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1 Determinants of Outcome after Surgical Treatment of Pulmonary Atresia with Ventricular Septal Defect and Major Aortopulmonary Collateral Arteries Presenter Disclosure Adriano Carotti, MD The following relationships exist related to this presentation: Relationships to Disclose Department of Pediatric Cardiology and Cardiac Surgery Bambino Gesu Children s Hospital Roma, Italy
2 Aim of the Study Analysis of results of a fifteen-year experience with surgical treatment of PA-VSD-MAPCAs Identification of variables influencing both early and late outcome with a particular focus on the impact of unifocalization procedures
3 Management protocol PA-VSD-MAPCAs Angiography Hypoplastic dominant pulmonary arteries (PAI<100mm 2 /m 2 ) RVOTR or Central shunt Abnormal P.Art. arborization Dominant MAPCAs Exclusive MAPCAs Midline complete unifocalization and flow-study Unifocalization and staged intracardiac repair Low PA pressure ( 30 mmhg) Simultaneous intracardiac repair High PA pressure (>30 mmhg) Staged intracardiac repair
4 Demographics Number of pts January 1994 September (30%) 63 (70%) Chromosome 22q11 deletion (pts) 33 (37%) Median age Median weight at 1st treatment: at unifocalization: at 1st treatment: at unifocalization: 12 mos (20 dd - 35 yrs) 15 mos (20 dd - 35 yrs) 8.2 kg ( kg) 8.7 kg ( kg)
5 Characteristics of pulmonary blood supply N. pts % TNPAI = Cross sectional area of MAPCAs + PAs (mm 2 ) Body Surface Area (m 2 ) <150 mm 2 /m mm 2 /m 2 64 Dominant collateral or pulmonary arteries Spa:Sca <1 Spa:Sca >1 Central pulmonary artery/ies, single, bilaterally Confluent intrapericardial pulmonary arteries Confluent intraparenchimal pulmonary arteries
6 Surgical treatment Pts with PA-VSD-MAPCAs n = 90 Hypoplastic dominant pulmonary arteries n = 21 RVOTR n = 19 Central shunt n = 2 Complete unifocalization n = 17 Flow-study n = 15 Abnormal P.Art. arborization n = 17 Dominant MAPCAs n = 40 Exclusive MAPCAs n = 12 Complete unifocalization n = 69 Flow-study n = 56 Simultaneous VSD closure n = 15 In-staging n = 2 In-staging n = 4 Late death n = 2 Early death n = 1 Late death n = 4 Early death n = 6 Late death n = 1 Early death n = 1 Simultaneous VSD closure n = 44 VSD left open n = 25 Delayed VSD closure n = 11 In-staging n = 7
7 Outcome N.pts Staging to promote native pulmonary arterial growth 21 23% Total unifocalization - 1 st stage - 2 nd stage VSD management at unifocalization - Intraoperative pulmonary flow study - VSD closed - VSD left open % 80% 20% % 69% 31% VSD delayed closure 11 41% Complete repair 70 78% Postoperative early prv/plv ratio after repair (all patients) Postoperative early prv/plv ratio after repair (patients who entered paired t test analysis) ± ±0.14
8 Follow-up N.Observations 78 Median interval (mos) 46 ( ) Conduit reinterventions: N.Pts 25 Percutaneous intervention on PAs: N.Pts - Once (N. Pts) - Twice or more times (N. Pts) Bronchial stenting: N.Pts
9 Survival 30 days: 92% PA-VSD-MAPCAs Kaplan-Meier Estimates months 95% CI Survival function months 95% CI Freedom from conduit reintervention months 95% CI Freedom from percutaneous intervention on PAs
10 prv/plv Comparison (paired t test) 1 0,9 0,8 0,7 0,6 0, ,4 0,3 0,2 0,1 0 Early p.o. Follow-up early p.o. median 95 mos f.u. ( mos) Variable Obs Mean Std.Err. Std.Dev. p Value prv/plv p.o. prv/plv f.u. Diff ,
11 End-points of univariate and multivariate analysis Suitability to one-stage unifocalization Suitability to simultaneous VSD closure Postoperative prv/plv ratio after VSD closure Survival
12 Variables examined Age Weight TNPAI Categorical 30 vs >30 days Continuous Categorical 3 vs >3 kgs Continuous <150 vs 150 mm 2 /m 2 Confluent intrapericardial pulmonary arteries vs Confluent intraparenchimal pulmonary arteries vs Central pulmonary artery/ies vs Chromosome 22q11.2 microdeletion vs Dominant collateral or pulmonary arteries MAPCAs vs PAs Era at treatment vs
