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Long term trends in prevalence of neural tube defects in Europe: population based study

Long term trends in prevalence of neural tube defects in Europe: population based study
Long term trends in prevalence of neural tube defects in Europe: population based study
Study question: what are the long term trends in the total (live births, fetal deaths, and terminations of pregnancy for fetal anomaly) and live birth prevalence of neural tube defects (NTD) in Europe, where many countries have issued recommendations for folic acid supplementation but a policy for mandatory folic acid fortification of food does not exist?

Methods: this was a population based, observational study using data on 11?353 cases of NTD not associated with chromosomal anomalies, including 4162 cases of anencephaly and 5776 cases of spina bifida from 28 EUROCAT (European Surveillance of Congenital Anomalies) registries covering approximately 12.5 million births in 19 countries between 1991 and 2011. The main outcome measures were total and live birth prevalence of NTD, as well as anencephaly and spina bifida, with time trends analysed using random effects Poisson regression models to account for heterogeneities across registries and splines to model non-linear time trends.

Summary answer and limitations: overall, the pooled total prevalence of NTD during the study period was 9.1 per 10?000 births. Prevalence of NTD fluctuated slightly but without an obvious downward trend, with the final estimate of the pooled total prevalence of NTD in 2011 similar to that in 1991. Estimates from Poisson models that took registry heterogeneities into account showed an annual increase of 4% (prevalence ratio 1.04, 95% confidence interval 1.01 to 1.07) in 1995-99 and a decrease of 3% per year in 1999-2003 (0.97, 0.95 to 0.99), with stable rates thereafter. The trend patterns for anencephaly and spina bifida were similar, but neither anomaly decreased substantially over time. The live birth prevalence of NTD generally decreased, especially for anencephaly. Registration problems or other data artefacts cannot be excluded as a partial explanation of the observed trends (or lack thereof) in the prevalence of NTD.

What this study adds: in the absence of mandatory fortification, the prevalence of NTD has not decreased in Europe despite longstanding recommendations aimed at promoting peri-conceptional folic acid supplementation and existence of voluntary folic acid fortification
0959-8138
h5949
Khoshnood, Babak
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Loane, Maria
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Walle, Hermien de
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Arriola, Larraitz
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Addor, Marie-Claude
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Barisic, Ingeborg
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Beres, Judit
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Bianchi, Fabrizio
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Dias, Carlos
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Draper, Elizabeth
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Garne, Ester
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Gatt, Miriam
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Haeusler, Martin
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Klungsoyr, Kari
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Latos-Bielenska, Anna
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McDonnell, Bob
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Nelen, Vera
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Neville, Amanda J.
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O’Mahony, Mary T.
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Queisser-Luft, Annette
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Rankin, Judith
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Rissmann, Anke
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Ritvanen, Annukka
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Rounding, Catherine
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Sipek, Antonin
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Tucker, David
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Verellen-Dumoulin, Christine
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Wellesley, Diana
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Dolk, Helen
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Khoshnood, Babak
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Loane, Maria
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Walle, Hermien de
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Arriola, Larraitz
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Addor, Marie-Claude
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Barisic, Ingeborg
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Beres, Judit
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Bianchi, Fabrizio
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Dias, Carlos
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Draper, Elizabeth
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Garne, Ester
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Gatt, Miriam
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Haeusler, Martin
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Klungsoyr, Kari
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Latos-Bielenska, Anna
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Lynch, Catherine
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McDonnell, Bob
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Nelen, Vera
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Neville, Amanda J.
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O’Mahony, Mary T.
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Rankin, Judith
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Rissmann, Anke
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Ritvanen, Annukka
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Rounding, Catherine
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Sipek, Antonin
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Tucker, David
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Verellen-Dumoulin, Christine
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Wellesley, Diana
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Dolk, Helen
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Khoshnood, Babak, Loane, Maria, Walle, Hermien de, Arriola, Larraitz, Addor, Marie-Claude, Barisic, Ingeborg, Beres, Judit, Bianchi, Fabrizio, Dias, Carlos, Draper, Elizabeth, Garne, Ester, Gatt, Miriam, Haeusler, Martin, Klungsoyr, Kari, Latos-Bielenska, Anna, Lynch, Catherine, McDonnell, Bob, Nelen, Vera, Neville, Amanda J., O’Mahony, Mary T., Queisser-Luft, Annette, Rankin, Judith, Rissmann, Anke, Ritvanen, Annukka, Rounding, Catherine, Sipek, Antonin, Tucker, David, Verellen-Dumoulin, Christine, Wellesley, Diana and Dolk, Helen (2015) Long term trends in prevalence of neural tube defects in Europe: population based study. British Medical Journal, 351, h5949. (doi:10.1136/bmj.h5949).

