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Paediatric subaortic stenosis : long-term outcome and risk factors for reoperation

Rik De Wolf, Katrien Francois (UGent) , Thierry Bové (UGent) , Ilse Coomans (UGent) , Katya De Groote (UGent) , Hans De Wilde (UGent) , Joseph Panzer (UGent) , Kristof Vandekerckhove (UGent) and Daniël De Wolf (UGent)
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Abstract
Abstract Objectives: Surgical repair of subaortic stenosis (SAS) is associated with a substantial reoperation risk. We aimed to identify risk factors for reintervention in relation to discrete and tunnel-type SAS morphology. Methods: Single-centre retrospective study of paediatric SAS diagnosed between 1992 and 2017. Multivariable Cox regression analysis was performed to identify reintervention risk factors. Results: Eighty-five children [median age 2.5 (0.7-6.5) years at diagnosis] with a median follow-up of 10.1 (5.5-16.4) years were included. Surgery was executed in 83% (n = 71). Freedom from reoperation was 88 ± 5% at 5 years and 82 ± 6% at 10 years for discrete SAS, compared to, respectively, 33 ± 16% and 17 ± 14% for tunnel-type SAS (log-rank P < 0.001). Independent risk factors for reintervention were a postoperative gradient >20 mmHg [hazard ratio (HR) 6.56, 95% confidence interval (CI) 1.41-24.1; P = 0.005], tunnel-type SAS (HR 7.46, 95% CI 2.48-22.49; P < 0.001), aortic annulus z-score <-2 (HR 11.07, 95% CI 3.03-40.47; P < 0.001) and age at intervention <2 years (HR 3.24, 95% CI 1.09-9.86; P = 0.035). Addition of septal myectomy at initial intervention was not associated with lesser reintervention. Fourteen children with a lower left ventricular outflow tract (LVOT) gradient (P < 0.001) and older age at diagnosis (P = 0.024) were followed expectatively. Conclusions: Children with SAS remain at risk for reintervention, despite initially effective LVOT relief. Regardless of SAS morphology, age <2 years at first intervention, a postoperative gradient >20 mmHg and presence of a hypoplastic aortic annulus are independent risk factors for reintervention. More extensive LVOT surgery might be considered at an earlier stage in these children. SAS presenting in older children with a low LVOT gradient at diagnosis shows little progression, justifying an expectative approach
Keywords
Surgery, Pulmonary and Respiratory Medicine, Cardiology and Cardiovascular Medicine, Subaortic stenosis, Congenital heart disease, Left ventricular outflow tract, SUBVALVULAR AORTIC-STENOSIS, NATURAL-HISTORY, FOLLOW-UP, CHILDREN, REPAIR, ADULTS, OBSTRUCTION, PREVALENCE, RESECTION, RELIEF

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MLA
De Wolf, Rik, et al. “Paediatric Subaortic Stenosis : Long-Term Outcome and Risk Factors for Reoperation.” INTERACTIVE CARDIOVASCULAR AND THORACIC SURGERY, vol. 33, no. 4, 2021, pp. 588–96, doi:10.1093/icvts/ivab121.
APA
De Wolf, R., Francois, K., Bové, T., Coomans, I., De Groote, K., De Wilde, H., … De Wolf, D. (2021). Paediatric subaortic stenosis : long-term outcome and risk factors for reoperation. INTERACTIVE CARDIOVASCULAR AND THORACIC SURGERY, 33(4), 588–596. https://doi.org/10.1093/icvts/ivab121
Chicago author-date
De Wolf, Rik, Katrien Francois, Thierry Bové, Ilse Coomans, Katya De Groote, Hans De Wilde, Joseph Panzer, Kristof Vandekerckhove, and Daniël De Wolf. 2021. “Paediatric Subaortic Stenosis : Long-Term Outcome and Risk Factors for Reoperation.” INTERACTIVE CARDIOVASCULAR AND THORACIC SURGERY 33 (4): 588–96. https://doi.org/10.1093/icvts/ivab121.
Chicago author-date (all authors)
De Wolf, Rik, Katrien Francois, Thierry Bové, Ilse Coomans, Katya De Groote, Hans De Wilde, Joseph Panzer, Kristof Vandekerckhove, and Daniël De Wolf. 2021. “Paediatric Subaortic Stenosis : Long-Term Outcome and Risk Factors for Reoperation.” INTERACTIVE CARDIOVASCULAR AND THORACIC SURGERY 33 (4): 588–596. doi:10.1093/icvts/ivab121.
Vancouver
1.
De Wolf R, Francois K, Bové T, Coomans I, De Groote K, De Wilde H, et al. Paediatric subaortic stenosis : long-term outcome and risk factors for reoperation. INTERACTIVE CARDIOVASCULAR AND THORACIC SURGERY. 2021;33(4):588–96.
IEEE
[1]
R. De Wolf et al., “Paediatric subaortic stenosis : long-term outcome and risk factors for reoperation,” INTERACTIVE CARDIOVASCULAR AND THORACIC SURGERY, vol. 33, no. 4, pp. 588–596, 2021.
@article{8708455,
  abstract     = {{Abstract
Objectives: Surgical repair of subaortic stenosis (SAS) is associated with a substantial reoperation risk. We aimed to identify risk factors for reintervention in relation to discrete and tunnel-type SAS morphology.

