PILOT STUDY: WEB-BASED TREATMENT OF SEVERE FATIGUE AMONG ADOLESCENT PATIENTS WITH AN IMMUNE DYSREGULATION DISORDER: A SINGLE-CASE EXPERIMENTAL DESIGN WITH MULTIPLE MEASUREMENTS.

Chronic severe fatigue is a frequently reported complaint among adolescents
with paediatric immune dysregulation disorders (paediatric rheumatic diseases
and immune deficiencies). Chronic severe fatigue is described as extreme
persistent or recurrent fatigue for at least 3 months with a profound,
debilitating effect on daily life and should therefore be seen as a clinically
relevant symptom. We found that severe fatigue affects 25.1% of adolescents
with a paediatric rheumatic disease, thus making fatigue a common problem among
this patient population. Importantly, this percentage is significantly higher
than in the general population. Furthermore, fatigue among these adolescent
patients is associated with a significant decrease in school presence and
physical activity. Another important result of our study is the finding that in
our subgroup of adolescents with Juvenile Idiopathic Arthritis (JIA), fatigue
was not associated independently with disease activity (measured using
JADAS-criteria), whereas a significant positive correlation was found between
fatigue and pain.
The correlation between fatigue and disabilities among patients suggests that
fatigue may be a viable target for therapeutic interventions designed to
improve physical functioning and school participation in this patient
population. One approach in the treatment of severe fatigue that is promising
is cognitive behaviour therapy (CBT). CBT is a highly effective intervention in
adolescents with chronic fatigue syndrome (CVS), such as FITNET, a web-based
treatment program, but also in adult severely fatigued patients with chronic
conditions like Rheumatoid Arthritis or Multiple Sclerosis. The complaint of
disabling severe fatigue among paediatric immune dysregulation disorders (with
a low and stable disease activity) and chronic fatigue syndrome (CFS) in
adolescents share several similarities in physical symptoms, such as the high
level of experienced fatigue, pain and chronicity. Functional impairment is a
key aspect of both paediatric conditions, thereby affecting most areas of
adolescents lives. It is worth noting that even though the abovementioned CBT
program was not designed specifically to target pain, both fatigue and pain
were reduced in adolescent CFS patients. The proven efficacy of a web-based
treatment programme for adolescent patients with CFS (FITNET) might be an
excellent opportunity for adjusting this treatment programme for adolescent
with disabling fatigue PLUS a paediatric immune dysregulation disorder
(FITNET-PLUS).
In this pilot study web-based CBT is applied to severely fatigued adolescent
patients with an immune dysregulation disorder to test if CBT will lead to a
reduction of fatigue and disabilities.

1. To investigate the feasibility of web-based cognitive CBT among severely
fatigued adolescent patients with an immune dysregulation disorder, and
2. To estimate intervention effects on reducing fatigue, associated chronic
pain and fatigue related disabilities in adolescent patients with an immune
dysregulation disorders.

The research methodology used in the proposed research is a single-case
experiment (SCE). SCEs are experimental, and its purpose is to document causal
relationships between independent and dependent variables.

Participants will receive web-based CBT for fatigue in addition to usual care
for their primary disease. The CBT treatment consists of 20 interactive
treatment modules, over a period of six months. Trained and licensed (by the
Expert Centre of Chronic Fatigue) cognitive behavioural therapists at the
Wilhelmina Children*s Hospital will give the CBT. This treatment is already
part of routine care for severely fatigued adolescents patients with chronic
fatigue syndrome, and adult patients with severe fatigue plus chronic
conditions like rheumatic diseases and multiple scleroses. The treatment is
adapted to the specific characteristics of adolescent patients with an immune
dysregulation disorder.

There are no or only minimal risks involved in participating in the CBT
intervention. The burden is limited and consists of extra travel time for four
face to face sessions with a cognitive behavioural therapist (intake,
consultation session, evaluation post intervention and follow-up), completion
of questionnaires, wearing an actometer for 12 days before the start of the
treatment and doing home-work assignments when the web-based treatment get
started. The program will be adapted to the individual situation of the
patient. Patients fill out several questionnaires at baseline measurement, in
order to determine which topics need to be included in the CBT. During the
complete study period (min. 49 to max. 68 weeks) patients will complete a short
weekly questionnaire. This short weekly questionnaire will be completed online,
within approximately 5-10 minutes. Besides the weekly measures, patients will
fill out three longer questionnaires (approximately 30 minutes), three times in
total (at baseline, post treatment and follow-up).
There are potential benefits for participants, as CBT had already proved to be
a highly effective intervention in reducing fatigue severity and related
disabilities among other chronic diseases, disease-free cancer survivors and
chronic fatigue syndrome. The intervention may also lead to reducing fatigue
severity and increasing social (community) participation among adolescent
patients with an immune dysregulation disorder.