First name
Bryan
Middle name
H
Last name
Goldstein

Title

Comparison of management strategies for neonates with symptomatic tetralogy of Fallot and weight <2.5 kg.

Year of Publication

2021

Date Published

2021 Feb 03

ISSN Number

1097-685X

Abstract

OBJECTIVE: To compare management strategies for neonates <2.5 kg with tetralogy of Fallot and symptomatic cyanosis who either undergo staged repair (SR) (initial palliation followed by later complete repair) or primary repair (PR).

METHODS: Consecutive neonates with tetralogy of Fallot and symptomatic cyanosis weighing <2.5 kg at initial intervention and between 2005 and 2017 were retrospectively reviewed from the Congenital Cardiac Research Collaborative. Primary outcome was mortality and secondary outcomes included component (eg, initial palliation, complete repair, or primary repair) and cumulative (SR: initial palliation followed by later complete repair) hospital and intensive care unit lengths of stay, durations of ventilation, inotrope use, cardiopulmonary bypass time, procedural complications, and reintervention. Outcomes were compared with propensity score adjustments with PR as the reference group.

RESULTS: The cohort included 76 SR (initial palliation: 53 surgical and 23 transcatheter) and 44 PR patients. The observed risk of overall mortality was similar between SR and PR groups (15.8% vs 18.2%: P = .735). The adjusted hazard of mortality remained similar between groups overall (hazard ratio, 0.59; 95% confidence interval, 0.26-1.36; P = .214), as well as during short-term (<4 months: hazard ratio, 0.37; 95% confidence interval, 0.13-1.09; P = .071) and midterm (>4 months: hazard ratio, 1.32; 95% confidence interval, 0.30-5.79; P = .717) follow-up. Reintervention in the first 18 months was common in both groups (53.2% vs 48.4%; hazard ratio, 1.69; 95% confidence interval, 0.96-2.28; P = .072). Adjusted procedural complications and neonatal morbidity burden were overall lower in the SR group. Cumulative secondary outcome burdens largely favored the PR group.

CONCLUSIONS: In this study comparing SR and PR treatment strategies for neonates with tetralogy of Fallot and symptomatic cyanosis and weight <2.5 kg, mortality and reintervention burden was highly independent of treatment strategy. Other potential advantages were observed with each approach.

DOI

10.1016/j.jtcvs.2021.01.100

Alternate Title

J Thorac Cardiovasc Surg

PMID

33726912

Title

Comparative Costs of Management Strategies for Neonates With Symptomatic Tetralogy of Fallot.

Year of Publication

2022

Number of Pages

1170-1180

Date Published

2022 Mar 29

ISSN Number

1558-3597

Abstract

BACKGROUND: Recent data have demonstrated that overall mortality and adverse events are not significantly different for primary repair (PR) and staged repair (SR) approaches to management of neonates with symptomatic tetralogy of Fallot (sTOF). Cost data can be used to compare the relative value (cost for similar outcomes) of these approaches and are a potentially more sensitive measure of morbidity.

OBJECTIVES: This study sought to compare the economic costs associated with PR and SR in neonates with sTOF.

METHODS: Data from a multicenter retrospective cohort study of neonates with sTOF were merged with administrative data to compare total costs and cost per day alive over the first 18 months of life in a propensity score-adjusted analysis. A secondary analysis evaluated differences in department-level costs.

RESULTS: In total, 324 subjects from 6 centers from January 2011 to November 2017 were studied (40% PR). The 18-month cumulative mortality (P = 0.18), procedural complications (P = 0.10), hospital complications (P = 0.94), and reinterventions (P = 0.22) did not differ between PR and SR. Total 18-month costs for PR (median $179,494 [IQR: $121,760-$310,721]) were less than for SR (median: $222,799 [IQR: $167,581-$327,113]) (P < 0.001). Cost per day alive (P = 0.005) and department-level costs were also all lower for PR. In propensity score-adjusted analyses, PR was associated with lower total cost (cost ratio: 0.73; P < 0.001) and lower department-level costs.

CONCLUSIONS: In this multicenter study of neonates with sTOF, PR was associated with lower costs. Given similar overall mortality between treatment strategies, this finding suggests that PR provides superior value.

DOI

10.1016/j.jacc.2021.12.036

Alternate Title

J Am Coll Cardiol

PMID

35331412

Title

Factors Influencing Reintervention Following Ductal Artery Stent Implantation for Ductal-Dependent Pulmonary Blood Flow: Results From the Congenital Cardiac Research Collaborative.

