New Study Compares Costs of Management Strategies for Symptomatic Tetralogy of Fallot in Neonates

A new study published in the Journal of the American College of Cardiology (JACC) details procedural and care cost comparisons for the treatment of neonates with symptomatic tetralogy of Fallot (sTOF) through either a primary or staged repair strategy.¹

Bryan H. Goldstein, MD, FACC, FSCAI, FAAP, Director of the Cardiac Catheterization Laboratory in the Heart Institute at UPMC Children's Hospital of Pittsburgh, was the senior author of the study conducted by the Congenital Cardiac Research Collaborative (CCRC), which Dr. Goldstein co-founded and continues to co-lead.

This new study builds upon previous work from Dr. Goldstein et al. published in JACC in 2021, and in the Journal of Thoracic and Cardiovascular Surgery (JTCVS) in 2022, which found similarities in outcomes and mortality rates of staged (SR) versus primary repair (PR) for sTOF.^2,3 That 2021 JACC study did find several significant differences between the treatment groups. The staged approach to repair was associated with a reduced hazard of early mortality, but this benefit was lost over time when comparisons of longer-term outcomes were made. A crucial aspect of the study examined a range of procedural morbidities between the PR and SR cohorts. The 2021 study found a clear benefit to risks of neonatal morbidities with the staged repair pathway. There was a reduced burden of potentially toxic exposures such as volatile anesthetics, cardiopulmonary bypass, ICU and hospital length of stay, and duration of mechanical ventilatory support – all during the critical neonatal period, when organ maturity and brain development are at their most vulnerable. However, the cumulative burden of exposure to these morbidities was generally lower for the primary repair group. Likewise, reinterventions were more common in the staged repair group, but this largely reflected reinterventions during the interstage, before definitive TOF repair. Following definitive repair, there was no difference in the rate of reintervention. The 2022 JTCVS study of sTOF neonates < 2.5 kg in weight also demonstrated relative parity in outcomes between the treatment groups, but a striking 16% to 18% mortality risk, overall.

Given the findings from the earlier investigations, and the continuing debate in the field as to the preferred management strategy in the sTOF population, Dr. Goldstein and his CCRC colleagues delved into an investigation of the costs of care for staged versus primary repair pathways to understand the potentially disparate economic impact of these management strategies. The group felt that cost was a metric that was not only important to stakeholders (providers, families, payers), but it also may be the single measure that best reflects the burden and complexity of care provided for a given patient and treatment pathway. Stated another way, cost incorporates both length of stay and complexity of that stay, thus accounting for morbidities acquired, reinterventions performed, hospital days, etc.

“With our findings from the earlier studies, they begged the question: If the two procedural pathways provide roughly similar clinical outcomes, should we be concentrating our efforts primarily on the path that incurs fewer costs?” says Dr. Goldstein. 

Cost Comparisons of the Staged Versus Primary Repair Pathway

The new study examined the total costs of care of the two repair pathways from birth hospitalization to 18 months of age. In order to execute this ambitious project, investigators from the CCRC had to combine patient-level clinical data from their sTOF project with patient-level cost data derived from the Pediatric Health Information System (PHIS) database.

The CCRC found that mean costs for the primary repair approach were $179,494, while mean costs of care for the staged repair pathway were found to be higher at $222,799.

“The primary approach to repair ends up costing less because there are fewer incidents of care required, even though, on average, the primary repair patients stay in the hospital longer after the first intervention," says Dr. Goldstein. "Adding in the second component of the staged repair strategy, and potentially other factors such as interstage reinterventions, these drive up the costs, which ultimately favor the primary repair strategy from this perspective.”

As Dr. Goldstein points out, however, not all hospitals or centers have the capability to perform a neonatal primary repair of sTOF with the same high-level of clinical outcomes that the tertiary-care academic centers that participated in the CCRC study can achieve. In this regard, the findings from the analysis are not necessarily generalizable to every institution. 

“However, for those centers that can realize similar clinical outcomes, our study suggests that with an equivalence of clinical outcomes, the lower cost strategy ought to reflect a solid, single metric that considers all the aspects of care between the two treatment strategies.”

In the absence of additional data, the study suggests that the primary repair approach is the optimal pathway, in cases where a primary repair is a medically feasible option, and there exist no confounding medical complexities that would preclude a primary repair approach. Dr. Goldstein is quick to note, however, that this study fails to account for potentially impactful late outcomes, which could reflect yet unmeasured differences between the treatment strategies, such as school-age neurodevelopmental outcomes, or adolescent cardiopulmonary exercise performance.

“We think this study will influence, at the hospital level, which pathway makes the most sense to pursue. If an institution can perform primary repair and achieve the same average level of outcomes as the academic centers in our study, from a cost perspective, it makes the case that primary repair ought to be the approach used when feasible,” says Dr. Goldstein.

More Information

Please read the new study and its complete analysis and conclusions using the following reference. Reference for the earlier 2021 and 2022 studies on primary versus staged repair outcomes also follows for interested readers.

Learn more about Dr. Goldstein, the Heart Institute at UPMC Children’s, and the Congenital Cardiac Research Collaborative.

References

1. O’Byrne ML, Glatz AC, Huang Y-S V, Kelleman MS, Petit CJ Qureshi AM, Shahanavaz S, Nicholson GT, Batlivala S, Meadows JJ, Zampi JD, Law MA, Romano JC, Mascio CE, Chai PJ, Maskatia S, Asztalos IB, Beshish A, Pettus J, Pajk AL, Healan SJ, Eilers LF, Merritt T, McCracken CE, Goldstein BH. Comparative Costs of Management Strategies for Neonates With Symptomatic Tetralogy of Fallot. J Am Coll Cardiol. 2022; 79: 1170-1180.

2. Goldstein BH, Petit CJ, Qureshi AM, McCracken CE, Kelleman MS, Nicholson GT, Law MA, Meadows JJ, Zampi JD, Shahanavaz S, Mascio CE, Chai PJ, Romano JC, Asztalos IB, Kamsheh AM, Healan SJ, Smith J, Ligon RA, Pettus JA, Juma S, Raulston JJ, Huskey JL, Batlivala SP, Pajk AL, Eilers LF, Khan HQ, Merritt TC, Candor M, Juergensen S, Rinderknecht FA, Bauser-Heaton H, Glatz AC.  Comparison of Management Strategies for the Neonate with Symptomatic Tetralogy of Fallot. J Am Coll Cardiol. 2021 Mar 2; 77(8): 1093-1106. 

3. Qureshi AM, Caldarone CA, Romano JC, Chai PJ, Mascio CE, Glatz AC, Petit CJ, McCracken CE, Kelleman MS, Nicholson GT, Meadows JJ, Zampi JD, Shahanavaz S, Law MA, Batlivala SP, Goldstein BH; Congenital Cardiac Research Collaborative Investigators. Comparison of Management Strategies for the Neonate With Symptomatic Tetralogy of Fallot and Weight Less Than 2.5 kg. J Thorac Cardiovasc Surg. 2022 Jan; 163(1): 192-207.e3.