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巴西南部先天性肾上腺皮质增生症公共新生儿筛查项目中新生儿17-羟孕酮临界值测定的影响:3年经验

The impact of neonatal 17-hydroxyprogesterone cutoff determination in a public newborn screening program for congenital adrenal hyperplasia in Southern Brazil: 3 years' experience.

作者信息

Castro Simone Martins de, Wiest Paloma, Spritzer Poli Mara, Kopacek Cristiane

机构信息

Hospital Materno Infantil Presidente Vargas, Newborn Screening Referral Center, Porto Alegre, RS, Brazil.

Department of Analysis, Universidade Federal do Rio Grande do Sul (UFRGS), School of Pharmacy, Porto Alegre, RS, Brazil.

出版信息

Endocr Connect. 2023 Oct 30;12(12). doi: 10.1530/EC-23-0162. Print 2023 Dec 1.

Abstract

Congenital adrenal hyperplasia (CAH) occurs due to enzyme defects in adrenal steroidogenesis. The 21-hydroxylase deficiency accounts for 90-95% of cases, triggering accumulation of 17-hydroxyprogesterone (17-OHP). Early diagnosis through neonatal screening allows adequate treatment and reduced mortality. The purpose of the study was to determine 17-OHP cutoffs for the diagnosis of CAH in a public newborn screening program in Southern Brazil. A retrospective, descriptive, cross-sectional study was conducted to analyze 17-OHP levels in dried blood samples collected on filter paper of 317,745 newborns screened at a public newborn screening center from May 2014 to April 2017. Neonatal 17-OHP was measured in DBS samples using a time-resolved fluoroimmunoassay (GSP® kit 3305-0010; PerkinElmer). Different cutoffs were determined and stratified by birth weight. The incidence of CAH was 1:15,887 live births in the state of Rio Grande do Sul, with 20 cases of classical CAH diagnosed during the study period. Most newborns (80.73%) were white, and the prematurity rate was 9.8% in the study population. The combination of different percentiles, 98.5th for birth weight 2001-2500 g and 99.8th for the other birth weight groups, decreased false-positive results and increased specificity compared with current reference values to identify classical CAH cases. The local 17-OHP cutoffs determined were higher than those currently used by this screening program for all birth weight groups. The calculation of reference values from local population data and the combination of percentiles proved to be a valuable tool for proper diagnosis of CAH and reduction in the number of false positives.

摘要

先天性肾上腺皮质增生症(CAH)是由于肾上腺类固醇生成过程中的酶缺陷所致。21-羟化酶缺乏症占病例的90-95%,会引发17-羟孕酮(17-OHP)的蓄积。通过新生儿筛查进行早期诊断可实现充分治疗并降低死亡率。本研究的目的是确定巴西南部一项公共新生儿筛查项目中用于诊断CAH的17-OHP临界值。开展了一项回顾性、描述性横断面研究,以分析2014年5月至2017年4月在一家公共新生儿筛查中心接受筛查的317745名新生儿滤纸干血样本中的17-OHP水平。使用时间分辨荧光免疫分析法(GSP®试剂盒3305-0010;珀金埃尔默公司)对干血斑样本中的新生儿17-OHP进行检测。确定了不同的临界值,并按出生体重进行分层。在南里奥格兰德州,CAH的发病率为1:15887活产,在研究期间诊断出20例经典型CAH。大多数新生儿(80.73%)为白人,研究人群中的早产率为9.8%。与当前用于识别经典型CAH病例的参考值相比,对于出生体重2001-2500 g的新生儿采用第98.5百分位数,其他出生体重组采用第99.8百分位数的不同百分位数组合,减少了假阳性结果并提高了特异性。所确定的当地17-OHP临界值高于该筛查项目目前对所有出生体重组所使用的临界值。根据当地人群数据计算参考值并结合百分位数,被证明是正确诊断CAH和减少假阳性数量的宝贵工具。

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