Thirty-Year Survival after Cardiac Surgery for Patients with Turner Syndrome
To evaluate long-term survival in patients with Turner syndrome after congenital heart surgery with a focus on left heart obstructive lesions (LHOLs). We queried the Pediatric Cardiac Care Consortium, a US-based registry of congenital heart surgery, for patients with Turner syndrome undergoing conge...
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Published in | The Journal of pediatrics Vol. 239; pp. 187 - 192.e1 |
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Main Authors | , , , , , , , |
Format | Journal Article |
Language | English |
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Elsevier Inc
01.12.2021
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Abstract | To evaluate long-term survival in patients with Turner syndrome after congenital heart surgery with a focus on left heart obstructive lesions (LHOLs).
We queried the Pediatric Cardiac Care Consortium, a US-based registry of congenital heart surgery, for patients with Turner syndrome undergoing congenital heart surgery at <21 years of age between 1982 and 2011. Outcomes were obtained from the Pediatric Cardiac Care Consortium and from national death and transplant registries through 2019. Survival of patients with Turner syndrome and nonsyndromic patients with similar LHOL was compared by Kaplan-Meier survival curves and Cox regression adjusted for age, congenital heart disease, and era.
We identified 179 patients with Turner syndrome operated for LHOL: 161 with 2-ventricle lesions (coarctation n = 149, aortic stenosis n = 12) and 18 with hypoplastic left heart (HLH) variants. There were 157 with 2-ventricle LHOL and 6 with HLH survived to discharge. Among survivors to hospital discharge, the 30-year transplant-free survival was 90.4% for Turner syndrome with 2-ventricle lesions and 90.9% for nonsyndromic comparators (adjusted hazard ratio [aHR] 1.15, 95% CI 0.64-2.04). The postdischarge survival for HLH was 33% for Turner syndrome and 51% for nonsyndromic patients, with these numbers being too small for meaningful comparisons. There was a higher risk for cardiovascular disease events in patients with Turner syndrome vs male (aHR 3.72, 95% CI 1.64-8.39) and female comparators (aHR 4.55, 95% CI 1.87-11.06) excluding heart failure deaths.
The 30-year transplant-free survival is similar for patients with Turner syndrome and nonsyndromic comparators with operated 2-ventricle LHOL without excess congenital heart disease risk. However, patients with Turner Syndrome still face increased cardiovascular disease morbidity, stressing the importance of lifelong comorbidity surveillance in this population. |
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AbstractList | To evaluate long-term survival in patients with Turner syndrome after congenital heart surgery with a focus on left heart obstructive lesions (LHOLs).
We queried the Pediatric Cardiac Care Consortium, a US-based registry of congenital heart surgery, for patients with Turner syndrome undergoing congenital heart surgery at <21 years of age between 1982 and 2011. Outcomes were obtained from the Pediatric Cardiac Care Consortium and from national death and transplant registries through 2019. Survival of patients with Turner syndrome and nonsyndromic patients with similar LHOL was compared by Kaplan-Meier survival curves and Cox regression adjusted for age, congenital heart disease, and era.
We identified 179 patients with Turner syndrome operated for LHOL: 161 with 2-ventricle lesions (coarctation n = 149, aortic stenosis n = 12) and 18 with hypoplastic left heart (HLH) variants. There were 157 with 2-ventricle LHOL and 6 with HLH survived to discharge. Among survivors to hospital discharge, the 30-year transplant-free survival was 90.4% for Turner syndrome with 2-ventricle lesions and 90.9% for nonsyndromic comparators (adjusted hazard ratio [aHR] 1.15, 95% CI 0.64-2.04). The postdischarge survival for HLH was 33% for Turner syndrome and 51% for nonsyndromic patients, with these numbers being too small for meaningful comparisons. There was a higher risk for cardiovascular disease events in patients with Turner syndrome vs male (aHR 3.72, 95% CI 1.64-8.39) and female comparators (aHR 4.55, 95% CI 1.87-11.06) excluding heart failure deaths.
