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Canadian Paediatric Surveillance Program
Overview and History
Overview
Surveillance is an important part of the practice of medicine, allowing for the tracking and studying of conditions. Not only can surveillance help determine the burden of disease and assess interventions to prevent the occurrence of a disorder, the information collected can also allow development of future health policies to address the needs of patients with these conditions.
The difficulty in recognizing rare disease can result in delayed diagnosis, increasing the risk of preventable complications or death. Emerging infections will initially be rare and may remain undetected allowing infection to spread extensively before action is taken. All such diseases are difficult to study as their low frequency often means that little may be known about their etiology, clinical spectrum, sequelae, pathological features, diagnosis, treatment and management. Data collection from a large and often geographically diverse population is required to generate a sufficient number of cases to derive meaningful data.
The CPSP is designed to study uncommon disorders with high morbidity and mortality in childhood or rare complications of more common diseases of such low frequency that data collection nationally is required to generate a sufficient number of cases to derive meaningful results. More than 2,500 paediatricians and paediatric subspecialists have been enrolled as participants in this active surveillance system. Other participants, such as paediatric neurologists, allergists, medical geneticists and intensivists are enrolled in the program when research studies indicate their participation. These physicians provide health care to over seven million Canadian children and youth.
History
1986 – Establishment of the British Paediatric Surveillance Unit
National surveillance of rare paediatric conditions had its inception in 1986 with the establishment of the British Paediatric Surveillance Unit (BPSU). The formal establishment was precipitated by the failure of paediatricians to report cases of necrotizing fasciitis, Reye’s syndrome, Kawasaki disease, and hemolytic/hemorrhagic shock syndromes to the British Paediatric Association and the Public Health Laboratory Service.
1996 – Establishment of a joint pilot program between the Canadian Paediatric Society and the Laboratory Centre for Disease Control
In 1995, the Canadian Paediatric Society and the Laboratory Centre for Disease Control (Health Canada) formed a small working group to set up a national paediatric surveillance program modeled on the British Paediatric Surveillance Unit. After several months of planning and consultation, a joint pilot program for the surveillance of rare paediatric diseases and conditions was established and commenced activity in January 1996. Three conditions were selected for the pilot: acute flaccid paralysis (AFP), congenital rubella syndrome (CRS), and group B streptococcal infection (GBS). AFP was selected because the CPSP provided a means of both monitoring suspected cases of paralytic poliomyelitis and providing evidence of the elimination of indigenous wild poliovirus transmission towards certification of a polio-free status in Canada and the rest of the Americas in September 1994. CRS was selected because of the need to monitor progress towards attaining the goal of elimination of indigenous rubella infection during pregnancy by the year 2000. GBS was chosen because no Canadian data existed on the incidence of the infection.
1996 – Establishment of the Steering Committee
During 1996, a Steering Committee was established to ensure that the CPSP would be developed to serve the health needs of Canadian children and youth as well as the research needs of the health care community whose prime concern is the care and health of children. Membership on the Steering Committee included representation from the Canadian Paediatric Society, the Laboratory Centre for Disease Control, the Federal/Provincial Advisory Committee on Epidemiology, Chief Medical Officers of Health, and the Assembly of Canadian University Paediatric Department Heads. Also included were liaison representatives from various organizations, such as the British Paediatric Surveillance Unit, the Canadian Association of Child Neurology, and the Canadian College of Medical Geneticists.
1996 – Response rate
The overall response rate of 76% was very much in line with the results of other international surveillance units during the first year of their operations, but below the CPSP goal of 90% response.
1996 – International developments
Following the success of the British Paediatric Surveillance Unit, the same methodology was adopted, and adapted in the 1990s to other countries whose paediatric services were amenable to active surveillance. By 1996/97 six other units, including Canada, had been established: Australia, Germany, the Netherlands, Switzerland and Malaysia. A meeting of the European Units was held in Leiden during January 1995 with a view to sharing and standardizing protocols, case definitions and study periods.
1996 – Lessons learned
The pilot study highlighted the importance of mailing early reminders to participants who did not send in their monthly report cards, as demonstrated by response rates of 89% and 88% for January and February, respectively, compared to 61% and 64% for November and December. Three reminders were sent for the first two months while no reminders were sent for the final two months of the year. The pilot phase enabled the CPSP to evolve into a smoother, more efficient system as a result of the experience gained throughout the year.
