Assessing Health Care Utilization and Compliance in Kawasaki Disease.

OBJECTIVE: To characterize health care utilization and costs associated with care after diagnosis of Kawasaki disease including adherence to guidelines for echocardiograms. STUDY DESIGN: We analyzed children hospitalized for Kawasaki disease using 2015-2017 national Truven MarketScan commercial claims data. The mean 90-day prehospitalization utilization and costs were quantified and compared with the 90 days posthospitalization via Wilcoxon 2-sample test. Adherence to echocardiogram guidelines was examined using multivariable logistic regression to identify factors associated with adherence. RESULTS: The mean total payments 90 days prior to hospitalization ($2090; n = 360) were significantly lower than those after discharge ($3778), though out of pocket costs were higher ($400 vs $270) (P < .0001). There was an increase in office visits, medical procedures, and echocardiograms after discharge. A majority of health care utilization before hospitalization occurred in the 7 days immediately prior to the date of admission; 51% obtained an echocardiogram within the first 2 weeks, and 14% were completely adherent with recommendations. Children with greater utilization prior to admission were more likely to adhere to American Heart Association guidelines for follow-up echocardiograms (OR 1.03, 95% CI 1.01-1.06). CONCLUSIONS: Outpatient health care expenditure nearly doubles after Kawasaki disease hospital discharge when compared with prehospitalization, suggesting the financial ramifications of this diagnosis persist beyond costs incurred during hospitalization. A significant portion of patients do not receive guideline recommended follow-up echocardiograms. This issue should be explored in more detail given the morbidity and mortality associated with this diagnosis.

A Cost Comparison of Infliximab Versus Intravenous Immunoglobulin for Refractory Kawasaki Disease Treatment.

BACKGROUND AND OBJECTIVES: In 10% to 20% of cases, Kawasaki disease is refractory to intravenous immunoglobulin (IVIg), an expensive medication under a national shortage. Data suggest that infliximab is a viable alternative to a second dose of IVIg, with similar efficacy and safety. We compared the cost of a second IVIg dose to that of infliximab in the treatment of refractory Kawasaki disease (rKD). METHODS: A decision analysis model was used to compare rKD treatments: a second dose of IVIg at 2 g/kg versus infliximab at 10 mg/kg. Infliximab monitoring times were 24, 36, and 48 hours. Direct hospital costs beginning at rKD diagnosis were estimated by using 2016-2017 Truven MarketScan data. Redbook was used for drug costs. Calculations were applied to 3 hypothetical cohorts of 100 patients aged 2 (12.5 kg), 4 (16 kg), and 8 years (25.5 kg). Indirect costs included parental missed workdays. RESULTS: The total direct cost for children receiving IVIg was $1 677 801, $1 791 652, and $2 100 675 for the 2-, 4-, and 8-year-old cohorts. The direct cost of infliximab with 24 hours of monitoring was $853 042, $899 096, and $1 024 101, respectively. A 20% bidirectional sensitivity analysis revealed stability of our model, with overall cost savings with use of infliximab. With monitoring 48 hours after infliximab treatment, 20% changes in length of stay (LOS) tipped the balance for the 2- and 4-year-old cohorts. Overall, IVIg and infliximab LOS had the most influence on our model. CONCLUSIONS: Infliximab has potential to yield shorter LOS and significant cost savings in the treatment of rKD. Infliximab treatment, followed by 24 hours of monitoring, nearly halved hospital costs, regardless of age.

An update on the epidemiology, length of stay, and cost of Kawasaki disease hospitalisation in the United States.

BACKGROUND: Kawasaki disease is an acute vasculitis of childhood and is the leading cause of acquired heart disease in the developed countries. METHODS: Data from hospital discharge records were obtained from the National Kids Inpatient Database for years 2009 and 2012. Hospitalisations by months, hospital regions, timing of admission, insurance types, and ethnicity were analysed. Length of stay and total charges were also analysed. RESULTS: There were 10,486 cases of Kawasaki disease from 12,678,005 children hospitalisation. Kawasaki disease was more common between 0 and 5 years old, in male, and in Asian. The January-March quarter had the highest rate compared to the lowest in the July-September quarter (OR=1.62, p < 0.001). Admissions on the weekend had longer length of stay [4.1 days (95 % CI: 3.97-4.31)] as compared to admissions on a weekday [3.72 days (95 % CI: 3.64-3.80), p < 0.001]. Blacks had the longest length of stay and whites had the shortest [4.33 days (95 % CI: 4.12-4.54 days) versus 3.60 days (95 % CI: 3.48-3.72 days), p < 0.001]. Coronary artery aneurysm was identified in 2.7 % of all patients with Kawasaki disease. Children with coronary artery aneurysm were hospitalised longer and had higher hospital charge. Age, admission during weekend, and the presence of coronary artery aneurysm had significant effect on the length of stay. CONCLUSIONS: This report provides the most updated epidemiological information on Kawasaki disease hospitalisation. Age, admissions during weekend, and the presence of coronary artery aneurysm are significant contributors to the length of stay.

