Montesanti M, Domenici R, Matteucci L, Mei E
Ospedale Campo di Marte,Regione Toscana-USL 6-Piana di Lucca.
Minerva Pediatr. 1996 Nov;48(11):475-83.
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.
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.
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.
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.
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.
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%。
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。
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。
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。
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。