{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,11,4]],"date-time":"2026-11-04T12:37:09Z","timestamp":1793795829483,"version":"4.6.1"},"reference-count":56,"publisher":"Oxford University Press (OUP)","issue":"1","license":[{"start":{"date-parts":[[2023,10,27]],"date-time":"2023-10-27T00:00:00Z","timestamp":1698364800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"funder":[{"name":"Erasmus University Medical Centre"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2024,1,2]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p\/>\n                  <jats:sec>\n                    <jats:title>OBJECTIVES<\/jats:title>\n                    <jats:p>Congenital supravalvular aortic stenosis (SVAS) is a rare form of congenital outflow tract obstruction and long-term outcomes are scarcely reported. This study aims to provide an overview of outcomes after surgical repair for congenital SVAS.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>METHODS<\/jats:title>\n                    <jats:p>A systematic review of published literature was conducted, including observational studies reporting long-term clinical outcome (&amp;gt;2\u2009years) after SVAS repair in children or adults considering &amp;gt;20 patients. Early risks, late event rates and time-to-event data were pooled and entered into a microsimulation model to estimate 30-year outcomes. Life expectancy was compared to the age-, sex- and origin-matched general population.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>RESULTS<\/jats:title>\n                    <jats:p>Twenty-three publications were included, encompassing a total of 1472 patients (13\u00a0125 patient-years; pooled mean follow-up: 9.0 (6.2) years; median follow-up: 6.3\u2009years). Pooled mean age at surgical repair was 4.7 (5.8) years and the most commonly used surgical technique was the single-patch repair (43.6%). Pooled early mortality was 4.2% (95% confidence interval: 3.2\u20135.5%) and late mortality was 0.61% (95% CI: 0.45\u20130.83) per patient-year. Based on microsimulation, over a 30-year time horizon, it was estimated that an average patient with SVAS repair (mean age: 4.7\u2009years) had an observed life expectancy that was 90.7% (95% credible interval: 90.0\u201391.6%) of expected life expectancy in the matched general population. The microsimulation-based 30-year risk of myocardial infarction was 8.1% (95% credible interval: 7.3\u20139.9%) and reintervention 31.3% (95% credible interval: 29.6\u201333.4%), of which 27.2% (95% credible interval: 25.8\u201329.1) due to repair dysfunction.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>CONCLUSIONS<\/jats:title>\n                    <jats:p>After surgical repair for SVAS, 30-year survival is lower than the matched-general-population survival and the lifetime risk of reintervention is considerable. Therefore, lifelong monitoring of the cardiovascular system and in particular residual stenosis and coronary obstruction is recommended.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1093\/ejcts\/ezad360","type":"journal-article","created":{"date-parts":[[2023,10,26]],"date-time":"2023-10-26T08:52:50Z","timestamp":1698310370000},"source":"Crossref","is-referenced-by-count":19,"title":["Long-term surgical outcomes of congenital supravalvular aortic stenosis: a systematic review, meta-analysis and microsimulation study"],"prefix":"10.1093","volume":"65","author":[{"given":"Frederike","family":"Meccanici","sequence":"first","affiliation":[{"name":"Department of Cardiology, Erasmus University Medical Centre , Rotterdam, Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3828-6775","authenticated-orcid":false,"given":"Maximiliaan L","family":"Notenboom","sequence":"additional","affiliation":[{"name":"Department of Cardiothoracic Surgery, Erasmus University Medical Centre Rotterdam, Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jade","family":"Meijssen","sequence":"additional","affiliation":[{"name":"Department of Cardiology, Erasmus University Medical Centre , Rotterdam, Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vernon","family":"Smit","sequence":"additional","affiliation":[{"name":"Department of Cardiology, Erasmus University Medical Centre , Rotterdam, Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0826-7837","authenticated-orcid":false,"given":"Pieter C","family":"van de Woestijne","sequence":"additional","affiliation":[{"name":"Department of Cardiothoracic Surgery, Erasmus University Medical Centre Rotterdam, Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Annemien E","family":"van den Bosch","sequence":"additional","affiliation":[{"name":"Department of Cardiology, Erasmus University Medical Centre , Rotterdam, Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1023-1537","authenticated-orcid":false,"given":"Willem A","family":"Helbing","sequence":"additional","affiliation":[{"name":"Department of Paediatrics, Division of Paediatric Cardiology, Erasmus University Medical Centre , Rotterdam, Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6200-1998","authenticated-orcid":false,"given":"Ad J J C","family":"Bogers","sequence":"additional","affiliation":[{"name":"Department of Cardiothoracic Surgery, Erasmus University Medical Centre Rotterdam, Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9809-5623","authenticated-orcid":false,"given":"Johanna J M","family":"Takkenberg","sequence":"additional","affiliation":[{"name":"Department of Cardiothoracic Surgery, Erasmus University Medical Centre Rotterdam, Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6770-3830","authenticated-orcid":false,"given":"Jolien W","family":"Roos-Hesselink","sequence":"additional","affiliation":[{"name":"Department of Cardiology, Erasmus University Medical Centre , Rotterdam, Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2023,10,27]]},"reference":[{"key":"2024011113244354100_ezad360-B1","first-page":"107","article-title":"Aortic stenosis in children: 19-year experience","volume":"59","author":"Liu","year":"1997","journal-title":"Zhonghua Yi Xue Za Zhi (Taipei)"},{"key":"2024011113244354100_ezad360-B2","doi-asserted-by":"crossref","first-page":"195","DOI":"10.1016\/S1010-7940(00)00647-3","article-title":"Congenital supravalvar aortic stenosis: a simple lesion?","volume":"19","author":"Stamm","year":"2001","journal-title":"Eur J Cardiothorac Surg"},{"key":"2024011113244354100_ezad360-B3","doi-asserted-by":"crossref","first-page":"692","DOI":"10.1161\/CIRCGENETICS.112.962860","article-title":"Supravalvular aortic stenosis: elastin arteriopathy","volume":"5","author":"Merla","year":"2012","journal-title":"Circ Cardiovasc Genet"},{"key":"2024011113244354100_ezad360-B4","doi-asserted-by":"crossref","first-page":"142","DOI":"10.1161\/01.CIR.92.1.142","article-title":"Genetic approaches to cardiovascular disease. 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