1997 – Continued growth
The CPSP continued to grow and build in 1997. Three additional diseases were added to the program: Creutzfeldt-Jakob disease, hemorrhagic disease of the newborn and neural tube defects. At the same time, surveillance of group B strep was discontinued, partly in response to an increase in the number of investigators who had commenced studies on GBS following the publication of guidelines for the management of GBS during pregnancy and delivery. Initial overall response rates by paediatricians climbed significantly from 76% in 1996 to 82% in 1997. Follow-up rates for detailed reporting forms improved from 54% in 1996 to 89% in 1997.
A poster presentation at the Canadian Paediatric Society’s (CPS) annual meeting profiled the evolution of the CPSP and its findings to date. The presentation provided CPS members with the opportunity for direct feedback on current and future directions of the program.
On the international scene, Latvia and New Zealand established national paediatric surveillance units.
1998 – Increased self-direction
While no new studies were added to the CPSP, surveillance of neural tube defects concluded in 1998. The program continued to evolve becoming more self-directed, as in the summer of 1998 a call was issued for research proposals. The call was successful with six additional new studies approved for inclusion in the program pending confirmation of financial support and ethical approval: anaphylaxis, cerebral edema in diabetic ketoacidosis, idiopathic interstitial lung disease, perinatal hemochromatosis, pyridoxine-dependent status epilecticus, and vitamin D-deficiency rickets. With more than 2,100 paediatricians participating in the program, the CPSP became the largest paediatric surveillance unit in the world.
The response rate for initial reports was 86%, a slight increase over the 1997 rate of 82%. Detailed reporting forms were received for 93% of the 258 cases reported in 1998.
The British Paediatric Surveillance Unit requested that Canada serve as a control for one of its studies; consequently, the study on the surveillance of Creutzfeldt-Jakob disease was expanded to include cases of progressive, intellectual and neurological deterioration. At the same time, the CPSP welcomed the participation of child neurologists for this study through a partnership with the Canadian Association of Child Neurology.
In August 1998, during the 22nd International Congress of Paediatrics in Amsterdam, the International Network of Paediatric Surveillance Units (INoPSU) was formed. The founding units were: Australia, United Kingdom, Canada, Germany, Latvia, Malaysia, the Netherlands, New Zealand, Papua New Guinea, and Switzerland. The CPSP invited INoPSU to host its first scientific meeting during the Canadian Paediatric Society’s annual meeting in June 2000, affording Canadian paediatricians an excellent opportunity to benefit first-hand from this research dissemination.
1998 – Lessons learned
Final study results on neural tube defects indicated that case ascertainment was incomplete. In retrospect, it became clear that establishing a network of collaborators is of prime importance when studying the occurrence of conditions that involve a number of health care professionals. To ensure that case ascertainment is complete, all collaborators must be involved. In this case, extending the list of participants to include other subspecialities, such as obstetricians and geneticists, would have ensured that case ascertainment results were more complete.
1999 – CPSP gains recognition
The CPSP started to gain recognition among paediatric researchers as the number of queries about the program increased throughout the year. A number of new studies were approved to commence in 2000: anaphylaxis, Smith-Lemli-Opitz, hemolytic uremic syndrome with either diarrhea or strep pneumoniae, and neonatal herpes. Paediatric subspecialists in allergy, medical genetics, and intensive care were added as participants in the program for these new studies. Two letters of intent were received for surveillance studies on primary pulmonary hypertensive angiopathy and lead toxicity.
The International Network of Paediatric Surveillance Units met during the British Paediatrics and Child Health meeting in Edinburgh, Scotland, in November 1999. Luxembourg and Belgium expressed interest in establishing national paediatric surveillance units.
1999 – Lessons learned
Overall, study results from 1999 were most encouraging. Not only were cases of such rare conditions as Creutzfeldt-Jakob disease and subacute sclerosing panencephalitis found, but CPSP participants, recognizing the importance of the program, completed and returned a record number of questionnaires, providing investigators with valuable detailed case-specific data. The CPSP even received queries about whether similar counterpart surveillance systems exist in other areas of health care.
