Cumulative In-Hospital Costs Associated With Single-Ventricle Palliation

Michael L O'Byrne^{1

2

3}, Kimberly E McHugh⁴, Jing Huang^{1

5}, Lihai Song⁶, Heather Griffis⁶, Brett R Anderson⁷, Emily M Bucholz⁸, Nikhil K Chanani⁹, Justin J Elhoff¹⁰, Stephanie S Handler¹¹, Jeffery P Jacobs¹², Jennifer S Li¹³, Alan B Lewis¹⁴, Brian W McCrindle¹⁵, Nelangi M Pinto¹⁶, Peter Sassalos¹⁷, David S Spar¹⁸, Sara K Pasquali¹⁹, Andrew C Glatz^{1

2}

Affiliations

¹ Division of Cardiology, The Children's Hospital of Philadelphia and Department of Pediatrics, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, Pennsylvania, USA.
² Center for Pediatric Clinical Effectiveness, The Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, USA.
³ Leonard Davis Institute of Health Economics and Cardiovascular Outcomes, Quality, and Evaluative Research Center, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, Pennsylvania, USA.
⁴ Division of Cardiology, Medical University of South Carolina, Charleston, South Carolina, USA.
⁵ Department of Biostatistics, Center for Clinical Epidemiology and Biostatistics, Perelman School of Medicine, Philadelphia, Pennsylvania, USA.
⁶ Data Science and Biostatistics Unit, Department of Biomedical and Health Informatics, The Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, USA.
⁷ Division of Cardiology, New York-Presbyterian Morgan-Stanley Children's Hospital/Columbia University Irving Medical Center, New York, New York, USA.
⁸ Department of Cardiology, Children's Hospital Boston and Harvard University Medical School, Boston, Massachusetts, USA.
⁹ Children's Healthcare of Atlanta, Sibley Heart Center and Emory University School of Medicine, Atlanta, Georgia, USA.
¹⁰ Sections of Critical Care and Cardiology, Texas Children's Hospital and Baylor College of Medicine, Houston, Texas, USA.
¹¹ Children's Hospital of Wisconsin, Milwaukee, Wisconsin, USA.
¹² Division of Cardiothoracic Surgery, Departments of Surgery and Pediatrics, Congenital Heart Center, University of Florida, Gainesville, Florida, USA.
¹³ Division of Pediatric Cardiology, Duke University School of Medicine, Durham, North Carolina, USA.
¹⁴ Division of Cardiology, Department of Pediatrics, Children's Hospital of Los Angeles, Keck School of Medicine of University of Southern California, Los Angeles, California, USA.
¹⁵ Department of Pediatrics, Labatt Family Heart Centre, The Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada.
¹⁶ Division of Cardiology, Primary Children's Hospital and University of Utah School of Medicine, Salt Lake City, Utah, USA.
¹⁷ Division of Pediatric Cardiothoracic Surgery, C.S. Mott Children's Hospital and University of Michigan School of Medicine, Ann Arbor, Michigan, USA.
¹⁸ The Heart Institute, Cincinnati Children's Hospital Medical Center and Department of Pediatrics, University of Cincinnati School of Medicine, Cincinnati, Ohio, USA.
¹⁹ Division of Cardiology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, USA.

PMID: 38939312
PMCID: PMC11198056
DOI: 10.1016/j.jacadv.2022.100029

Cumulative In-Hospital Costs Associated With Single-Ventricle Palliation

Michael L O'Byrne et al. JACC Adv. 2022.

. 2022 May 26;1(2):100029.

doi: 10.1016/j.jacadv.2022.100029. eCollection 2022 Jun.

