[加利西亚新生儿筛查 20 年的评估与展望。计划成果]

IF 0.9 4区 医学 Q4 PUBLIC, ENVIRONMENTAL & OCCUPATIONAL HEALTH Revista Espanola De Salud Publica Pub Date : 2020-12-16
Paula Sánchez Pintos, José Ángel Cocho de Juan, M Dolores Bóveda Fontán, Daisy E Castiñeiras Ramos, Cristóbal Colón Mejeras, Agustin Javier Iglesias Rodríguez, María José de Castro López, José Ramón Alonso Fernández, José María Fraga Bermúdez, María Luz Couce Pico
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引用次数: 0

摘要

加利西亚新生儿筛查计划始于 1978 年,旨在早期发现内分泌和代谢疾病,2000 年 7 月,该计划采用了质谱技术,成为西班牙扩大新生儿筛查范围的先驱。该计划的首要目标是筛查 28 种疾病,其中包括建议进行 SNS 筛查的疾病,但镰状细胞性贫血除外,因为该疾病正处于纳入阶段。在其 20 年的历史中,共对 404 616 名新生儿(nb)进行了分析,确定了 547 个受所含疾病影响的病例,全球新生儿发病率为 1:739,筛查出的先天性代谢错误(IEM)发病率为 1:1.237(如果不包括良性高苯丙氨酸血症-HPA,则为 1:1.580nb),平均参与率为 99.35%,在分析期间逐步上升。在所筛查的病症中,先天性甲状腺功能减退症(1:2.211 nb)、胱氨酸尿症(1:4.129 nb)和高苯丙氨酸血症(1:5.699 nb)的发病率较高,其次是苯丙酮尿症和囊性纤维化(1:10,936 nb)。共发现66例假阳性(其中17例与母体病理有关)和5例假阴性,该计划的总体PPV和NPV分别为89.2%和99.99%,灵敏度为99.09%,特异性为99.98%。确诊的 CME 患者死亡率为 1.52%,其中有 11 例在筛查结果出来之前就出现了症状(2%)。有神经系统受累风险的 IEM 患者的智商在 95% 以上的病例中都是正常的。
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[Evaluation and perspective of 20 years of neonatal screening in Galicia. Program results.]

Galician newborn screening program for early detection of endocrine and metabolic diseases began in 1978 and was a pioneer in expanded newborn screening in Spain with the incorporation of mass spectrometry in July 2000. As a primary objective, 28 diseases are screened, including those recommended SNS except sickle cell anemia which is in the inclusion phase. In its 20-year history, 404,616 newborns (nb) have been analyzed, identifying 547 cases affected by the diseases included, with a global incidence of 1: 739 newborns and 1: 1.237 of the screened inborn errors of metabolism (IEM) (1:1.580 nb if excluding benign hyperphenylalaninemia-HPA), with an average participation of 99.35%, progressively higher during the analyzed period. Among the pathologies screened, congenital hypothyroidism (1:2.211 nb), cystinuria (1:4.129 nb) and HPA (1:5.699 nb), followed by phenylketonuria and cystic fibrosis (1:10,936 nb) stand out for their incidence. Sixty-six cases of false positives were identified (seventeen of them in relation to maternal pathology) and five false negatives, being the overall PPV and NPV of the program respectively of 89.2% and 99.99%, with a sensitivity of 99.09% and a specificity of 99.98%. The mortality rate of diagnosed CME patients is 1.52%, with eleven cases presenting symptoms prior to the screening result (2%). The intelligence quotient of IEM patients at risk of neurological involvement is normal in more than 95% of cases.

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来源期刊
Revista Espanola De Salud Publica
Revista Espanola De Salud Publica PUBLIC, ENVIRONMENTAL & OCCUPATIONAL HEALTH-
CiteScore
2.00
自引率
0.00%
发文量
106
审稿时长
12 weeks
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