Value based health care in systemic sclerosis: what is the optimal follow-up for patients with systemic sclerosis?

Systemic sclerosis (SSc) is a complex multisystem rheumatic autoimmune disease.
Currently, evidence based guidelines for frequency and intensity of follow-up
of SSc patients are not available. Based on expert consensus annual extensive
evaluation is recommended. To provide comprehensive multidisciplinary care
integrated with evaluation of organ involvement and as such, reducing health
care utilization while improving the quality of care for the patient, the
*Leiden Combined Care in SSc (CCISS) pathway* was started in 2009. Data
collected on disease progression in the patients that participate in this care
pathway show that 50% of the patients have relatively mild disease, without any
disease progression over time. Therefore there is a need for tailormade care in
SSc patients in accordance to disease activity. To enable this, a prediction
model was developed that can identify patients with low risk for disease
progression.

To evaluate in SSc patients with low risk for disease progression 1) whether
assessment in an outpatient clinic setting is an acceptable alternative for
evaluation in the Care Pathway. Outcome parameters we will evaluate include 1)
health care utilization, 2) patients* perception of the disease and delivery
of care, 3) health-related quality of life and 4) disease progression. Health
care utilization as primary outcome is defined as number of contacts with heath
care providers during 12 months.

This study consists of two parts:
1) Applying our prediction model to categorize SSc patients, who have had at
least two Care Pathway visits, into three risk categories for disease
progression: low, intermediate or high risk group.
2) Among the SSc patients in the low and intermediate risk category for disease
progression, a non-inferiority randomized trial will be performed. Patients
with a low or intermediate risk score will be randomized for an outpatient
clinic visit or a visit in the care pathway. The year after the *study visit*,
everyone one will be seen in the care pathway.

Based on a machine learning assisted prediction model (unpublished data of our
research group) the patients identified as low or intermediate risk profile for
disease progression are randomized in two groups: 1) follow-up in the
outpatient clinic (intervention) and 2) usual care at the care pathway (control
group)

This is a non-therapeutic study and the risks for participants are considered
low. Only SSc patients who have had two care pathway evaluations and with a
predicted low or intermediate risk for disease progression will randomize for
the intervention. The cut-offs for defining risk groups were chosen with the
aim not to miss disease progression (high negative predictive value); amongst
patients classified as low risk no disease progression was observed, while
among the patients classified as intermediate risk the chance for progression
was < 10%. At all times, patients are allowed to contact the care providers of
the Care Pathway in case of questions or symptoms. The number of physical
examinations and site visits will not be different between the groups according
to the study design. During the follow-up period patients will be asked to
complete two additional questionnaires to collect data on health care
utilization, and two additional questionnaire on illness perceptions. We try to
limit the physiological discomfort associated with participation by given
advice and information on the low risks of this study and by assuring patients
that they will receive the high quality care in both arms of the study. We will
also investigate physiological discomfort with the use of the brief illness
perception questionnaire (BIPQ). After one year, all patients will be evaluated
at the SSc care pathway.