BACKGROUND JIA includes all chronic arthritis of unknown cause starting before the
age of 16. JIA is the most common rheumatic disease of childhood, affecting
approximately 1 in 1,000 Canadian children. Despite trends to more favorable
outcomes, JIA continues to have disabling sequelae that reduce mobility, quality of
life and future productivity, affecting the child, their families and society as a
whole. After a diagnosis of JIA, families have many questions about the prognosis of
their child, treatments that may be required and their potential side-effects.
Despite numerous studies, our ability to answer these questions remains limited.
International League of Associations for Rheumatology (ILAR) criteria classify
children with JIA into seven categories based on clinical manifestations and
laboratory tests. Current practice recommendations propose use of medications
tailored according to JIA category or according to treatment groups defined by
number of joints affected and features of poor prognosis. In general, initial
treatment with intraarticular corticosteroids and nonsteroidal anti-inflammatory
drugs (NSAIDs) is recommended for JIA affecting a few joints; while methotrexate and
biologic agents are recommended when many joints are affected and for children with
features of poor prognosis.
CAPRI is the collaborative network of all pediatric rheumatology investigators
across Canada, with an outstanding track record of successful collaboration in a
number of research projects. Since 2005 CAPRI investigators have worked on a project
called ReACCH-Out, a team grant funded by the Canadian Institutes of Health Research
(CIHR) to study the impact of arthritis on Canadian children. CAPRI developed a
robust web-based longitudinal data base and enrolled 1,500 newly diagnosed children
with JIA during a five year period. This study has resulted in one of the largest
longitudinal JIA cohorts in the world and six published scientific manuscripts to
date. Most importantly it has provided essential new answers about disease
presentation and short to medium-term prognosis. The initial study was leveraged
into two subsequent studies also funded by CIHR: Biologically Based Outcomes
Predictors in JIA (BBOP) and Linking Exercise, Activity, and Pathophysiology in
Canadian children with Arthritis (LEAP).
Continuation of this effort with a new registry can illuminate longer term prognosis
for JIA patients. The rapidly changing landscape of available therapies necessitates
maintenance and extension of the registry to elucidate the changing prognosis in
different therapeutic eras, and to enable contemporary prognostic counseling to new
patients and their families.
In April 2015, The Arthritis Society provided an infrastructure award to continue,
expand and strengthen a harmonized Canadian registry of patients with JIA, to
support pediatric rheumatology research. This award has been enriched with other
sources to include assessment of medication adverse events, thus providing the
funding that will be used for this CAPRI national JIA registry.
GOALS AND PRINCIPLES
The goal of the CAPRI JIA registry is to prospectively collect information on disease
course, outcomes and medication adverse events among Canadian children with JIA. The
registry will:
Provide answers about the expected disease outcomes and the risks of medication
adverse events for patients and care providers.
Contribute to new insights about JIA and its treatment through yearly updates of
registry findings and publication of scientific papers.
Allow Canadian researchers to answer their own research questions about JIA.
Allow comparison of JIA outcomes and treatments across Canada and with other
jurisdictions, in support of quality improvement initiatives.
The registry will operate following five principles:
Universal: all children newly diagnosed with JIA across Canada will be eligible.
Simple: the registry will require minimum training and time to collect information.
High-Quality: the registry will emphasize quality over quantity of data and the quality
of data will be continually monitored.
Clinician-centered: the registry will ensure that physicians who follow children with JIA
are engaged and see value from their contribution to the registry.
Linkable: the registry will make provisions for future linkages to existing research and
healthcare cohorts, to Statistics Canada data and to other databases.
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3.1 Registry oversight
The registry will be managed through CAPRI. A registry committee of CAPRI has been
established, with the following responsibilities:
Oversight of all registry functions.
Report to CAPRI executive.
Supervision of the registry coordinator.
The registry committee consists of CAPRI members who volunteer for a 2-year renewable
term. The group has elected a chair (Dr. Jaime Guzman) who will be the direct contact for
the registry coordinator, organize teleconferences and provide reports to the chair of
CAPRI. The registry committee chair will sit on the CAPRI executive committee.
Funding from registry sponsors will come to a central registry account. The registry
committee and its chair will be responsible for fund payments, as per agreed CAPRI
budget. Financial accounting will be managed by the registry coordinator.
The registry will be subject to the following reporting processes:
Annual report to registry sponsors including budget use and progress.