Authors

Affiliations

¹ Division of Cardiology, The Children's Hospital of Philadelphia and Department of Pediatrics, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, Pennsylvania, USA.
² Center for Pediatric Clinical Effectiveness, The Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, USA.
³ Leonard Davis Institute of Health Economics and Cardiovascular Outcomes, Quality, and Evaluative Research Center, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, Pennsylvania, USA.
⁴ Division of Cardiology, Medical University of South Carolina, Charleston, South Carolina, USA.
⁵ Department of Biostatistics, Center for Clinical Epidemiology and Biostatistics, Perelman School of Medicine, Philadelphia, Pennsylvania, USA.
⁶ Data Science and Biostatistics Unit, Department of Biomedical and Health Informatics, The Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, USA.
⁷ Division of Cardiology, New York-Presbyterian Morgan-Stanley Children's Hospital/Columbia University Irving Medical Center, New York, New York, USA.
⁸ Department of Cardiology, Children's Hospital Boston and Harvard University Medical School, Boston, Massachusetts, USA.
⁹ Children's Healthcare of Atlanta, Sibley Heart Center and Emory University School of Medicine, Atlanta, Georgia, USA.
¹⁰ Sections of Critical Care and Cardiology, Texas Children's Hospital and Baylor College of Medicine, Houston, Texas, USA.
¹¹ Children's Hospital of Wisconsin, Milwaukee, Wisconsin, USA.
¹² Division of Cardiothoracic Surgery, Departments of Surgery and Pediatrics, Congenital Heart Center, University of Florida, Gainesville, Florida, USA.
¹³ Division of Pediatric Cardiology, Duke University School of Medicine, Durham, North Carolina, USA.
¹⁴ Division of Cardiology, Department of Pediatrics, Children's Hospital of Los Angeles, Keck School of Medicine of University of Southern California, Los Angeles, California, USA.
¹⁵ Department of Pediatrics, Labatt Family Heart Centre, The Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada.
¹⁶ Division of Cardiology, Primary Children's Hospital and University of Utah School of Medicine, Salt Lake City, Utah, USA.
¹⁷ Division of Pediatric Cardiothoracic Surgery, C.S. Mott Children's Hospital and University of Michigan School of Medicine, Ann Arbor, Michigan, USA.
¹⁸ The Heart Institute, Cincinnati Children's Hospital Medical Center and Department of Pediatrics, University of Cincinnati School of Medicine, Cincinnati, Ohio, USA.
¹⁹ Division of Cardiology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, USA.

PMID: 38939312
PMCID: PMC11198056
DOI: 10.1016/j.jacadv.2022.100029

Abstract

Background: In the SVR (Single Ventricle Reconstruction) Trial, 1-year survival in recipients of right ventricle to pulmonary artery shunts (RVPAS) was superior to that in those receiving modified Blalock-Taussig-Thomas shunts (MBTTS), but not in subsequent follow-up. Cost analysis is an expedient means of evaluating value and morbidity.

Objectives: The purpose of this study was to evaluate differences in cumulative hospital costs between RVPAS and MBTTS.

Methods: Clinical data from SVR and costs from Pediatric Health Information Systems database were combined. Cumulative hospital costs and cost-per-day-alive were compared serially at 1, 3, and 5 years between RVPAS and MBTTS. Potential associations between patient-level factors and cost were explored with multivariable models.

Results: In total, 303 participants (55% of the SVR cohort) from 9 of 15 sites were studied (48% MBTTS). Observed total costs at 1 year were lower for MBTTS ($701,260 ± 442,081) than those for RVPAS ($804,062 ± 615,068), a difference that was not statistically significant (P = 0.10). Total costs were also not significantly different at 3 and 5 years (P = 0.21 and 0.32). Similarly, cost-per-day-alive did not differ significantly for either group at 1, 3, and 5 years (all P > 0.05). In analyses of transplant-free survivors, total costs and cost-per-day-alive were higher for RVPAS at 1 year (P = 0.05 for both) but not at 3 and 5 years (P > 0.05 for all). In multivariable models, aortic atresia and prematurity were associated with increased cost-per-day-alive across follow-up (P < 0.05).

Conclusions: Total costs do not differ significantly between MBTTS and RVPAS. The magnitude of longitudinal costs underscores the importance of efforts to improve outcomes in this vulnerable population.

Keywords: economic analysis; hypoplastic left heart syndrome; outcomes research; pediatrics.

