Aortic dilatation and outcome in women with Turner syndrome.


Journal

Heart (British Cardiac Society)
ISSN: 1468-201X
Titre abrégé: Heart
Pays: England
ID NLM: 9602087

Informations de publication

Date de publication:
05 2019
Historique:
received: 08 06 2018
revised: 13 09 2018
accepted: 27 09 2018
pubmed: 29 10 2018
medline: 12 5 2020
entrez: 29 10 2018
Statut: ppublish

Résumé

Women with Turner syndrome (TS) are at increased risk of aortic dissection, which is related to ascending aortic diameter. However, the relation between aortic diameter and outcome is not well determined. This study evaluates the prevalence of aortic dilatation, the growth rate of the aorta and the risk of aortic complications in adults with TS. Single centre, retrospective study of all women with TS followed with a strict protocol in an outpatient TS clinic. Aortic diameters were analysed using advanced imaging. The primary outcome was a combined endpoint of aortic-related mortality, aortic dissection and preventive aortic surgery. The secondary endpoint was aortic growth and prevalence of aortic dilatation, defined as an aortic size index >20 mm/m At least one cardiac MR/CT was available in 268 women with TS, having median age of 28.7 (IQR: 21.3-39.7) years. Aortic dilatation was present in 22%. Linear regression identified independent factors associated with larger aortic diameters: age (coefficient=0.23; p<0.001), hypertension (coefficient=2.7; p<0.001), bicuspid aortic valve (coefficient=3.3; p<0.001), 45XO karyotype (coefficient=1.7; p=0.002), weight (coefficient=0.075; p<0.001) and growth hormone treatment (coefficient=1.4; p=0.044). During follow-up (6.8±3.2 years), five women (2%) reached the primary endpoint (two dissections, three aortic surgery). Women withmore than one scan (n=171; 1015 patient-years follow-up), the median aortic growth was 0.20 (IQR: 0.00-0.44) mm/year. In multivariate analysis, aortic growth was not associated with baseline aortic diameter or other variables. Aortic dilatation is common and known associations were confirmed in large adult TS cohort However, aortic dissection, related mortality and preventive aortic surgery are rare. Growth hormone treatment in childhood was associated with aortic dimensions.

Sections du résumé

BACKGROUND
Women with Turner syndrome (TS) are at increased risk of aortic dissection, which is related to ascending aortic diameter. However, the relation between aortic diameter and outcome is not well determined. This study evaluates the prevalence of aortic dilatation, the growth rate of the aorta and the risk of aortic complications in adults with TS.
METHODS
Single centre, retrospective study of all women with TS followed with a strict protocol in an outpatient TS clinic. Aortic diameters were analysed using advanced imaging. The primary outcome was a combined endpoint of aortic-related mortality, aortic dissection and preventive aortic surgery. The secondary endpoint was aortic growth and prevalence of aortic dilatation, defined as an aortic size index >20 mm/m
RESULTS
At least one cardiac MR/CT was available in 268 women with TS, having median age of 28.7 (IQR: 21.3-39.7) years. Aortic dilatation was present in 22%. Linear regression identified independent factors associated with larger aortic diameters: age (coefficient=0.23; p<0.001), hypertension (coefficient=2.7; p<0.001), bicuspid aortic valve (coefficient=3.3; p<0.001), 45XO karyotype (coefficient=1.7; p=0.002), weight (coefficient=0.075; p<0.001) and growth hormone treatment (coefficient=1.4; p=0.044). During follow-up (6.8±3.2 years), five women (2%) reached the primary endpoint (two dissections, three aortic surgery). Women withmore than one scan (n=171; 1015 patient-years follow-up), the median aortic growth was 0.20 (IQR: 0.00-0.44) mm/year. In multivariate analysis, aortic growth was not associated with baseline aortic diameter or other variables.
CONCLUSIONS
Aortic dilatation is common and known associations were confirmed in large adult TS cohort However, aortic dissection, related mortality and preventive aortic surgery are rare. Growth hormone treatment in childhood was associated with aortic dimensions.

Identifiants

pubmed: 30368486
pii: heartjnl-2018-313716
doi: 10.1136/heartjnl-2018-313716
doi:

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

693-700

Informations de copyright

© Author(s) (or their employer(s)) 2019. No commercial re-use. See rights and permissions. Published by BMJ.

Déclaration de conflit d'intérêts

Competing interests: None declared.

Auteurs

Anthonie L Duijnhouwer (AL)

Department of Cardiology, Radboud University Medical Centre, Nijmegen, The Netherlands.

Lidia R Bons (LR)

Department of Cardiology, Erasmus MC, Rotterdam, The Netherlands.

Henri J L M Timmers (HJLM)

Department of Endocrinology, Radboud University Medical Centre, Nijmegen, The Netherlands.

Roland R L van Kimmenade (RRL)

Department of Cardiology, Radboud University Medical Centre, Nijmegen, The Netherlands.

Miranda Snoeren (M)

Department of Radiology, Radboud University Medical Centre, Nijmegen, The Netherlands.

Janneke Timmermans (J)

Department of Cardiology, Radboud University Medical Centre, Nijmegen, The Netherlands.

Allard T van den Hoven (AT)

Department of Cardiology, Radboud University Medical Centre, Nijmegen, The Netherlands.

Marlies Kempers (M)

Department of Medical Genetics, Radboud University Medical Centre, Nijmegen, The Netherlands.

Kathrin Fleischer (K)

Department of Gynaecology, Radboud University Medical Centre, Nijmegen, Netherlands.

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