Treatment of severely fatigued adolescents with an immune dysregulation disorder

ISRCTN	ISRCTN12612526
DOI	https://doi.org/10.1186/ISRCTN12612526
Secondary identifying numbers	NL56462.041.16

Submission date: 27/12/2016
Registration date: 11/04/2017
Last edited: 11/04/2017

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

Prospectively registered

Protocol

Statistical analysis plan

Results

Individual participant data

Record updated in last year

Plain English Summary

Background and study aims
Paediatric immune dysregulation disorders (PIDD) are a group of childhood disorders where the immune system does not function properly. This may involve autoimmune rheumatic diseases (where the immune system mistakenly attacks healthy joints) and immune deficiencies (where the immune system’s ability to fight infections and disease is compromised or entirely absent). Chronic severe fatigue is a frequently reported complaint amount adolescents with PIDD. It involves extreme, persistent or recurrent fatigue (extreme tiredness) which has lasted for at least three months. It has a profound, debilitating effect on daily life and can lead to a decrease in school attendance and physical activity in those affected. The relationship between fatigue and disabilities among PIDD patients suggests that fatigue may be a viable target for therapeutic interventions designed to improve physical functioning and school participation. One approach in the treatment of severe fatigue that is promising is cognitive behaviour therapy (CBT), a type of talking therapy that aims to change the way people think and behave. The aim of this study is to look at the effectiveness of a web-based CBT treatment programme for adolescent PIDD patients with chronic fatigue at reducing fatigue and disabilities.

Who can participate?
Patients aged between 11 and 18 who have a PIDD and have been suffering from severe fatigue for at least three months.

What does the study involve?
All participants take part in the web-based CBT programme for six months. They are randomly allocated to start receiving the programme at different timepoints. The programme involves 19 interactive modules which include topics such as regulation of sleep-wake pattern; adjustment to a chronic (long-term) disease and managing the uncertainty about disease progression; adjustment to chronic (long-term) pain; reformulation of fatigue related thought patterns; activity regulation and increasing physical activity; and improving social interactions in relation to fatigue. Within a treatment module the participant is asked to keep several journals, answer questions and do several assignments. All answers are sent to the therapist that participants are in weekly email contact with. The main emphasis in the treatment part will be on the setting of goals in behavioural terms to motivate a change in behaviour and accompanying them to normal daily activities including full time education. Patients work toward achieving these goals throughout the programme. At the start of the study and then every week until four weeks after the programme ends (including the time before they start the programme), participants complete a range of questionnaires about their fatigue, pain and disability levels.

What are the possible benefits and risks of participating?
Participants may benefit from a reduction in their fatigue and disability levels. There are no anticipated risks involved with participating in this study.

Where is the study run from?
Wilhelmina Children's Hospital, University Medical Center Utrecht (Netherlands)

When is the study starting and how long is it expected to run for?
August 2016 to January 2018

Who is funding the study?
Univerisity Medical Center Utrecht (Netherlands)

Who is the main contact?
Dr Elise van de Putte
e.vandeputte@umcutrecht.nl

Contact information

Dr Elise van de Putte
Scientific

Wilhelmina Children's Hospital (part of University Medical Center Utrecht)
Postbox 85090
Utrecht
3508 AB
Netherlands