Year of Publication

2021

Number of Pages

CIRCINTERVENTIONS120010086

Date Published

2021 Nov 18

ISSN Number

1941-7632

Abstract

<p><strong>BACKGROUND: </strong>Stenting of the patent ductus arteriosus (PDA) is an established palliative option for infants with ductal-dependent pulmonary blood flow. Following initial palliation, reintervention on the PDA stent is common, but risk factors have not been characterized.</p>

<p><strong>METHODS: </strong>Infants with ductal-dependent pulmonary blood flow palliated with PDA stent between 2008 and 2015 were reviewed within the Congenital Cardiac Research Collaborative. Rates and risk factors for reintervention were analyzed.</p>

<p><strong>RESULTS: </strong>Among 105 infants who underwent successful PDA stenting, 41 patients (39%) underwent a total of 53 reinterventions on the PDA stent, with all but one occurring within 6 months of the initial intervention. Stent redilation constituted the majority of reintervention (n=35; 66%) followed by additional stent placement (n=11; 21%) and surgical shunt placement (n=7; 13%). The majority of reintervention was nonurgent, and there were no deaths during the reintervention procedure. All but one reintervention occurred within 6 months of the initial procedure. On univariate analysis, risk factors for reintervention included anticipated single-ventricle physiology, lack of prior balloon pulmonary valvuloplasty, use of drug-eluting stent, and increased ductal tortuosity.</p>

<p><strong>CONCLUSIONS: </strong>In infants with ductal-dependent pulmonary blood flow palliated with PDA stent implantation, reintervention is common, can be performed safely, and is associated with both anatomic/procedural factors and anticipated final physiology.</p>

DOI

10.1161/CIRCINTERVENTIONS.120.010086

Alternate Title

Circ Cardiovasc Interv

PMID

34789017

Title

Comparison of Patent Ductus Arteriosus Stent and Blalock-Taussig Shunt as Palliation for Neonates with Sole Source Ductal-Dependent Pulmonary Blood Flow: Results from the Congenital Catheterization Research Collaborative.

Year of Publication

2021

Date Published

2021 Sep 15

ISSN Number

1432-1971

Abstract

<p>Patent ductus arteriosus (PDA) stenting is an accepted method for securing pulmonary blood flow in cyanotic neonates. In neonates with pulmonary atresia and single source ductal-dependent pulmonary blood flow (SSPBF), PDA stenting remains controversial. We sought to evaluate outcomes in neonates with SSPBF, comparing PDA stenting and surgical Blalock-Taussig shunt (BTS). Neonates with SSPBF who underwent PDA stenting or BTS at the four centers of the Congenital Catheterization Research Collaborative from January 2008 to December 2015 were retrospectively reviewed. Reintervention on the BTS or PDA stent prior to planned surgical repair served as the primary endpoint. Additional analyses of peri-procedural complications, interventions, and pulmonary artery growth were performed. A propensity score was utilized to adjust for differences in factors. Thirty-five patients with PDA stents and 156 patients with BTS were included. The cohorts had similar baseline characteristics, procedural complications, and mortality. Interstage reintervention rates were higher in the PDA stent cohort (48.6% vs. 15.4%, p &lt; 0.001).</p>

DOI

10.1007/s00246-021-02699-7

Alternate Title

Pediatr Cardiol

PMID

34524483

Title

Data quality methods through remote source data verification auditing: results from the Congenital Cardiac Research Collaborative.

Year of Publication

2021

Number of Pages

1-6

Date Published

2021 Mar 17

ISSN Number

1467-1107

Abstract

<p><strong>BACKGROUND: </strong>Multicentre research databases can provide insights into healthcare processes to improve outcomes and make practice recommendations for novel approaches. Effective audits can establish a framework for reporting research efforts, ensuring accurate reporting, and spearheading quality improvement. Although a variety of data auditing models and standards exist, barriers to effective auditing including costs, regulatory requirements, travel, and design complexity must be considered.</p>

<p><strong>MATERIALS AND METHODS: </strong>The Congenital Cardiac Research Collaborative conducted a virtual data training initiative and remote source data verification audit on a retrospective multicentre dataset. CCRC investigators across nine institutions were trained to extract and enter data into a robust dataset on patients with tetralogy of Fallot who required neonatal intervention. Centres provided de-identified source files for a randomised 10% patient sample audit. Key auditing variables, discrepancy types, and severity levels were analysed across two study groups, primary repair and staged repair.</p>

<p><strong>RESULTS: </strong>Of the total 572 study patients, data from 58 patients (31 staged repairs and 27 primary repairs) were source data verified. Amongst the 1790 variables audited, 45 discrepancies were discovered, resulting in an overall accuracy rate of 97.5%. High accuracy rates were consistent across all CCRC institutions ranging from 94.6% to 99.4% and were reported for both minor (1.5%) and major discrepancies type classifications (1.1%).</p>

<p><strong>CONCLUSION: </strong>Findings indicate that implementing a virtual multicentre training initiative and remote source data verification audit can identify data quality concerns and produce a reliable, high-quality dataset. Remote auditing capacity is especially important during the current COVID-19 pandemic.</p>

DOI

10.1017/S1047951121000974

Alternate Title

Cardiol Young

PMID

33726868

Title

Comparison of Management Strategies for Neonates With Symptomatic Tetralogy of Fallot.