Record type: Article

Abstract

Study question: what are the long term trends in the total (live births, fetal deaths, and terminations of pregnancy for fetal anomaly) and live birth prevalence of neural tube defects (NTD) in Europe, where many countries have issued recommendations for folic acid supplementation but a policy for mandatory folic acid fortification of food does not exist?

Methods: this was a population based, observational study using data on 11?353 cases of NTD not associated with chromosomal anomalies, including 4162 cases of anencephaly and 5776 cases of spina bifida from 28 EUROCAT (European Surveillance of Congenital Anomalies) registries covering approximately 12.5 million births in 19 countries between 1991 and 2011. The main outcome measures were total and live birth prevalence of NTD, as well as anencephaly and spina bifida, with time trends analysed using random effects Poisson regression models to account for heterogeneities across registries and splines to model non-linear time trends.

Summary answer and limitations: overall, the pooled total prevalence of NTD during the study period was 9.1 per 10?000 births. Prevalence of NTD fluctuated slightly but without an obvious downward trend, with the final estimate of the pooled total prevalence of NTD in 2011 similar to that in 1991. Estimates from Poisson models that took registry heterogeneities into account showed an annual increase of 4% (prevalence ratio 1.04, 95% confidence interval 1.01 to 1.07) in 1995-99 and a decrease of 3% per year in 1999-2003 (0.97, 0.95 to 0.99), with stable rates thereafter. The trend patterns for anencephaly and spina bifida were similar, but neither anomaly decreased substantially over time. The live birth prevalence of NTD generally decreased, especially for anencephaly. Registration problems or other data artefacts cannot be excluded as a partial explanation of the observed trends (or lack thereof) in the prevalence of NTD.

What this study adds: in the absence of mandatory fortification, the prevalence of NTD has not decreased in Europe despite longstanding recommendations aimed at promoting peri-conceptional folic acid supplementation and existence of voluntary folic acid fortification

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Accepted/In Press date: 19 October 2015
Published date: 24 November 2015
Organisations: Human Development & Health

Identifiers

Local EPrints ID: 384440
URI: http://eprints.soton.ac.uk/id/eprint/384440
ISSN: 0959-8138
PURE UUID: 507060b5-56b4-4c5b-bdf4-73fcf7ac49c5

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Date deposited: 21 Dec 2015 12:33
Last modified: 14 Mar 2024 21:58

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Contributors

Author: Babak Khoshnood
Author: Maria Loane
Author: Hermien de Walle
Author: Larraitz Arriola
Author: Marie-Claude Addor
Author: Ingeborg Barisic
Author: Judit Beres
Author: Fabrizio Bianchi
Author: Carlos Dias
Author: Elizabeth Draper
Author: Ester Garne
Author: Miriam Gatt
Author: Martin Haeusler
Author: Kari Klungsoyr
Author: Anna Latos-Bielenska
Author: Catherine Lynch
Author: Bob McDonnell
Author: Vera Nelen
Author: Amanda J. Neville
Author: Mary T. O’Mahony
Author: Annette Queisser-Luft
Author: Judith Rankin
Author: Anke Rissmann
Author: Annukka Ritvanen
Author: Catherine Rounding
Author: Antonin Sipek
Author: David Tucker
Author: Christine Verellen-Dumoulin
Author: Diana Wellesley
Author: Helen Dolk

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