Wellcome Trust/Cancer Research UK Gurdon Institute, University of Cambridge, Cambridge, UK.
Pediatr Pulmonol. 2010 Oct;45(10):951-8. doi: 10.1002/ppul.21271.
It is a matter of debate as to what extent the long-term outcome of cystic fibrosis (CF) is affected by presenting with meconium ileus (MI). We compared long-term clinical outcomes of CF children who presented with MI, to those presenting with other symptoms (non-MI) in an era of non new-born-screening (NBS).We collected annual lung function data between the ages of 8-15 years in terms of percent predicted first second forced expired volume (FEV1%pr), percent predicted forced vital capacity (FVC%pr), and between the ages of 2-15 years annual height and weight Z-scores (HtZ and WtZ respectively) for children attending the Royal Brompton Hospital CF clinic. To be included in the study, subjects had to have at least five pulmonary function tests and five anthropometric measurements recorded over this period.Thirty-eight MI and 76 non-MI subjects were compared. There were no significant differences in genotype, sex, chronic Pseudomonas infection, or pancreatic enzyme use between the two groups. The median age of diagnosis was 1 day (MI) versus 7 months (non-MI). There was a decline in spirometry and anthropometric variables over the study period for both MI and non-MI groups apart from WtZ score in the non-MI group. Mixed model analysis adjusting for potential confounders including genotype, pancreatic status, sex, chronic Pseudomonas aeruginosa lung infection, and age of diagnosis revealed no difference between the two groups in terms of lung function and growth during the time period of the study, however there was a non-significant trend for subjects presenting with MI to do better in all four parameters.We conclude that babies presenting with MI have no worse long-term outcome than those presenting symptomatically later in infancy, despite having undergone invasive procedures in the newborn period. This underscores the importance of early diagnosis and treatment in CF.
MI 组和非 MI 组 CF 患儿的长期临床结局比较
在非新生儿筛查(NBS)时代,我们比较了以 MI 起病和以其他症状(非 MI)起病的 CF 患儿的长期临床结局。我们收集了在 Royal Brompton 医院 CF 诊所就诊的患儿在 8-15 岁时的年度肺功能数据,以第一秒用力呼气量占预计值的百分比(FEV1%pr)、用力肺活量占预计值的百分比(FVC%pr)表示,在 2-15 岁时以身高和体重 Z 评分(HtZ 和 WtZ 分别)表示。为了纳入研究,受试者在这段时间内必须至少有 5 次肺功能检查和 5 次人体测量记录。比较了 38 例 MI 患儿和 76 例非 MI 患儿。两组之间的基因型、性别、慢性假单胞菌感染或胰酶使用无显著差异。MI 组的中位诊断年龄为 1 天(MI),而非 MI 组为 7 个月(非 MI)。在研究期间,除了非 MI 组的 WtZ 评分外,MI 组和非 MI 组的肺功能和人体测量指标均呈下降趋势。调整基因型、胰腺状态、性别、慢性铜绿假单胞菌肺部感染和诊断年龄等潜在混杂因素的混合模型分析显示,在研究期间,两组在肺功能和生长方面无差异,但 MI 组的 4 项参数均有更好的趋势。我们得出结论,与症状较晚出现的婴儿相比,MI 组婴儿的长期结局并无恶化,尽管他们在新生儿期接受了有创治疗。这凸显了 CF 早期诊断和治疗的重要性。