Year of Publication

2021

Number of Pages

1093-1106

Date Published

2021 Mar 02

ISSN Number

1558-3597

Abstract

<p><strong>BACKGROUND: </strong>Neonates with tetralogy of Fallot and symptomatic cyanosis (sTOF) require early intervention.</p>

<p><strong>OBJECTIVES: </strong>This study sought to perform a balanced multicenter comparison of staged repair (SR) (initial palliation [IP] and subsequent complete repair [CR]) versus primary repair (PR) treatment strategies.</p>

<p><strong>METHODS: </strong>Consecutive neonates with sTOF who underwent IP or PR at&nbsp;≤30&nbsp;days of age from 2005 to 2017 were retrospectively reviewed from the Congenital Cardiac Research Collaborative. The primary outcome was death. Secondary outcomes included component (IP, CR, PR) and cumulative (SR): hospital and intensive care unit lengths of stay; durations of cardiopulmonary bypass, anesthesia, ventilation, and inotrope use; and complication and reintervention rates. Outcomes were compared using propensity score adjustment.</p>

<p><strong>RESULTS: </strong>The cohort consisted of 342 patients who underwent SR (IP: surgical, n&nbsp;=&nbsp;256; transcatheter, n&nbsp;=&nbsp;86) and 230 patients who underwent PR. Pre-procedural ventilation, prematurity, DiGeorge syndrome, and pulmonary atresia were more common in the SR group (p&nbsp;≤0.01). The observed risk of death was not different between the groups (10.2% vs 7.4%; p&nbsp;=&nbsp;0.25) at median 4.3 years. After adjustment, the hazard of death remained similar between groups (hazard ratio: 0.82; 95% confidence interval: 0.49 to 1.38; p&nbsp;=&nbsp;0.456), but it favored SR during early follow-up (&lt;4&nbsp;months; p&nbsp;=&nbsp;0.041). Secondary outcomes favored the SR group in component analysis, whereas they largely favored PR in cumulative analysis. Reintervention risk was higher in the SR group (p&nbsp;=&nbsp;0.002).</p>

<p><strong>CONCLUSIONS: </strong>In this multicenter comparison of SR or PR for management of neonates with sTOF, adjusted for patient-related factors, early mortality and neonatal morbidity were lower in the SR group, but cumulative morbidity and reinterventions favored the PR group, findings suggesting potential benefits to each strategy.</p>

DOI

10.1016/j.jacc.2020.12.048

Alternate Title

J Am Coll Cardiol

PMID

33632484

Title

Impact of Treatment Strategy on Outcomes in Isolated Pulmonary Artery of Ductal Origin.

Year of Publication

2021

Date Published

2021 Jan 04

ISSN Number

1432-1971

Abstract

<p>Isolated pulmonary artery (PA) of ductal origin (IPADO) is a rare cardiac defect which requires surgical repair, with or without preceding palliation. We sought to determine the impact of treatment strategy on outcomes. Retrospective study of consecutive patients with IPADO that underwent staged or primary repair from 1/05 to 9/16 at 6 Congenital Cardiac Research Collaborative centers. Patients with single ventricle physiology, major aortopulmonary collaterals, or bilateral IPADO were excluded. Primary outcome was isolated PA z-score at late follow-up. Secondary outcomes included PA symmetry index (isolated:confluent PA diameter) and reintervention burden. Propensity score adjustment was used to account for baseline differences. Of 60 patients in the study cohort, 26 (43%) underwent staged and 34 (57%) primary repair. The staged and primary repair groups differed in weight at diagnosis and presence of other heart disease but not in baseline PA dimensions. Staged patients underwent ductal stent (n = 16) or surgical shunt (n = 10) placement followed by repair at 210 vs. 21 days in the primary repair group (p &lt; 0.001). At median follow-up of 4.5 years post-repair, after adjustment, isolated PA z-score (- 0.74 [- 1.75, - 0.26] vs. - 1.95 [- 2.91, - 1.59], p = 0.012) and PA symmetry index (0.81 [0.49, 1.0] vs. 0.55 [0.48, 0.69], p = 0.042) significantly favored the staged repair group. Freedom from PA reintervention was not different between groups (adjusted HR 0.78 [0.41, 1.48]; p = 0.445). A staged approach to repair of IPADO is associated with superior isolated PA size and symmetry at late follow-up. Consideration should be given to initial palliation in IPADO patients, when feasible.</p>

DOI

10.1007/s00246-020-02511-y

Alternate Title

Pediatr Cardiol

PMID

33394118

Title

Impact of Palliation Strategy on Interstage Feeding and Somatic Growth for Infants With Ductal-Dependent Pulmonary Blood Flow: Results from the Congenital Catheterization Research Collaborative.

