Financial costs
Arthritis and related conditions, such as juvenile arthritis, cost the U.S. economy nearly $128 billion per year in medical care and indirect expenses, including lost wages and productivity [
34]. Great financial burden is imposed on families of patients with JIA and may be attributed to the following: expensive biologic agents; frequent subspecialty outpatient visits; periodic laboratory and imaging tests; frequent visits to the pediatrician for increased URI, etc. due to immunosuppression; physical therapy; inpatient infusions and hospitalizations; and missed work days for the parents. The process of diagnosis and treatment can be very expensive [
35]. Minden et al measured direct costs (healthcare and non-healthcare costs) and indirect costs (productivity loss due to sick leave and work disability) in 215 JIA patients in adulthood [
36]. These patients were assessed on an average of 17 years after disease onset using clinical evaluation, a structured interview, and two self completion questionnaires [
36]. The authors assessed annual direct costs based on the reported use of healthcare services and resources, using average unit prices. Indirect costs were determined from the number of work days missed-that is, using the human capital approach. The mean total cost of late JIA was estimated to be 3500 Euros per patient and year, of which the direct cost contributed a large percentage. Ninety percent of the cost was attributable to patients with still active disease (55%). These patients incurred a mean total cost of 5700 Euros per patient year, and among them those under rheumatologic care incurred a cost of 9300 Euros. Independent contributors to increased costing patients with active JIA included the following variables: belonging to a certain JIA subgroup, functional disability, and receipt of specialized care. Highest mean total costs were found in patients with active seropositive polyarthritis (17,000 Euros) and extended oligoarthritis (11,000 Euros), while the lowest were found in active enthesitis related arthritis (1,500 Euros) and persistent oligoarthritis (2,700 Euros) [
36]. Authors concluded that the estimated one-year costs in are considerable and variable among the various JIA subgroups [
36].
Allaire et al collected data on direct costs, family costs and community (extra school) costs on 120 families with children with JRA in New England, and published the results in 1992 [
3]. The mean yearly direct cost per child was $7,905 (inpatient: $1,717; outpatient: $5,700; and nonmedical: $488). Family costs averaged $1,524/year (out of pocket medical and nonmedical: $1,196; lost salary: $328), which corresponded to 5% of mean family income. The mean extra school cost was $1,449/9 months. This study shows that the economic impacts of JRA is significant [
3].
Given the fluctuating nature of the disease, the need for treatment escalation, physical therapy, screening for ophthalmologic complications and surgery, cumulative costs need to be determined. Especially with the advent of biologic therapies, treatment strategies in JIA should be analyzed for their long-term cost effectiveness [
36].
Ambulatory health care visits, hospitalizations, and mortality
The National Ambulatory Medical Care Survey and National Hospital Ambulatory Medical Care Survey found that among children < 17 years in 1997-98, there was an average of 1.3 million AORC-related ambulatory care visits per year related to arthritis and other rheumatic conditions (AORC) [
37]. Hootman et al reported that pediatric arthritis-related visits were more likely to be made by girls (67%), whites (82%), non-Hispanics (66%) and children were aged 12-17 years (59%). Most visits occurred in physician offices (75%) compared to outpatient departments (18%) and emergency department (7%). The main three diseases seen were: soft tissue disorders excluding back (41%; 513,000), unspecified joint pain/effusion (31%; 387,000), and rheumatoid arthritis (10%; 122,000). Among physician office visits, 41% saw family practice/general practitioners/internal medicine; 33% saw rheumatologists/orthopedists/neurologists; and 26% saw pediatricians [
37].
Sacks et al estimated the prevalence of and the annual number of ambulatory health care visits (physician, outpatient, emergency) for children with significant pediatric arthritis and other rheumatologic conditions (SPARC) using data from the 2001-2004 National Ambulatory Medical Care Survey and 2001-2004 National Hospital Ambulatory Medical Care Survey [
2]. Visit estimates were converted into prevalence estimates using data on the number of prior annual visits per patient. Synthetic estimates for states were extrapolated using national rates. The average annualized number of children with SPARC was 294,000 (95% confidence interval [95% CI] 188,000-400,000); and ambulatory health-care visits were 827,000 (95% CI 609,000-1,044,000) [
2]. Study data also show that children diagnosed with arthritis and other rheumatologic conditions had an average of 83,000 emergency department room visits annually [
2]. With codes for AORC as defined in adults, in 1997, among children < 14 years, there were 21,000 hospitalizations with chief diagnosis of AORC (rate = 3.5/10,000) and 33,000 hospitalizations with any reference to AORC (rate = 9.2/10,000) out of 2,266,000 childhood hospitalizations (0.9%-1.45% of all hospitalizations)[
38]. Undoubtedly, the number of arthritis-related health care visits is significant and impacts the families' HRQOL and burdens the pediatric health care system. Detailed tracking of such data for different subtypes of JIA is important over the long-term [
2].
Sacks et al reviewed multiple cause of death tapes from the National Center for Health Statistics (NCHS) from 1979 to 1998. The authors found that the crude death rate from AORC was 2.46 per 100,000 in 1979, and by 1998, it was 3.48. The three categories of AORC that accounted for almost 80% of deaths were diffuse connective tissue diseases (34%), other specified rheumatic conditions (23%), and rheumatoid arthritis (22%) [
39].
Using AORC codes, the Centers for Disease Control and Prevention's (CDC) NCHS death data revealed that about 1,000 children younger than 15 years of age died from arthritis and other rheumatic conditions in the 20 years from 1979-1998 (average = 50 deaths/year). The juvenile AORC death rate fell 25% during the 20-year period from 1.2 per million to 0.9 per million (average = 1 death per million children per year) [
39] (study quoted in the CDC website-[
40]).
Hashkes et al described mortality rates, causes of death, and potential mortality risk factors in pediatric rheumatic diseases in the US using the Indianapolis Pediatric Rheumatology Disease Registry, which included 49,023 patients from 62 centers who were newly diagnosed between 1992 and 2001[
41]. Authors confirmed 110 deaths among 48,885 patients (0.23%). Patients had been followed up for a mean ± SD of 7.9 ± 2.7 years. The standardized mortality ratios (SMR) of the entire cohort was significantly decreased (0.65 [95% CI 0.53-0.78]), with differences in patients followed up for > or = 9 years. The SMR was significantly greater for systemic lupus erythematosus (3.06 [95% CI 1.78-4.90]) and dermatomyositis (2.64 [95% CI 0.86-6.17]) but not for systemic juvenile rheumatoid arthritis (1.8 [95% CI 0.66-3.92]). Causes of death were related to the rheumatic diagnosis (including complications) in 39 patients (35%), treatment complications in 11 (10%), non-natural causes in 25 (23%), background disease in 23 (21%), and were unknown in 12 patients (11%). Rheumatic diagnoses, age at diagnosis, sex, and early use of systemic steroids and methotrexate were significantly associated with the risk of death. Their findings showed that the overall mortality rate for pediatric rheumatic diseases was not increased. Even for the diseases and conditions associated with increased mortality, mortality rates were significantly lower than those reported in previous studies [
41]. Current data are required for patients on biologic treatments. Further, in order to elucidate the impact on cost, hospitalizations, ambulatory care and emergency room visits codes specific for childhood arthritis should be used and specified for particular disease and subtype.