PubMed Disclaimer

Conflict of interest statement

The SVR trial was funded by the Pediatric Heart Network/10.13039/100000050National Heart, Lung, and Blood Institute (HL068269, HL068270, HL068279, HL068281, HL068285, HL068288, HL068290, HL068292, and HL085057). Dr O'Byrne received support from 10.13039/100000050NHLBI (K23 HL130420-01). The project also received assistance from The Pediatric Heart Network’s Integrated CARDiac Data and Outcomes Collaborative (iCARD). The Pediatric Heart Network Ancillary Studies Committee and Single Ventricle Reconstruction Trial Committee reviewed the proposed research and manuscript for appropriateness but did not participate in the drafting of the paper. The views expressed are solely of the authors and do not reflect those of the funders and other supporting groups. All other authors have reported that they have no relationships relevant to the contents of this paper to disclose.PERSPECTIVESCOMPETENCY IN MEDICAL KNOWLEDGE: Costs (which are reflective of morbidity) overall did not differ between participants randomized to a Blalock-Taussig-Thomas shunt and those to a RVPAS. However, analysis of survivors suggests that the cost of living with a RVPAS is higher, consistent with higher reintervention rates, while higher mortality rates increased costs of the Blalock-Taussig-Thomas shunt strategy. Differences in cost were derived from differences in the first year of life where the majority of total costs were accrued. Efforts to improve care should focus on improving outcomes in this period. TRANSLATIONAL OUTLOOK: Although it did not reveal significant differences in costs between shunt types, this analysis of the SVR trial leads to a better understanding of the economic and medical impact of operative palliation for single ventricle heart disease.

Figures

**Central Illustration**
Association of Shunt Type and Cumulative In-Hospital Costs After Norwood Procedure **(A)** Norwood procedure with right ventricle to pulmonary artery shunt **(Top)** and modified Blalock-Taussig-Thomas shunt **(Bottom)**. Image from Menon et al.(B) Total costs and cost-per-day alive. Box-and-whiskers plot depicting the accumulation of total cumulative costs **(Top)** and cost-per-day-alive **(Bottom)** for Blalock-Taussig-Thomas Shunt (MBTTS) **(blue)** and right ventricle to pulmonary artery shunt (RVPAS) **(red)**. The **solid line in the box** depicts the median cost, the **open circle** depicts the mean cost, the **box** depicts the range between the 25th and 75th percentiles, and the **whiskers** depict 1.5 times the interquartile range. SVR = single ventricle reconstruction.

**Figure 1**
Study Population MBTT = modified Blalock-Taussig-Thomas shunt; PHIS = Pediatric Health Information Systems Database; RV PA = right ventricle to pulmonary artery; SVR = single ventricle reconstruction.

See this image and copyright information in PMC

References

1. Ohye R.G., Schonbeck J.V., Eghtesady P., et al. Cause, timing, and location of death in the single ventricle reconstruction trial. J Thorac Cardiovasc Surg. 2012;144:907–914. - PMC - PubMed
1. Ohye R.G., Sleeper L.A., Mahony L., et al. Comparison of shunt types in the Norwood procedure for single-ventricle lesions. N Engl J Med. 2010;362:1980–1992. - PMC - PubMed
1. O'Brien S.M., Clarke D.R., Jacobs J.P., et al. An empirically based tool for analyzing mortality associated with congenital heart surgery. J Thorac Cardiovasc Surg. 2009;138:1139–1153. - PubMed
1. Menon S.C., Erickson L.K., McFadden M., Miller D.V. Effect of ventriculotomy on right-ventricular remodeling in hypoplastic left heart syndrome: a histopathological and echocardiography correlation study. Pediatr Cardiol. 2013;34:354–363. - PubMed
1. Newburger J.W., Sleeper L.A., Frommelt P.C., et al. Transplantation-free survival and interventions at 3 years in the single ventricle reconstruction trial. Circulation. 2014;129:2013–2020. - PMC - PubMed

Grants and funding

T32 HL007572/HL/NHLBI NIH HHS/United States

LinkOut - more resources

Full Text Sources

Save citation to file

Email citation

Add to Collections

Add to My Bibliography

Your saved search

Create a file for external citation management software

Your RSS Feed

Cumulative In-Hospital Costs Associated With Single-Ventricle Palliation

Affiliations

Cumulative In-Hospital Costs Associated With Single-Ventricle Palliation

Authors

Affiliations

Abstract

Conflict of interest statement

Figures

References

Grants and funding

LinkOut - more resources

Full Text Sources