Prediction of responsiveness or non-responsiveness to treatment of acute Kawasaki disease using 1 gram per kilogram of immunoglobulin--an effective and cost-saving schedule of therapy.

Standard treatment of acute Kawasaki disease involves giving 2 grams per kilogram of immunoglobulin intravenously along with aspirin. More than half of the patients with acute Kawasaki disease, nonetheless, can be cured by giving only 1 gram per kilogram of immunoglobulin, thus reducing this aspect of the cost of treatment by half. Our purpose was to predict those patients with acute Kawasaki disease who would respond to treatment with 1 gram per kilogram of immunoglobulin given intravenously on the basis of their clinical profiles and laboratory findings prior to the initial treatment. We performed a retrospective review of the clinical records of consecutive patients with acute Kawasaki disease treated in our hospital with intravenous immunoglobulin from January, 2001, to December, 2005.During this period, we treated in this fashion 98 patients with acute Kawasaki disease. 65% of these needing immunoglobulin therapy were cured by giving 1 gram per kilogram. The neutrophil count and the percentage of white blood cells representing neutrophils, along with aspirate aminotransferase, alanine aminotransferase, bilirubin and C reactive protein, were all significantly lower, and sodium was significantly higher, in those responding to 1 gram per kilogram of immunoglobulin when compared to those who did not respond. The days of illness at the first intravenous treatment was later in those responding than in those failing to respond. We generated a score for prediction, assigning a point for each of C reactive protein equal to or greater than 10 mg/dl, sodium equal to or lower than 133 meq/l, alanine aminotransferase equal to or greater than 110 IU/l, and 2 points for the percentage of white blood cells representing neutrophils equal to or greater than 70%. Using a cut-off point of a score less than 2, we were able to identify those responding with 60% sensitivity, and 91% specificity.Thus, we are now able to predict those patients with acute Kawasaki disease who will respond to immunoglobulin given intravenously at 1 gram per kilogram using laboratory data, with a potential saving in medical costs.

The strategy of immune globulin resistant Kawasaki disease: a comparative study of additional immune globulin and steroid pulse therapy.

BACKGROUND: We compared the clinical utility of additional intravenous immune globulin (IVIG) therapy with the clinical utility of steroid pulse therapy in patients with IVIG-resistant Kawasaki disease. METHODS: We enrolled 164 patients with Kawasaki disease who were treated with a single dose of IVIG (2 g/kg) and aspirin (30 mg/kg per day). Twenty-seven of these patients (16%) were resistant to the initial IVIG treatment. We compared the effectiveness of treatment strategies for the initial IVIG-resistant 27 patients, 14 of these patients were treated with additional IVIG therapy, and the other 13 patients were treated with steroid pulse therapy (methylprednisolone 30 mg/kg per day for 3 days). RESULTS: Three patients in the group receiving additional IVIG treatment had coronary artery aneurysms (21.4%), no patients had coronary artery aneurysm in the steroid pulse therapy group; the difference in the incidence of coronary artery aneurysm was not statistically significant. The duration of high fever after additional treatment in the steroid pulse therapy group (1 ± 1.3 days) was significantly shorter than that in the additional IVIG treatment group (3 ± 2.4 days; P < 0.05). The medical costs were significantly lower in the steroid pulse therapy group than in the additional IVIG treatment group. CONCLUSION: Steroid pulse therapy was useful to reduce the fever duration and medical costs for patients with Kawasaki disease. Steroid pulse therapy and additional IVIG treatment were not significantly different in terms of preventing the development of coronary artery aneurysm.

Epidemiologic features of Kawasaki disease in Taiwan, 1996-2002.

OBJECTIVE: Kawasaki disease (KD) is the most common acquired heart disease in children worldwide. The incidence of KD varies among different countries, with Asian countries supposedly having higher incidences than Western countries. However, the incidence of KD in Taiwan has not been well investigated. METHODS: Since the implementation of Taiwan's National Health Insurance (NHI) in 1995, NHI has covered health care for >96% its population. Using the NHI database from 1996 to 2002, we investigated epidemiologic features of KD (International Classification of Diseases, Ninth Revision, code 446.1), the rate of coronary artery aneurysm formation (International Classification of Diseases, Ninth Revision, code 414.11), and the change in incidence during the recent 7 years. We also compared the annual incidences in Taiwan with those reported by other countries. RESULTS: During the 7-year study period, KD occurred most frequently in the summer and least frequently in winter. It is interesting that the highest peak occurred in the summer of 1998 at the same time that Taiwan's enterovirus 71 epidemic was occurring. Ninety-one percent of KD cases occurred in children who were <5 years old, and the male-to-female ratio was 1.70:1. Recurrence of KD was found in 1.3% (94 of 7305) of these children, and coronary artery aneurysm was found in 7.3% (536 of 7305). The annual incidence per 100,000 children was 146 in children <1 year old, 98 in 1-year-old children, 51 in 2-year-old children, 28 in 3-year-old children, 19 in 4-year-old children, and 5.3 in 5- to 9-year-old children; the incidence of KD decreased with increased age. The overall incidence was 66 cases per 100,000 children <5 years old from 1996 to 2002 with the annual incidence not differing significantly during the 7-year study period. CONCLUSIONS: KD in Taiwan occurs more frequently in boys and in the summer months. During the 7-year study period, the annual KD incidence in Taiwan of 66/100000 in children <5 years old was the second highest in the world after Japan.

