The changing epidemiology of Ebstein's anomaly and its relationship with maternal mental health conditions: a European registry-based study
Por:
Boyle B, Garne E, Loane M, Addor MC, Arriola L, Cavero-Carbonell C, Gatt M, Lelong N, Lynch C, Nelen V, Neville AJ, O'Mahony M, Pierini A, Rissmann A, Tucker D, Zymak-Zakutnia N and Dolk H
Publicada:
1 may 2017
Ahead of Print:
30 ago 2016
Resumen:
Objectives: The aim of this study was to describe the epidemiology of Ebstein's anomaly in Europe and its association with maternal health and medication exposure during pregnancy. Design: We carried out a descriptive epidemiological analysis of population-based data. Setting: We included data from 15 European Surveillance of Congenital Anomalies Congenital Anomaly Registries in 12 European countries, with a population of 5.6 million births during 1982-2011. Participants: Cases included live births, fetal deaths from 20 weeks gestation, and terminations of pregnancy for fetal anomaly. Main outcome measures: We estimated total prevalence per 10,000 births. Odds ratios for exposure to maternal illnesses/medications in the first trimester of pregnancy were calculated by comparing Ebstein's anomaly cases with cardiac and non-cardiac malformed controls, excluding cases with genetic syndromes and adjusting for time period and country. Results: In total, 264 Ebstein's anomaly cases were recorded; 81% were live births, 2% of which were diagnosed after the 1st year of life; 54% of cases with Ebstein's anomaly or a co-existing congenital anomaly were prenatally diagnosed. Total prevalence rose over time from 0.29 (95% confidence interval (CI) 0.20-0.41) to 0.48 (95% CI 0.40-0.57) (p < 0.01). In all, nine cases were exposed to maternal mental health conditions/medications (adjusted odds ratio (adjOR) 2.64, 95% CI 1.33-5.21) compared with cardiac controls. Cases were more likely to be exposed to maternal beta-thalassemia (adjOR 10.5, 95% CI 3.13-35.3, n = 3) and haemorrhage in early pregnancy (adjOR 1.77, 95% CI 0.93-3.38, n = 11) compared with cardiac controls. Conclusions: The increasing prevalence of Ebstein's anomaly may be related to better and earlier diagnosis. Our data suggest that Ebstein's anomaly is associated with maternal mental health problems generally rather than lithium or benzodiazepines specifically; therefore, changing or stopping medications may not be preventative. We found new associations requiring confirmation.
Filiaciones:
Boyle B:
1Centre for Maternal, Fetal and Infant Research,Institute of Nursing and Health Research,Ulster University,Belfast,UK
Garne E:
2Paediatric Department,Hospital Lillebaelt,Kolding,Denmark
Loane M:
1Centre for Maternal, Fetal and Infant Research,Institute of Nursing and Health Research,Ulster University,Belfast,UK
Addor MC:
3Division of Medical Genetics,CHUV,Lausanne,Switzerland
Arriola L:
4Registro Anomalías Congénitas de la CAPV, Public Health Division of Gipuzkoa,Instituto BIO-Donostia,Basque Government, CIBER Epidemiología y Salud Pública - CIBERESP,Bilbao,Spain
:
5Foundation for the Promotion of Health and Biomedical Research in the Valencian Region,Rare Diseases Research Area,Valencia,Spain
Gatt M:
6Department of Health Information and Research Guardamangia,Malta
Lelong N:
7Paris Registry of Congenital Malformations INSERM U953,Paris,France
Lynch C:
8Health Service Executive, Kilkenny,Ireland
Nelen V:
9Department of Environment,PIH,Antwerp,Province of Antwerp,Belgium
Neville AJ:
10Registro IMER - IMER Registry (Emila Romagna Registry of Birth Defects),Azienda Ospedaliero - Universitaria di FerraraCorso Giovecca,Ferrara,Italy
O'Mahony M:
11Health Service Executive, Cork,Ireland
Pierini A:
12CNR Institute of Clinical Physiology,Pisa,Italy
Rissmann A:
13Medical Faculty,Malformation Monitoring Centre,Otto-von-Guericke University,Magdeburg,Saxony-Anhalt,Germany
Tucker D:
14Public Health Wales. Swansea,UK
Zymak-Zakutnia N:
15Khmelnytsky Regional Medical Genetic Centre,Rivne,Ukraine
Dolk H:
1Centre for Maternal, Fetal and Infant Research,Institute of Nursing and Health Research,Ulster University,Belfast,UK
Green Submitted, Bronze, Green Accepted
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