Farrell Philip M, Rosenstein Beryl J, White Terry B, Accurso Frank J, Castellani Carlo, Cutting Garry R, Durie Peter R, Legrys Vicky A, Massie John, Parad Richard B, Rock Michael J, Campbell Preston W
Department of Pediatrics and Population Health Sciences, University of Wisconsin School of Medicine and Public Health, Madison, WI, USA.
J Pediatr. 2008 Aug;153(2):S4-S14. doi: 10.1016/j.jpeds.2008.05.005.
Newborn screening (NBS) for cystic fibrosis (CF) is increasingly being implemented and is soon likely to be in use throughout the United States, because early detection permits access to specialized medical care and improves outcomes. The diagnosis of CF is not always straightforward, however. The sweat chloride test remains the gold standard for CF diagnosis but does not always give a clear answer. Genotype analysis also does not always provide clarity; more than 1500 mutations have been identified in the CF transmembrane conductance regulator (CFTR) gene, not all of which result in CF. Harmful mutations in the gene can present as a spectrum of pathology ranging from sinusitis in adulthood to severe lung, pancreatic, or liver disease in infancy. Thus, CF identified postnatally must remain a clinical diagnosis. To provide guidance for the diagnosis of both infants with positive NBS results and older patients presenting with an indistinct clinical picture, the Cystic Fibrosis Foundation convened a meeting of experts in the field of CF diagnosis. Their recommendations, presented herein, involve a combination of clinical presentation, laboratory testing, and genetics to confirm a diagnosis of CF.
针对囊性纤维化(CF)的新生儿筛查(NBS)正在越来越多地实施,并且很快可能在美国全境使用,因为早期检测有助于获得专业医疗护理并改善治疗结果。然而,CF的诊断并非总是一目了然。汗液氯化物测试仍然是CF诊断的金标准,但并不总是能给出明确的答案。基因分型分析也并非总能提供清晰的结果;在囊性纤维化跨膜传导调节因子(CFTR)基因中已鉴定出1500多种突变,并非所有这些突变都会导致CF。该基因中的有害突变可表现为一系列病理状况,从成年后的鼻窦炎到婴儿期的严重肺部、胰腺或肝脏疾病。因此,出生后确诊的CF必须仍然是临床诊断。为了为NBS结果呈阳性的婴儿以及临床表现不明确的老年患者的诊断提供指导,囊性纤维化基金会召集了CF诊断领域的专家会议。本文介绍的他们的建议涉及临床表现、实验室检测和遗传学的综合运用,以确诊CF。