Kawasaki syndrome hospitalizations in the United States, 1997 and 2000.

OBJECTIVE: To estimate the incidence and describe the epidemiologic characteristics of Kawasaki syndrome (KS) among children in the United States. METHODS: Hospital discharge records with a KS diagnosis among patients <18 years of age were obtained from the 1997 and 2000 Kids' Inpatient Database and weighted to estimate the number and rate of KS-associated hospitalizations for the United States. RESULTS: In 2000, approximately 4248 hospitalizations associated with KS occurred in the United States, and the median age of patients at admission was 2 years. Children <5 years of age accounted for 3277 of these KS hospitalizations (77%) and had a KS hospitalization rate of 17.1 per 100,000 children. This rate was similar to the 1997 rate of 17.6 per 100,000 children. The KS hospitalization rate was significantly higher for infants <1 year of age than for children 1 to 4 years of age (19.8 and 16.4 per 100,000 children, respectively). The rate of KS hospitalizations among children aged <5 years was highest among Asian and Pacific Islander children and was followed by the rate for black children (39.0 and 19.7 per 100,000 children, respectively). No deaths associated with KS were reported among hospitalized children. The median charge for a KS hospitalization was 7779 dollars (mean 10,725 dollars) and the total annual charges for KS hospitalizations in 2000 were approximately 35 million dollars among children <5 years of age. CONCLUSIONS: Among children <5 years of age, the annual KS-associated hospitalization rates were similar for 1997 and 2000. The epidemiologic characteristics and hospitalization rates for KS at a national level were consistent with those reported from earlier studies, suggesting that the incidence for KS has not markedly changed in the United States during the past decade.

A cost-benefit analysis of intravenous immunoglobulin treatment in children with Kawasaki disease.

To determine the long-term cost-benefit of intravenous immunoglobulin (IVIG) treatment in Children with Kawasaki Disease (KD), a model was made to compare the total cost for management of these children with and without the use of IVIG. Long-term (10-21 years) follow-up of 594 KD patients treated in the pre-IVIG era reported by Kato, et al. was used to calculate cost using previous cost studies from Chulalongkorn Hospital. Reduction of CAA from 25 per cent to 4 per cent with IVIG treatment was assumed based on previous published data. Total cost was slightly lower for the non-IVIG treatment group compared to the IVIG treatment group (33,451,129 baht vs 35,001,195 baht) for the duration of follow-up in Kato's model. Cost per effectiveness analysis showed more effectiveness in the IVIG treatment group (359,576 baht vs 383,614 baht). Net cost analysis similarly demonstrated lower costs in the IVIG treatment group (25,365,215 baht vs 33,451,129 baht). Incremental cost-effectiveness analysis demonstrated supplementary costs of 13,663 baht for one case in the reduction of coronary involvement and 387,517 baht for one life saved in the IVIG-treated group. Estimation of total costs for follow-up and treatment for healthy life (until 60 years old) was more expensive in the non-IVIG treatment than the IVIG treated group (75,482,803 baht vs 29,883,833 baht). The authors conclude that treatment of all KD cases in Thailand with IVIG is likely to result in lower cost and better outcome when compared to no treatment with the IVIG policy.

Kawasaki syndrome hospitalizations and associated costs in the United States.

