Horizontal bars show each component of ambulatory health care cost (1a) and out-of-pocket cost (1b) for adults with congenital heart disease (CHD), by lesion category; the overall length of each bar indicates the total cost. Change in cost indicates the adjusted difference in overall cost compared with that for frequency-matched non-CHD patients. All costs and cost differences are adjusted for age, sex, US region, beneficiary status, and cardiac and noncardiac comorbidities. ED indicates emergency department. Lesions included within each CHD type are complex (Eisenmenger syndrome, common ventricle, hypoplastic left heart syndrome, transposition of great arteries, tetralogy of Fallot, truncus arteriosus, and endocardial cushion defect), simple (ventricular septal defect and patent ductus arteriosus), and moderately complex (Ebstein anomaly, coarctation of aorta, anomalies of the pulmonary artery, anomalies of the pulmonary valve, anomalies of the tricuspid valve, unspecified septal defects, anomalies of the great vein, subaortic stenosis, and aortic anomalies).
eTable. International Classification of Disease-10 Codes for Congenital Heart Disease (CHD) Lesions
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Agarwal A, Vittinghoff E, Myers JJ, et al. Ambulatory Health Care Service Use and Costs Among Commercially Insured US Adults With Congenital Heart Disease. JAMA Netw Open. 2020;3(9):e2018752. doi:10.1001/jamanetworkopen.2020.18752
Adults with congenital heart disease (CHD) are a rapidly increasing population,1 have many comorbidities,2 and require frequent monitoring.3 However, little is known about their ambulatory health care use and associated costs in the US.
International Statistical Classification of Diseases and Related Health Problems, 10th Revision (ICD-10) codes were used to identify patients with CHD, as described previously (eTable in the Supplement).2 The control group was selected from a random sample of age- and sex-matched individuals without CHD and with at least 1 year of data equivalent to that of the patients with CHD. Comorbidities were identified with Elixhauser comorbidity measures.3 The American Heart Association/American College of Cardiology anatomic classification was used to categorize adults with CHD as having simple, moderately complex, and complex disease.4
Wilcoxon and χ2 tests were used for comparisons of continuous and categoric variables, respectively. To estimate the independent associations of age, sex, US region, beneficiary status, comorbidities, and lesion type with costs, we used zero-inflated negative binomial models. These models accommodate the severe right skewing of costs, as well as the excess of observations with no costs, relative to the standard negative binomial distribution. Adjusted mean costs by lesion group were obtained by regression standardization, based on the fitted negative binomial models. Two-tailed P < .05 was considered statistically significant. Analyses were performed with Stata version 16.0. Data were analyzed on January 28, 2020.
The mean (SD) age of 33 892 patients included in the study cohort was 35.2 (14.2) years, and 48.8% were women. Of 16 946 patients, 5168 (30.5%) had complex CHD, 5719 (33.8%) had moderately complex CHD, and 6059 (35.7%) had simple CHD.
Compared with individuals without CHD, those with CHD had more comorbidities, more health care visits, and higher expenditures (Table). After multivariate adjustments, ambulatory costs remained significantly higher for all types of adults with CHD than for those without it (Figure).
Among patients with CHD, after multivariate adjustments, factors independently associated with ambulatory costs were 10-year increase in age (cost ratio, 1.17; 95% CI, 1.13-1.21), female sex (cost ratio, 1.14; 95% CI, 1.05-1.23), primary beneficiary (cost ratio, 0.88; 95% CI, 0.81-0.96), complex CHD (cost ratio, 1.43; 95% CI, 1.29-1.59), cardiac comorbidities (cost ratio, 2.17; 95% CI, 1.90-2.46), and noncardiac comorbidities (cost ratio, 1.92; 95% CI, 1.75-2.10) (P < .005 for all).
Annual ambulatory health care use and costs were significantly higher for commercially insured adults with CHD than those without it, even after adjusting for their baseline characteristics and comorbidities. Among adults with CHD, complex CHD and presence of comorbidities were independently associated with the highest cost ratio magnitude. This demonstrates the extraordinary health care needs of these patients with complex disease, who usually have multisystem disease,2 and underscores the importance of developing structured work flows to appropriately allocate resources. Our novel CHD severity–specific health care cost estimates may help patients in their personal financial planning (selecting a health insurance plan that will minimize their financial risk, such as opting for employee-provided health savings plans) and policy makers in designing affordable and appropriate health plans.
Our study limitations include reliance on ICD-10 codes and limited generalizability to patients who are not commercially insured. In contrast to previous studies of adults with CHD that primarily reported charges,5,6 our estimates are directly reflective of actual costs and therefore pertinent to understanding health resources required for these patients.
In conclusion, we provide data that could be useful to educate clinicians, health care organizations, and patients to guide resource allocation, enhance more efficient work flows, and inform realistic financial expectations.
Accepted for Publication: July 20, 2020.
Published: September 24, 2020. doi:10.1001/jamanetworkopen.2020.18752
Open Access: This is an open access article distributed under the terms of the CC-BY License. © 2020 Agarwal A et al. JAMA Network Open.
Corresponding Author: Anushree Agarwal, MD, Adult Congenital Heart Disease Section, Division of Cardiology, University of California, San Francisco, 500 Parnassus Ave, M-1177B, PO Box 0124, San Francisco, CA 94143-0124 (firstname.lastname@example.org).
Author Contributions: Drs Agarwal and Vittinghoff had full access to all of the data in the study and take responsibility for the integrity of the data and the accuracy of the data analysis.
Concept and design: Agarwal, John, Marcus.
Acquisition, analysis, or interpretation of data: Agarwal, Vittinghoff, Myers, Dudley, Khan, Marcus.
Drafting of the manuscript: Agarwal, Marcus.
Critical revision of the manuscript for important intellectual content: Agarwal, Vittinghoff, Myers, Dudley, Khan, John.
Statistical analysis: Agarwal, Vittinghoff.
Obtained funding: Agarwal, Marcus.
Administrative, technical, or material support: Agarwal, Myers, Marcus.
Supervision: Agarwal, Myers, Dudley, Marcus.
Conflict of Interest Disclosures: Dr Agarwal reports receiving grants from an American Heart Association/CHF AWRP Mentored Clinical & Population Research Award during the conduct of the study. Dr Vittinghoff reports receiving other from the University of California, San Francisco during the conduct of the study. Dr Marcus reports receiving grants from Jawbone Health outside the submitted work. No other disclosures were reported.
Funding/Support: This work was supported in part from an AHA/CHF AWRP Mentored Clinical & Population Research Award (17MCPRP33240000) (Drs Agarwal and Dudley).
Role of the Funder/Sponsor: The funder had no role in the design and conduct of the study; collection, management, analysis, and interpretation of the data; preparation, review, or approval of the manuscript; and decision to submit the manuscript for publication.
Additional Contributions: Robert Thombley, BS, provided access to the study cohort from the IBM MarketScan database. He was compensated for his work.
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