13 End-points of univariate and multivariate analysis Suitability to one-stage unifocalization Suitability to simultaneous VSD closure Postoperative prv/plv ratio after VSD closure Survival
14 Univariate model fit for suitability to one-stage unifocalization (χ 2 - Fisher) Group Obs (n=90) (n=4) (n=86) p Value Age 30 days >30 days Weight 3 kgs >3 kgs TNPAI <150 mm 2 /m mm 2 /m Confluent intrapericardial PAs Confluent intraparenchimal PAs Central pulmonary artery/ies, single, bilaterally Chromosome 22q11.2 microdeletion Dominant collateral or PAs MAPCAs PAs Era at treatment
15 Univariate model fit for suitability to one-stage unifocalization (two-sample t test) GROUP Obs Mean 95% CI p Value Age Weight
16 End-points of univariate and multivariate analysis Suitability to one-stage unifocalization Suitability to simultaneous VSD closure Postoperative prv/plv ratio after VSD closure Survival
17 Univariate model fit for suitability to simultaneous VSD closure (χ 2 - Fisher) Group Obs (n=86) (n=27) (n=59) p Value Age 30 days >30 days Weight 3 kgs >3 kgs TNPAI <150 mm 2 /m mm 2 /m Confluent intrapericardial PAs Confluent intraparenchimal PAs Central pulmonary artery/ies, single, bilaterally Chromosome 22q11.2 microdeletion Dominant collateral or PAs MAPCAs PAs Era at treatment
18 Univariate model fit for suitability to simultaneous VSD closure (two-sample t test) GROUP Obs Mean 95% CI p Value Age Weight
19 Logistic regression model fit for suitability to simultaneous VSD closure Group Odds Ratio p Value 95% CI Chromosome 22q11 deletion Era at treatment
20 End-points of univariate and multivariate analysis Suitability to one-stage unifocalization Suitability to simultaneous VSD closure Postoperative prv/plv ratio after VSD closure Survival
21 Univariate model fit for postoperative prv/plv (ANOVA) Group (Obs = 70) Coef. p Value 95% CI Age Categorical 30 vs >30 days Continuous Weight Categorical 3 vs >3 kgs Continuous TNPAI <150 vs 150 mm 2 /m Confluent intrapericardial PAs (0) vs (1) Confluent intraparenchimal PAs vs Central pulmonary artery/ies vs Chromosome 22q11 deletion vs Dominant collateral or pulmonary arteries MAPCAs vs PAs Era at treatment vs
22 End-points of univariate and multivariate analysis Suitability to one-stage unifocalization Suitability to simultaneous VSD closure Postoperative prv/plv ratio after VSD closure Survival
23 Kaplan-Meier survival estimates comparison (log-rank test) p = p = months months Age at unifocalization: 30 days >30 days Weight at unifocalization: 3 kg >3 kg p = p = 0.01 months months del 22q: Simultaneous VSD closure:
24 Cox regression model fit for mortality Group Haz.ratio p Value 95% CI Age (continous) Weight (continuous) Chromosome 22q11 deletion Dominant collateral or PAs Chromosome 22q11 deletion Era at treatment
25 Limitations of the Study Single-center retrospective study in which a treatment protocol was analyzed in the absence of a control group Follow-up was incomplete for 8 out of 78 patients (10%), due to insufficient data retrieval from foreign countries Some results of the analysis are probably biased by the use of specific parameters (e.g. TNPAI) as selection criteria
26 Conclusions 1 Results of one-stage complete unifocalization for PA- VSD-MAPCAs are satisfactory and durable despite the need of repeated percutaneous or surgical reinterventions The use of MAPCAs for neo-pulmonary arterial reconstruction assures functional pulmonary blood flow, compensates for defects in true pulmonary arterial distribution, and is the only option in cases with absent pulmonary arteries
27 Conclusions 2 Avoiding neonatal age and low body weight at unifocalization may increase the chance of positive outcome, as it does the simultaneous closure of ventricular septal defect, when feasible Presence of confluent intrapericardial pulmonary arteries does not improve the hemodynamic result after VSD closure Chromosome 22q11 deletion remains an independent variable affecting survival
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