OBJECTIVES: To describe the epidemiologic characteristics of patients hospitalized with Kawasaki syndrome (KS) and estimate associated costs in the United States, using a large national hospital discharge dataset. METHODS: Hospitalization discharge records with KS for 1997 through 1999 for U.S. residents <18 years of age were selected from Solucient's hospital discharge records. These records are collected from most of the self-governing children's hospitals and approximately one-third of short-term, non-federal general hospitals in the United States. RESULTS: A total of 7,431 hospital discharges with a KS diagnosis were identified; 2,270 of the discharges were in 1997, 2,700 in 1998, and 2,461 in 1999. Boys comprised 60.0% of the discharges, and 76.4% of discharges were among children ages <5 years. For the 44 states and the District of Columbia with at least one hospital reporting KS, the average annual KS hospitalization rate was 10.2 per 100,000 children ages <5 years. The KS hospitalization rate for boys (12.0 per 100,000) was higher than that for girls (8.3 per 100,000) (risk ratio 1.45; 95% confidence interval 1.37, 1.52). Extrapolation to the U.S. population showed an estimated average annual KS hospitalization rate of 21.6. The median KS hospitalization cost for children <5 years of age during the study period was $6,169 US dollars. CONCLUSIONS: The KS hospitalization rate was consistent with that of previous U.S. studies, although the extrapolated rate may be an overestimation. The median hospitalization cost for KS was higher than that for respiratory syncytial virus-associated bronchiolitis and diarrheal diseases. Large hospitalization datasets can be used to monitor the occurrence of KS in the United States.

Cost-effective use of echocardiography in children with Kawasaki disease.

OBJECTIVE: To determine whether a third echocardiogram, performed 6 months to 1 year after the onset of Kawasaki disease (KD), as recommended by current American Heart Association guidelines, identified any case of coronary artery abnormalities when previous echocardiograms were normal. METHODS: Children diagnosed with KD were identified by searching our institution's database. Cases were included in the study if diagnosed between June 1988 and December 1996 and if at least two echocardiograms were documented, including at least one study between 2 weeks and 2 months from the onset of KD and another in follow-up. The patients' charts were reviewed and videotapes of the echocardiograms were reviewed if reports were unclear or contradictory. McNemar's test for discordant pairs was used for statistical analysis. Additionally, a complete review was performed in all other cases of KD in the database in which a coronary artery abnormality had been identified. RESULTS: There were 203 patients diagnosed during the study period who had 2 or more echocardiograms performed, and 67 had the requisite studies in the subacute period and later follow-up. The median age at onset of KD was 3.0 years (range: 0.2-16), the median duration of follow-up was 12.5 months (range: 1.7-100), and the median number of echocardiograms performed was 3 (range: 2-8). Intravenous immunoglobulin was given in 62 cases, and high-dose aspirin was given in 63. There were 35 children with no echocardiographic abnormalities at any point, and 15 other children had early abnormalities (including coronary ectasia, perivascular brightness, pericardial effusion, and ventricular dysfunction) but had a normal echocardiogram between 2 weeks and 2 months. Of these 50 children, none were noted to have abnormalities on later studies. Three children had effusion and/or perivascular brightness after 2 weeks; follow-up studies were normal in each. Six children had coronary ectasia after 2 weeks; it persisted on follow-up in 1 child and had resolved in 5 children. Eight children had coronary aneurysms on studies after 2 weeks; in 3 children, the aneurysm resolved on later follow-up. No coronary abnormalities were demonstrated on a late follow-up echocardiogram in any child with normal coronaries between 2 weeks and 2 months. CONCLUSIONS: All children with KD should have an echocardiogram at the time of diagnosis with a follow-up study 4 to 6 weeks after the onset of fever. In the current environment of cost-containment, additional echocardiographic studies are justified only if abnormalities are present at 4 to 6 weeks. Kawasaki disease, coronary artery aneurysm, echocardiography, coronary ectasia.

Economic evaluation of intravenous immune globulin therapy for Kawasaki syndrome.

OBJECTIVE: To determine the costs and clinical outcomes of three alternative treatments of the acute phase of Kawasaki syndrome: aspirin alone; low doses of intravenously administered immune globulin (IVIG-LD), 400 mg/kg per dose for 4 days; and high doses of intravenously administered immune globulin (IVIG-HD), 2.0 gm/kg for one dose. DESIGN: A model was developed that assumed the inclusion of 100 patients with acute Kawasaki syndrome in each treatment option. Costs were valued by using the Chedoke-McMaster Corporate Cost Model in 1992 Canadian dollars. Clinical outcome, based on the published literature, was measured by the prevalence of coronary artery dilation at 7 weeks from the diagnosis of Kawasaki syndrome. RESULTS: For every 100 patients with Kawasaki syndrome, the cost was reduced by $323,400 when aspirin therapy alone was changed to IVIG-HD therapy and 14 cases of coronary artery dilation were thereby prevented. When IVIG-HD therapy was compared with IVIG-LD therapy, the cost was reduced by $118,200 because two cases of coronary artery aneurysm were prevented. This latter result was sensitive to the duration of hospitalization, with IVIG-HD costing $8500 more for every 100 patients than IVIG-LD when it was assumed that both groups were hospitalized for 5 days, an unlikely occurrence. CONCLUSIONS: Treatment with IVIG-HD for Kawasaki syndrome is preferred because it results in both lower costs and lower rates of coronary artery dilation.