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[卢卡周边地区中学生乳糜泻筛查]

[Screening for celiac disease in children attending secondary school around Lucca].

作者信息

Montesanti M, Domenici R, Matteucci L, Mei E

机构信息

Ospedale Campo di Marte,Regione Toscana-USL 6-Piana di Lucca.

出版信息

Minerva Pediatr. 1996 Nov;48(11):475-83.

PMID:9064494
Abstract

UNLABELLED

A growing number of clinical and epidemiological data point to the fact that coeliac disease (CD) is a pauci- or asymptomatic occurrence, relatively more frequent than it was supposed in the past, manifested in atypical, silent and latent forms which are often undiagnosed or diagnosed only at a later age. The fact that resolutive treatment is now available means that CD is an ideal field for the application of a screening method. In this context the study by Catassi et al. (1994) is particularly important since it reported a prevalence of 0.38% of CD in healthy children in the Pesaro-Urbino area.

AIM

The aim of this study was to verify the incidence of CD in a nonselected pediatric population in the province of Lucca, using the aforesaid screening method.

METHOD

The eligible population consisted of 1585 students from 5 secondary schools around Lucca, aged between 10 and 15 years old, none of whom were known to be affected by CD. In the first phase of the study anti-gliadin-IgA (AGA-IgA) and IgG antibodies were assayed in capillary blood (collected at school) using Alfa-Gliatest (Eurospital); children with AGA-IgA, AGA-IgG over 7 and 15 U/ml respectively were considered positive. In the second phase children with positive results underwent a further assay of AGA-IgA, AGA-IgG, anti-endomysium antibodies (EMA) and total IgA in venous blood. Lastly, children positive for AGA-IgA and/or EMA, or those positive for AGA-IgG with IgA deficit underwent duodenal jejunal biopsy.

RESULTS

41 children were positive on screening (2.6% of the eligible population, 3.8% of subjects effectively tested). Of these, 39 were assayed for AGA (IgA and IgG), EMA and total IgA in peripheral blood, identifying 4 subjects positive for AGA-IgA and EMA. Of the 4 children selected in this way, only 2 underwent jejunal biopsy and both presented "duodenal mucosa with chronic phlogosis and subtotal villous atrophy". Two cases of CD were formally ascertained with a prevalence of 1 out of 793 (0.13%) of the eligible population and an estimated prevalence of 1 out of 546.5 (0.18%) of the subjects undergoing screening. The cost was approximately Lit. 23,000 per child screened and approximately Lit. 6,100,000 for each coeliac child diagnosed.

COMMENTS AND CONCLUSIONS

The diagnostic iter proved efficacious and enabled 4 "high-risk" children to be selected. If the two subjects who did not undergo biopsy are also formally considered as coeliacs, the prevalence would be 1 out of 396 (0.25%) of the eligible subjects, namely 1 out of 273 (0.37%) of effectively tested subjects. This is a figure which is very similar to that reported by other studies. The 4 children identified here as strongly suspected of CD did not possess any anamnestic and/or objective elements which might have suggested "ex ante" a diagnosis of CD. If confirmed, these data provide concrete evidence of the need to perform mass screenings to identify CD. The economic convenience of this procedure depends on a careful analysis of the costs of the failure to diagnose CD.

摘要

未标注

越来越多的临床和流行病学数据表明,乳糜泻(CD)多为轻度或无症状,比过去认为的更为常见,常以非典型、隐匿和潜伏形式出现,往往未被诊断或仅在较晚年龄才被诊断出来。现在有了根治性治疗方法,这意味着CD是筛查方法应用的理想领域。在这种背景下,卡塔西等人(1994年)的研究尤为重要,因为该研究报告了佩萨罗 - 乌尔比诺地区健康儿童中CD的患病率为0.38%。

目的

本研究的目的是使用上述筛查方法,核实卢卡省非特定儿科人群中CD的发病率。

方法

符合条件的人群包括来自卢卡周围5所中学的1585名学生,年龄在10至15岁之间,均无已知的CD患者。在研究的第一阶段,使用阿尔法 - 麦醇溶蛋白检测试剂盒(欧罗斯皮塔尔公司)在学校采集的毛细血管血中检测抗麦醇溶蛋白IgA(AGA - IgA)和IgG抗体;AGA - IgA、AGA - IgG分别超过7和15 U/ml的儿童被视为阳性。在第二阶段,检测结果呈阳性的儿童在静脉血中进一步检测AGA - IgA、AGA - IgG、抗肌内膜抗体(EMA)和总IgA。最后,AGA - IgA和/或EMA呈阳性的儿童,或AGA - IgG呈阳性且有IgA缺乏的儿童接受十二指肠空肠活检。

结果

41名儿童筛查呈阳性(占符合条件人群的2.6%,实际检测对象的3.8%)。其中,39名儿童接受了外周血中AGA(IgA和IgG)、EMA和总IgA的检测,确定4名儿童AGA - IgA和EMA呈阳性。以这种方式挑选出的4名儿童中,只有2名接受了空肠活检,两人均表现为“十二指肠黏膜慢性炎症和绒毛部分萎缩”。正式确诊2例CD,符合条件人群的患病率为793人中1例(0.13%),接受筛查对象的估计患病率为546.5人中1例(0.18%)。筛查每个儿童的成本约为23,000里拉,诊断出每个乳糜泻儿童的成本约为6,100,000里拉。

评论与结论

诊断流程证明是有效的,能够挑选出4名“高危”儿童。如果未接受活检的两名儿童也被正式视为乳糜泻患者,符合条件对象的患病率将为396人中1例(0.25%),即实际检测对象中273人中1例(0.37%)。这一数字与其他研究报告的数字非常相似。这里确定的4名高度疑似CD的儿童没有任何病史和/或客观因素可能“事前”提示CD诊断。如果得到证实,这些数据为进行大规模筛查以识别CD提供了具体证据。该程序的经济可行性取决于对未诊断出CD的成本进行仔细分析。

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