Year of Publication

2020

Number of Pages

e013807

Date Published

2020 Jan 07

ISSN Number

2047-9980

Abstract

<p><strong>Background</strong> In infants with ductal-dependent pulmonary blood flow, the impact of palliation strategy on interstage growth and feeding regimen is unknown.</p>

<p><strong>Methods and Results</strong> This was a retrospective multicenter study of infants with ductal-dependent pulmonary blood flow palliated with patent ductus arteriosus (PDA) stent or Blalock-Taussig shunt (BTS) from 2008 to 2015. Subjects with a defined interstage, the time between initial palliation and subsequent palliation or repair, were included. Primary outcome was change in weight-for-age -score. Secondary outcomes included % of patients on: all oral feeds, feeding-related medications, higher calorie feeds, and feeding-related readmission. Propensity score was used to account for baseline differences. Subgroup analysis was performed in 1- (1V) and 2-ventricle (2V) groups. The cohort included 66 PDA stent (43.9% 1V) and 195 BTS (54.4% 1V) subjects. Prematurity was more common in the PDA stent group (=0.051). After adjustment, change in weight-for-age -score did not differ between groups over the entire interstage. However, change in weight-for-age -score favored PDA stent during the inpatient interstage (=0.005) and BTS during the outpatient interstage (=0.032). At initial hospital discharge, PDA stent treatment was associated with all oral feeds (&lt;0.001) and absence of feeding-related medications (=0.002). Subgroup analysis revealed that 2V but not 1V patients demonstrated significant increase in weight-for-age -score. In the 2V cohort, feeding-related readmissions were more common in the BTS group (=0.008).</p>

<p><strong>Conclusions </strong>In infants with ductal-dependent pulmonary blood flow who underwent palliation with PDA stent or BTS, there was no difference in interstage growth. PDA stent was associated with a simpler feeding regimen and fewer feeding-related readmissions.</p>

DOI

10.1161/JAHA.119.013807

Alternate Title

J Am Heart Assoc

PMID

31852418

Title

Use of carotid and axillary artery approach for stenting the patent ductus arteriosus in infants with ductal-dependent pulmonary blood flow: A multicenter study from the congenital catheterization research collaborative.

Year of Publication

2019

Date Published

2019 Dec 09

ISSN Number

1522-726X

Abstract

<p><strong>BACKGROUND: </strong>Carotid artery (CA) and axillary artery (AA) access are increasingly used for transcatheter stenting of the patent ductus arteriosus (PDA), although reports are limited.</p>

<p><strong>METHODS: </strong>The Congenital Catheterization Research Collaborative (CCRC) reviewed multicenter data from infants who underwent PDA stenting via the CA or AA approach from 2008 to 2017, and compared outcomes to those of infants undergoing PDA stenting via the femoral artery (FA) approach. Post-procedure ultrasound (US) imaging was reviewed.</p>

<p><strong>RESULTS: </strong>Forty-nine infants underwent PDA stenting from the CA (n = 43) or AA (n = 6) approach, compared with 55 infants who underwent PDA stenting from the FA approach. The PDA was the sole pulmonary blood flow (PBF) source in 61% of infants in the CA/AA cohort, compared with 33% of the FA cohort (p &lt; .01). Ductal tortuosity for CA/AA cohort was Type I (straight) in 10 (20%), Type II (one turn) in 17 (35%), and Type III (multiple turns) in 22 (45%) infants and reflected a greater degree of tortuosity when compared to the FA cohort (p &lt; .01). In 17 infants with CA/AA approach, the "flip technique" was used, and was associated with shorter procedure times for highly tortuous PDA (Type III) patients. Rates of procedural complications were similar across access sites. Most common complications were access site injury (thrombus or bleeding) and stent malposition. No complications were specifically related to the "flip technique."</p>

<p><strong>CONCLUSIONS: </strong>Use of CA and AA approach for PDA stenting was found to be more commonly employed in sole source PBF and highly tortuous PDAs. Procedural modifications such as the "flip technique" may lead to shorter procedure times. CA and AA approaches are associated with a similar burden of procedural or late complications. Post-procedural surveillance of the CA and AA is suggested, given the incidence of vascular findings on US.</p>

DOI

10.1002/ccd.28631

Alternate Title

Catheter Cardiovasc Interv

PMID

31815357

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