Bi-annual report to the CAPRI membership on progress, registry statistics, and
budget. A copy of the bi-annual report will be sent to registry sponsors.
Annual research output report after the first year of registry operations.
Annual update on registry findings to be shared with families and care providers.
3.2 Set up of registry centres All CAPRI centres and pediatric rheumatology practices in
Canada are eligible to participate in the registry. Participating sites will designate a
registry site director for communication and management purposes. Each site will be
responsible for patient enrollment, data entry, and maintenance of appropriate registry
records for their site. The registry site director will manage their own site budget, and
be required to provide annual budget records.
The registry coordinator will assist each registry site in completing the following steps
necessary for set up:
Submission of registry protocol for ethics review by the local ethics board and
amendments as needed
Deciding on the logistics of patient recruitment and registration that best fit with
the centre current practices and processes.
Deciding on the logistics of data collection that best fit their local practices and
processes, including timelines for data submission and locking of the data.
Review and agree on the processes for data quality monitoring.
3.3 Patient recruitment and registration At the medical visit where a new diagnosis of
JIA is made, the pediatric rheumatologist making the diagnosis will introduce the
registry to the patient. The registry site director or delegate will then provide the
family with full information about the registry and ask for their written consent.
ReACCh-Out and LEAP participants will be asked to re-consent for the registry at their
first regular visit.
Registration includes three steps:
Review and signing of the consent form
Assignment of a unique registry identification number by an automated online system
Collection of initial registry data as described below
3.4 Data collection The registry will collect data from the pediatric rheumatologist
caring for the child, and from the child and the child's parent or guardian. Data will be
collected from the physician and families at registration followed by a minimum set of
data at every subsequent clinic visit. Additional data once yearly at a visit close to
the anniversary of the diagnosis will be collected from families. All data will be
collected at clinic visits and there will be no visits booked specifically for registry
purposes.
The primary method of data collection from physicians will be direct entry online using a
secure web site located at a Canadian academic centre, via computer or smart phone. The
secondary method of data collection will be in paper forms for physicians who prefer that
method. Data in paper forms will then be entered online by research assistants.
The primary method of data collection from families will be in paper forms. The answers
will be verified by research assistants and then entered online. The secondary method of
data collection will be direct entry online for centres where existing infrastructure
allows for direct entry by families at the time of the visit.
3.5 Protection of privacy and confidentiality All data collected will be handled in
accordance with federal and provincial personal information legislation. Information that
would allow identification of a child, such as name, date of birth and address will only
be collected in the consent form. This information will be separate from registry
information and will not be available in any online system. All subsequent data
collection will have no personal identifying information, only the registry
identification number.
The de-identified registry information will be stored online in a dedicated RedCap server
at a Canadian academic centre that complies with all encryption and audit regulatory
requirements. As this is an ongoing registry de-identified electronic records will be
kept indefinitely in the secure servers. There is a possibility in the future to have
international collaborations to help with data analysis. In this case, data sent outside
of Canada will not contain any personal identifying information and will be strictly
de-identified.
Any de-identified paper records used during the process of entering information in the
registry will be kept under locked storage for ten years and then destroyed. They are to
be used in case of need for confirmation of the veracity of the data for scientific
publication purposes.
3.6 Data quality monitoring
The continuous monitoring of data quality is an essential function of the registry and
the major responsibility of the registry coordinator. The registry committee and its
chair bear final accountability for the quality of the data entered in the registry. The
following four complementary methods will ensure the data contained in the registry is
complete and of the highest possible quality:
Quality control during data entry: Defines the automatic quality controls set up as
part of data entry for each questionnaire or form.
Daily quality control by registry coordinator: Defines the daily activities of the
registry coordinator to monitor data quality and actions to follow through on issues
identified.
Quality control at periodic centre audits: Defines indications for quality audits,
their periodicity and how their results are assessed and followed through.
Monthly update on quality control to registry chair: defines the components of the
monthly quality update, how it is prepared and how it is assessed by the chair.
3.7 Access to data To assist healthcare provision: Patients and their physicians will
have access to their data to obtain estimates of expected outcomes and risks of side
effects based on analyses of registry data.
To answer research questions: CAPRI and non-CAPRI researchers may request access to
registry data to answer research questions.
To assist planning and program development: Healthcare organizations, registry sponsors,
health authorities and ministries of health services, may request access to summary
statistics or de-identified group data for use in knowledge translation activities or to
assist in their planning of programs and delivery of care.