2000 – Five years into the program
The CPSP’s fifth anniversary provided an excellent opportunity to reflect on both its accomplishments to-date and on its challenges for the future. For the first time since the CPSP’s inception, results from concluded studies on hemorrhagic disease of the newborn and subacute sclerosing panencephalitis (SSPE) emphasized the importance of undertaking timely epidemiological data collection to affect, and confirm, public health policy decisions. For example, with only five confirmed cases in four years, hemorrhagic disease of the newborn study results supported the Canadian Paediatric Society’s guidelines on the administration of intramuscular vitamin K to newborn babies. Two studies, neonatal herpes simples virus infection and SSPE, clearly indicated respectively how data collection can monitor the incidence of disease before vaccination, as well as the effectiveness of immunization programs. In fact, the rarity of SSPE cases (two in four years) is both a tribute to the success of the measles immunization program, as well as reassurance about the safety of the measles vaccine.
With increased concerns about the protection of individual privacy, an important issue for paediatric surveillance became the need to balance the goal of data collection for the common good against the need for confidentiality. While health-related surveillance existed for centuries, the rapidly increasing technological ability to link, analyze and spread data had to be considered. At their fall meeting, CPSP Steering Committee members, attending a workshop on ethics, reaffirmed their commitment to maintaining patient confidentiality; only non-nominal patient information, such as the date of birth and sex of the patient are requested, to identify and eliminate duplicates. The CPSP renewed its commitment to ensuring the privacy and the non-labelling of individuals, localities, and provinces in either rare encounters of a condition or localized outbreaks, citing that only pan-Canadian national data is used in presentations and publications.
2001 – Publications and promotions
Through a series of publications and presentations, the CPSP demonstrated its commitment to sharing information that would showcase the value of active surveillance and keep participants engaged in the program; examples are:
- Monthly highlights published in Paediatrics & Child Health
- Monthly articles published in CPS News
- May/June issue of Paediatrics & Child Health dedicated to surveillance. Issue included: editorial, advocacy paper, paediatric infectious disease note, neonatology note, two original articles focusing on the international perspective and beyond collecting numbers and a commentary on genetics and the CPSP.
- Two abstracts presented at the CPS 2001 Annual Meeting in Vancouver
- Dr. Victor Marchessault represented INoPSU at the International Paediatrics Association’s (IPA) meeting in Beijing where his proposal that INoPSU join the IPA was approved.
- The Canadian Paediatric Surveillance Program (CPSP): An epidemiological tool in action, promoting the CPSP, presented at the IPA meeting in Beijing.
- Hemorrhagic disease of the newborn - an internatinal comparison of prophylactic regimens of vitamin K, Congenital rubella syndrome - an international perspective: are the results comparable?, and The Canadian and British perspectives of paediatric intellectual and neurological deterioration (PIND): Are the results comparable? submitted for presentation at the CPS 2002 meeting in Toronto.
- Congenital rubella syndrome - an international perspective: are the results comparable? submitted for presentation at the Royal College of Paediatrics & Child Health meeting in York, England, 2002.
- Concurrent session on the public health and clinical implications of the CPSP approved for the CPS 2002 meeting in Toronto.
2002 – CPSP expands
The CPSP was expanded to allow investigators a cost-effective tool to survey participants on a one-time basis in order to identify the prevalence of a problem or to answer a specific question. In 2002, the Injury and Child Maltreatment Section, Health Surveillance and Epidemiology Division of the Centre for Healthy Human Development at Health Canada, with the cooperation and support of the Product Safety Bureau, Healthy Environments and Consumer Safety Branch, posed a question to better understand the frequency and extent of injuries associated with baby walkers in Canada. A total of 1,214 paediatricians returned the survey, representing a 53.4% response rate. The next scheduled question, in early 2003, will verify for investigators whether children with lap-belt syndrome are seen by paediatricians at some point during their hospitalization.
Through national and international presentations, including those at the second INoPSU meeting, the CPSP continued to showcase its work. See presentations for a complete list.
2003 - Program evaluation
A formal program evaluation was undertaken to answer important questions concerning the program’s strengths, weaknesses and future course of action. An extensive review by an international Expert Advisory Group (EAG) of the program’s policies, objectives, targets, strategies, action plans, performance and output resulted in a unanimous opinion that the CPSP represents excellent value for money. The achievement in this respect was seen as excellent and unsurpassed by any comparable program known to the EAG. The CPSP was seen as representing an important collaborative tool for surveillance, research and policy development. In this role, it was perceived as unique in Canada. In other words, it provides an important activity that would disappear in its absence, unless a much larger investment is made to replace it.
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