people with severe haemophilia
Intervention	This is a feasibility study design with no randomisation included. The intervention consists of a 12-session, low impact, moderate-intensity exercise programme. Each participant will have a personalised programme delivered to them by their own haemophilia physiotherapist. Using an interval training approach, it will include lower and upper body strengthening and general cardiovascular activity, with an additional exercise added every 2 weeks. Each session will last no longer than 30 minutes. The programme will be delivered virtually using a telerehabilitation approach. Each week will have a 1:1 session as well as a group exercise session (max. 5 people). In addition, there will be three knowledge sharing and discussion sessions with the topics of pain with haemophilia, physical activity with haemophilia, arthritis and pain and pacing with pain. The study design incorporates behaviour change techniques. Participants will be asked to record weekly short self-evaluations in a diary for the duration of the exercise intervention. Assessments will be carried out at three timepoints: baseline, end of exercise sessions (6 weeks) and 12 weeks after timepoint 2. Following completion of the 6-week exercise component, participants will be asked to participate in an individual semi-structured interview. This is in keeping with the feasibility design and is to investigate their views and experiences of taking part in the study.
Intervention type	Behavioural
Primary outcome measure(s)	The feasibility aspects of the intervention will be evaluated as follows: 1. Recruitment rate (% uptake) 2. Adherence rate to the intervention procedures 3. Acceptability of the intervention 4. Attrition due to the intervention 5. Safety of the intervention 6. Loss to follow up All will be evaluated from data from screening/recruitment logs, attendance records, participant diaries, participant interviews and review of any recorded adverse events at the end of the study intervention
Key secondary outcome measure(s)	Measured at baseline, week 6 and week 18: 1. Severity and impact of pain measured using the Brief Pain Inventory (BPI) 2. Confidence to be active even with pain measured using the Pain Self Efficacy Questionnaire (PSEQ) 3. Health-related quality of life measured using the EQ 5D 5L questionnaire 4. Pain and quality of life with arthritic joint pain measured using the Musculoskeletal Health Questionnaire (MSK-HQ) 5. Impact of haemophilia on perceived physical function measured using the Haemophilia Activities List (HAL) 6. Self-reporting functional goals for rehabilitation measured using the Patient-Specific Functional Scale (PSFS) 7. Rating change in one's own clinical status measured by Patient Global Impression of Change (PGIC)
Completion date	31/05/2023

Eligibility

Participant type(s)	Patient
Age group	Adult
Lower age limit	18 Years
Sex	Male
Target sample size at registration	15
Total final enrolment	10
Key inclusion criteria	1. People with severe haemophilia A or B (with or without an inhibitor) 2. Aged 18 years and over 3. Self-reported symptoms of chronic pain associated with haemophilic arthropathy (any joint) 4. Willing and able to give informed consent for participation in this study 5. Able to follow instructions 6. Have a good command of written and spoken English 7. Registered at a UK located haemophilia comprehensive care centre with a named physiotherapist 8. Have access to a laptop/tablet at home and sufficient internet connection
Key exclusion criteria	1. Mild or moderate haemophilia A or B 2. Any other inherited bleeding disorder 3. A diagnosis of chronic pain that is not associated with haemophilic arthropathy 4. Severe and/or unstable cardiovascular disease 5. Severe and/or unstable pulmonary disease 6. Uncontrolled diabetes mellitus
Date of first enrolment	01/07/2021
Date of final enrolment	01/12/2022

Locations

Countries of recruitment

United Kingdom
England

Study participating centres

Royal Free Hospital

Royal Free London NHS Foundation Trust
Pond St
London
NW3 2QG
United Kingdom

The Royal London Hospital

Barts Health NHS Trust
80 Newark St
London
E1 2ES
United Kingdom

Basingstoke and North Hampshire Hospital

Hampshire Hospitals NHS Foundation Trust
Aldermaston Rd
Basingstoke
RG24 9NA
United Kingdom

Results and Publications

Individual participant data (IPD) Intention to share	No
IPD sharing plan summary	Data sharing statement to be made available at a later date
IPD sharing plan	The data-sharing plans for the current study are unknown and will be made available at a later date.

Study outputs

Output type	Details	Date created	Date added	Peer reviewed?	Patient-facing?
Results article		08/10/2024	10/10/2024	Yes	No
HRA research summary			28/06/2023	No	No
Participant information sheet	Participant information sheet	11/11/2025	11/11/2025	No	Yes

Editorial Notes

10/10/2024: Publication reference and total final enrolment added.
18/10/2022: The following changes were made to the trial record:
1. The recruitment end date was changed from 28/02/2022 to 01/12/2022.
2. The overall end date was changed from 31/07/2022 to 31/05/2023.
3. The intention to publish date was changed from 01/10/2023 to 01/12/2023.
4. The plain English summary was updated to reflect these changes.
10/01/2022: The following changes were made to the trial record:
1. The recruitment end date was changed from 31/12/2021 to 28/02/2022.
2. The overall end date was changed from 31/05/2022 to 31/07/2022.
3. The intention to publish date was changed from 01/08/2023 to 01/10/2023.
4. The plain English summary was updated to reflect these changes.
5. The ethics approval was added.
21/05/2021: Trial's existence confirmed by the NIHR.