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欧洲两个地区囊性纤维化的出生发病率时间趋势:来自新生儿筛查项目的数据。

Time trends in birth incidence of cystic fibrosis in two European areas: data from newborn screening programs.

作者信息

Scotet Virginie, Assael Baroukh M, Duguépéroux Ingrid, Tamanini Anna, Audrézet Marie-Pierre, Férec Claude, Castellani Carlo

机构信息

Inserm U613, Brest, France.

出版信息

J Pediatr. 2008 Jan;152(1):25-32. doi: 10.1016/j.jpeds.2007.07.046. Epub 2007 Oct 22.

DOI:10.1016/j.jpeds.2007.07.046
PMID:18154893
Abstract

OBJECTIVE

To determine the incidence of cystic fibrosis (CF) and its time trends over a 16-year period (1990 to 2005) in 2 European regions with a long history of newborn screening (NBS) for CF, and to investigate the impact of some external factors.

STUDY DESIGN

This study focused on data from NBS and prenatal diagnosis (PD) in Brittany (western France) and Veneto/Trentino Alto-Adige (northeastern Italy).

RESULTS

Similar birth incidences of CF were observed in the 2 regions (1/3153 vs 1/3540; P = .245). Time trend analysis using Poisson regression revealed that the birth incidence decreased significantly in the Italian area only (average annual percent change [AAPC] = -4.7%; 95% confidence interval [CI] = -7.3 to -2; P = .0008). The use of PD appeared more common in Brittany, and considering the terminations of CF-affected fetuses, the adjusted incidence was 1/2191 in Brittany and 1/3116 in Veneto/Trentino, corresponding to variations of 30.5% (highly significant; P = .0002) and 12% (not significant; P = .16), respectively. Recording the reason for each PD allowed ready assessment of the affect of various public health policies on incidence. The affect of population mixing also appeared to be relevant in the Italian area.

CONCLUSIONS

This study highlights how the incidence of CF has evolved in 2 European regions that have different attitudes toward PD and immigration policy.

摘要

目的

确定在两个有长期囊性纤维化(CF)新生儿筛查(NBS)历史的欧洲地区,16年期间(1990年至2005年)CF的发病率及其时间趋势,并调查一些外部因素的影响。

研究设计

本研究重点关注法国西部布列塔尼和意大利东北部威尼托/特伦蒂诺-上阿迪杰的NBS和产前诊断(PD)数据。

结果

在这两个地区观察到相似的CF出生发病率(1/3153对1/3540;P = 0.245)。使用泊松回归进行的时间趋势分析显示,仅在意大利地区出生发病率显著下降(平均年变化百分比[AAPC] = -4.7%;95%置信区间[CI] = -7.3至-2;P = 0.0008)。PD的使用在布列塔尼似乎更为常见,考虑到受CF影响胎儿的终止妊娠情况,调整后的发病率在布列塔尼为1/2191,在威尼托/特伦蒂诺为1/3116,分别对应30.5%(高度显著;P = 0.0002)和12%(不显著;P = 0.16)的变化。记录每次PD的原因有助于随时评估各种公共卫生政策对发病率的影响。人口混合的影响在意大利地区似乎也很重要。

结论

本研究突出了CF发病率在两个对PD和移民政策态度不同的欧洲地区是如何演变的。

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