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

被引：156

作者：

Khoshnood, Babak ^{[1
]}

Loane, Maria ^{[2
]}

de Walle, Hermien ^{[3
]}

Arriola, Larraitz ^{[4
]}

Addor, Marie-Claude ^{[5
]}

Barisic, Ingeborg ^{[6
]}

Beres, Judit ^{[7
]}

Bianchi, Fabrizio ^{[8
]}

Dias, Carlos ^{[9
]}

Draper, Elizabeth ^{[10
]}

Garne, Ester ^{[11
]}

Gatt, Miriam ^{[12
]}

Haeusler, Martin ^{[13
]}

Klungsoyr, Kari ^{[14
,15
]}

Latos-Bielenska, Anna ^{[16
]}

Lynch, Catherine ^{[17
]}

McDonnell, Bob ^{[18
]}

Nelen, Vera ^{[19
]}

Neville, Amanda J. ^{[20
]}

O'Mahony, Mary T. ^{[21
]}

Queisser-Luft, Annette ^{[22
]}

Rankin, Judith ^{[23
]}

Rissmann, Anke ^{[24
]}

Ritvanen, Annukka ^{[25
]}

Rounding, Catherine ^{[26
]}

Sipek, Antonin ^{[27
]}

Tucker, David ^{[28
]}

Verellen-Dumoulin, Christine ^{[29
]}

Wellesley, Diana ^{[30
,31
]}

Dolk, Helen ^{[2
]}

机构：

[1] Maternite Port Royal, Ctr Biostat & Epidemiol, Obstet Perinatal & Pediat Epidemiol Res Team, INSERM U1153, F-75014 Paris, France

[2] Univ Ulster, Inst Nursing Res, Ctr Maternal Fetal & Infant Res, EUROCAT Cent Registry, Newtownabbey, North Ireland

[3] Univ Groningen, Univ Med Ctr Groningen, Dept Genet, EUROCAT Northern Netherlands Registry, Groningen, Netherlands

[4] CIBER Epidemiol & Salud Publ CIBERESP, Inst BIO Donostia Basque Govt, Publ Hlth Div Gipuzkoa, San Sebatian, Spain

[5] CHU Vaudois, Serv Genet Med Maternite, Lausanne, Switzerland

[6] Childrens Univ Hosp Zagreb, Clin Hosp Sisters Mercy, Zagreb, Croatia

[7] Dept Hungarian Congenital Abnormal Registry & Sur, Natl Inst Hlth Dev, Budapest, Hungary

[8] Gabrielle Monasterio Fdn, CNR Inst Clin Physiol, Pisa, Italy

[9] Inst Nacl Saude Dr Ricardo Jorge, Lisbon, Portugal

[10] Univ Leicester, Dept Hlth Sci, Leicester, Leics, England

[11] Hosp Lillebaelt, Kolding, Denmark

[12] Dept Hlth Informat & Res, Guardamangia, Malta

[13] Med Univ Graz, Graz, Austria

[14] Univ Bergen, Norwegian Inst Publ Hlth, Med Birth Registry Norway, Bergen, Norway

[15] Univ Bergen, Dept Global Publ Hlth & Primary Care, Bergen, Norway

[16] Univ Med Sci, Dept Med Genet, Poznan, Poland

[17] HSE South, Dept Publ Hlth, Lacken, Kilkenny, Ireland

[18] Hlth Serv Execut, Dublin, Ireland

[19] Prov Inst Hyg, Antwerp, Belgium

[20] Univ Ferrara, Ctr Clin & Epidemiol Res, Registro IMER IMER Registry Emilia Romagna Regist, I-44100 Ferrara, Italy

[21] Hlth Serv Execut South, Dept Publ Hlth, Dublin, Ireland

[22] Johannes Gutenberg Univ Mainz, Childrens Hosp, Univ Med Ctr, Birth Registry Mainz Model, D-55122 Mainz, Germany

[23] Newcastle Univ, Inst Hlth & Soc, Newcastle Upon Tyne NE1 7RU, Tyne & Wear, England

[24] Univ Magdeburg, Fac Med, Malformat Monitoring Ctr Saxony Anhalt, D-39106 Magdeburg, Germany

[25] Natl Inst Hlth & Welf, Helsinki, Finland

[26] Univ Oxford, Natl Perinatal Epidemiol Unit, Oxford, England

[27] Thomayer Univ Hosp, Dept Med Genet, Natl Registry Congenital Anomalies Czech Republ, Prague, Czech Republic

[28] Publ Hlth Wales, Swansea, W Glam, Wales

[29] Inst Rech Sci Pathol & Genet, Ctr Human Genet, Charleroi, Belgium

[30] Southampton Univ Hosp, Fac Med, Southampton, Hants, England

[31] Wessex Clin Genet Serv, Southampton, Hants, England

来源：

BMJ-BRITISH MEDICAL JOURNAL | 2015年 / 351卷

关键词：

FOLIC-ACID FORTIFICATION; CONGENITAL-MALFORMATIONS; FOOD FORTIFICATION; SPINA-BIFIDA; PREGNANCY; ANOMALIES; EXPOSURE; IMPACT;

D O I：

10.1136/bmj.h5949

中图分类号：

R5 [内科学];

学科分类号：

1002 ; 100201 ;

摘要：

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.

引用

页数：6

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