Methods: Single-centre retrospective study of paediatric SAS diagnosed between 1992 and 2017. Multivariable Cox regression analysis was performed to identify reintervention risk factors.

Results: Eighty-five children [median age 2.5 (0.7-6.5) years at diagnosis] with a median follow-up of 10.1 (5.5-16.4) years were included. Surgery was executed in 83% (n = 71). Freedom from reoperation was 88 ± 5% at 5 years and 82 ± 6% at 10 years for discrete SAS, compared to, respectively, 33 ± 16% and 17 ± 14% for tunnel-type SAS (log-rank P < 0.001). Independent risk factors for reintervention were a postoperative gradient >20 mmHg [hazard ratio (HR) 6.56, 95% confidence interval (CI) 1.41-24.1; P = 0.005], tunnel-type SAS (HR 7.46, 95% CI 2.48-22.49; P < 0.001), aortic annulus z-score <-2 (HR 11.07, 95% CI 3.03-40.47; P < 0.001) and age at intervention <2 years (HR 3.24, 95% CI 1.09-9.86; P = 0.035). Addition of septal myectomy at initial intervention was not associated with lesser reintervention. Fourteen children with a lower left ventricular outflow tract (LVOT) gradient (P < 0.001) and older age at diagnosis (P = 0.024) were followed expectatively.

Conclusions: Children with SAS remain at risk for reintervention, despite initially effective LVOT relief. Regardless of SAS morphology, age <2 years at first intervention, a postoperative gradient >20 mmHg and presence of a hypoplastic aortic annulus are independent risk factors for reintervention. More extensive LVOT surgery might be considered at an earlier stage in these children. SAS presenting in older children with a low LVOT gradient at diagnosis shows little progression, justifying an expectative approach}},
  author       = {{De Wolf, Rik and Francois, Katrien and Bové, Thierry and Coomans, Ilse and De Groote, Katya and De Wilde, Hans and Panzer, Joseph and Vandekerckhove, Kristof and De Wolf, Daniël}},
  issn         = {{1569-9293}},
  journal      = {{INTERACTIVE CARDIOVASCULAR AND THORACIC SURGERY}},
  keywords     = {{Surgery,Pulmonary and Respiratory Medicine,Cardiology and Cardiovascular Medicine,Subaortic stenosis,Congenital heart disease,Left ventricular outflow tract,SUBVALVULAR AORTIC-STENOSIS,NATURAL-HISTORY,FOLLOW-UP,CHILDREN,REPAIR,ADULTS,OBSTRUCTION,PREVALENCE,RESECTION,RELIEF}},
  language     = {{eng}},
  number       = {{4}},
  pages        = {{588--596}},
  title        = {{Paediatric subaortic stenosis : long-term outcome and risk factors for reoperation}},
  url          = {{http://dx.doi.org/10.1093/icvts/ivab121}},
  volume       = {{33}},
  year         = {{2021}},
}

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