The 30-year transplant-free survival is similar for patients with Turner syndrome and nonsyndromic comparators with operated 2-ventricle LHOL without excess congenital heart disease risk. However, patients with Turner Syndrome still face increased cardiovascular disease morbidity, stressing the importance of lifelong comorbidity surveillance in this population. OBJECTIVETo evaluate long-term survival in patients with Turner syndrome after congenital heart surgery with a focus on left heart obstructive lesions (LHOLs). STUDY DESIGNWe queried the Pediatric Cardiac Care Consortium, a US-based registry of congenital heart surgery, for patients with Turner syndrome undergoing congenital heart surgery at <21 years of age between 1982 and 2011. Outcomes were obtained from the Pediatric Cardiac Care Consortium and from national death and transplant registries through 2019. Survival of patients with Turner syndrome and nonsyndromic patients with similar LHOL was compared by Kaplan-Meier survival curves and Cox regression adjusted for age, congenital heart disease, and era. RESULTSWe identified 179 patients with Turner syndrome operated for LHOL: 161 with 2-ventricle lesions (coarctation n = 149, aortic stenosis n = 12) and 18 with hypoplastic left heart (HLH) variants. There were 157 with 2-ventricle LHOL and 6 with HLH survived to discharge. Among survivors to hospital discharge, the 30-year transplant-free survival was 90.4% for Turner syndrome with 2-ventricle lesions and 90.9% for nonsyndromic comparators (adjusted hazard ratio [aHR] 1.15, 95% CI 0.64-2.04). The postdischarge survival for HLH was 33% for Turner syndrome and 51% for nonsyndromic patients, with these numbers being too small for meaningful comparisons. There was a higher risk for cardiovascular disease events in patients with Turner syndrome vs male (aHR 3.72, 95% CI 1.64-8.39) and female comparators (aHR 4.55, 95% CI 1.87-11.06) excluding heart failure deaths. CONCLUSIONSThe 30-year transplant-free survival is similar for patients with Turner syndrome and nonsyndromic comparators with operated 2-ventricle LHOL without excess congenital heart disease risk. However, patients with Turner Syndrome still face increased cardiovascular disease morbidity, stressing the importance of lifelong comorbidity surveillance in this population. |
Author | Sassis, Leandros Kochilas, Lazaros Wechsler, Stephanie Burns Kefala-Karli, Pelagia Claxton, J'Neka S. Silberbach, Michael Alam, Sabikha Mortillo, Michael |
AuthorAffiliation | 2 Children’s Healthcare of Atlanta and Sibley Heart Center Cardiology, Atlanta, GA 5 Doernbecher Children’s Hospital, Oregon Health & Sciences University, Portland, OR 4 University of Nicosia Medical School, Nicosia, Cyprus 6 Department of Human Genetics, Emory University School of Medicine, Atlanta, GA 1 Department of Pediatrics, Emory University School of Medicine, Atlanta, GA 3 Rollins School of Public Health Emory University, Atlanta, GA |
AuthorAffiliation_xml | – name: 2 Children’s Healthcare of Atlanta and Sibley Heart Center Cardiology, Atlanta, GA – name: 6 Department of Human Genetics, Emory University School of Medicine, Atlanta, GA – name: 5 Doernbecher Children’s Hospital, Oregon Health & Sciences University, Portland, OR – name: 4 University of Nicosia Medical School, Nicosia, Cyprus – name: 1 Department of Pediatrics, Emory University School of Medicine, Atlanta, GA – name: 3 Rollins School of Public Health Emory University, Atlanta, GA |
Author_xml | – sequence: 1 givenname: Sabikha orcidid: 0000-0001-8516-3279 surname: Alam fullname: Alam, Sabikha email: sabikha.alam@gmail.com organization: Department of Pediatrics, Emory University School of Medicine, Atlanta, GA – sequence: 2 givenname: J'Neka S. orcidid: 0000-0002-2029-9754 surname: Claxton fullname: Claxton, J'Neka S. organization: Department of Pediatrics, Emory University School of Medicine, Atlanta, GA – sequence: 3 givenname: Michael orcidid: 0000-0002-5917-158X surname: Mortillo fullname: Mortillo, Michael organization: Rollins School of Public Health Emory University, Atlanta, GA – sequence: 4 givenname: Leandros orcidid: 0000-0003-3064-1434 surname: Sassis fullname: Sassis, Leandros organization: University of Nicosia Medical School, Nicosia, Cyprus – sequence: 5 givenname: Pelagia orcidid: 0000-0003-4265-9033 surname: Kefala-Karli fullname: Kefala-Karli, Pelagia organization: University of Nicosia Medical School, Nicosia, Cyprus – sequence: 6 givenname: Michael surname: Silberbach fullname: Silberbach, Michael organization: Doernbecher Children's Hospital, Oregon Health & Sciences University, Portland, OR – sequence: 7 givenname: Lazaros surname: Kochilas fullname: Kochilas, Lazaros organization: Department of Pediatrics, Emory University School of Medicine, Atlanta, GA – sequence: 8 givenname: Stephanie Burns orcidid: 0000-0002-9321-0220 surname: Wechsler fullname: Wechsler, Stephanie Burns organization: Department of Pediatrics, Emory University School of Medicine, Atlanta, GA |
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Keywords | HLH HRs aHR Turner syndrome CVD NDI PCCC left heart obstructive lesions congenital heart disease mortality LHOL PAPVR CHD OPTN |
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Snippet | To evaluate long-term survival in patients with Turner syndrome after congenital heart surgery with a focus on left heart obstructive lesions (LHOLs).
We... OBJECTIVETo evaluate long-term survival in patients with Turner syndrome after congenital heart surgery with a focus on left heart obstructive lesions (LHOLs).... |
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SubjectTerms | Adolescent Cardiac Surgical Procedures Child Child, Preschool congenital heart disease Female Follow-Up Studies Humans Infant Infant, Newborn left heart obstructive lesions Male mortality Registries Retrospective Studies Survival Analysis Treatment Outcome Turner syndrome Turner Syndrome - mortality Turner Syndrome - surgery Young Adult |
Title | Thirty-Year Survival after Cardiac Surgery for Patients with Turner Syndrome |
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