Phone	+31 88 75 545 55
Email	e.vandeputte@umcutrecht.nl

Study information

Study design	Single-centre randomised stepped wedge trial
Primary study design	Interventional
Secondary study design	Randomised controlled trial
Study setting(s)	Hospital
Study type	Treatment
Scientific title	Pilot study: web-based treatment of severe fatigue among adolescent patients with an immune dysregulation disorder: a single-case experimental design with multiple measurements
Study acronym	FITNET-plus
Study hypothesis	The main aim of this pilot study is to estimate the feasibility of web-based cognitive behaviour therapy (CBT) for severe disabling fatigue among a limited number of adolescent patients with an immune dysregulation disorder. The secondary aim is to estimate intervention effects of web-based CBT for fatigue, associated pain, and disabilities.
Ethics approval(s)	University Medical Center Utrecht, 02/08/2016, ref: 16-237-M, NL56462.041.16
Condition	Immune dysregulation disorder
Intervention	All participants will be randomized to receive the web-based cognitive behaviour therapy intervention after 7, 8, 10, 11, 13 (3x), 16, 18 or 24 weeks of randomization (the participants are their own controls during waiting time). The duration of the cognitive behavioural programme is limited to 6 months and the effect will be monitored during the waiting time and the intervention by weekly questionnaires. The web-based programme is developed for both the adolescents and the parents. The programme consists of two parts, a psycho-educational part and a cognitive behavioural part consisting of 19 treatment modules. The therapist activates one or more treatment modules per week, dependent on the progress of the participant. Within a treatment module the participant will keep several journals, answer questions and do several assignments. All answers are sent to the therapist, with whom a weekly email contact will be realised. The follow-up duration is four weeks and will be measured by weekly questionnaires and finished by a follow-up assessment.
Intervention type	Behavioural
Primary outcome measure	Fatigue severity is measured with the fatigue severity scale of the CIS-20 weekly from baseline assessment the end of follow up (four weeks).
Secondary outcome measures	1. School presence is expressed in attended hours / obliged hours * 100% last two weeks measured weekly from baseline assessment the end of follow up (four weeks) 2. Physical functioning is measured with the physical functioning scale of the CHQ weekly from baseline assessment the end of follow up (four weeks) 3. Pain is measured with the with the VAS scores for pain (with scores ranging from 0-100 mm) weekly from baseline assessment the end of follow up (four weeks)
Overall study start date	22/08/2016
Overall study end date	01/01/2018

Eligibility

Participant type(s)	Patient
Age group	Child
Lower age limit	11 Years
Upper age limit	18 Years
Sex	Both
Target number of participants	10
Participant inclusion criteria	1. Age of 11.5 - 18 years old 2. Able to speak, read, and write Dutch 3. Diagnosed with JIA, JDM or CVID 4. Being severely fatigued for at least 3 months (CIS fatigue ≥ 40) 5. Physical functioning subscale (Child Health Questionnaire) score ≤ 85 and/or school absence > 10% 6. At least 3 months before treatment on stable medication (type and dosage) 7. Diagnosed with unexplained fatigue by the general paediatrician
Participant exclusion criteria	1. Suspicion of active disease requiring adaptation of treatment (at the time of inclusion) or a possible visit to the paediatric rheumatologist (during treatment) 2. Cognitive impairment, estimated IQ <70 3. A somatic cause assessed by the general paediatrician or co-morbid psychiatric disorder based on questionnaires that can explain fatigue at baseline assessment before the start of CBT 4. An anxiety score above 44 on the Spielberger State-Trait Anxiety Inventory for Children (STAIC), (average score of Dutch healthy adolescents plus 2 SD) 5. A depression score above 15 on the Children’s Depression Inventory (CDI), (average score of Dutch healthy adolescents plus 2 SD) 6. Presence of suicidal risk, as assessed by CDI and general paediatrician 7. Receiving treatment for a psychiatric disorder at time of inclusion 8. No availability of computer with internet access
Recruitment start date	02/08/2016
Recruitment end date	01/01/2018

Locations

Countries of recruitment

Netherlands

Study participating centre

Wilhelmina Children's Hospital (part of Univerisity Medical Center Utrecht)

Postbox 85090
Utrecht
3508 AB
Netherlands

Sponsor information

University Medical Center Utrecht
Hospital/treatment centre

Postbox 85090
Utrecht
3508 AB
Netherlands

"ROR"	https://ror.org/0575yy874

Funders

Funder type

Hospital/treatment centre

Univerisity Medical Center Utrecht

No information available

Results and Publications

Intention to publish date	31/12/2018
Individual participant data (IPD) Intention to share	Yes
IPD sharing plan summary	Available on request
Publication and dissemination plan	Planned publication is in a high-impact peer reviewed journal.
IPD sharing plan	The datasets generated during and/or analysed during the current study are/will be available upon request from Ms. Linde Nijhof (l.n.nijhof@umcutrecht.nl)