EA7402, Laboratoire de Génétique de Maladies Rares (LGMR), University of Montpellier, Montpellier, France.
CRCM, Arnaud de Villeneuve Hospital, Montpellier, France.
PLoS One. 2020 Apr 17;15(4):e0231285. doi: 10.1371/journal.pone.0231285. eCollection 2020.
Cystic fibrosis (CF) is a rare genetic disease that affects the respiratory and digestive systems. Lung disease is variable among CF patients and associated with the development of comorbidities and chronic infections. The rate of lung function deterioration depends not only on the type of mutations in CFTR, the disease-causing gene, but also on modifier genes. In the present study, we aimed to identify genes and pathways that (i) contribute to the pathogenesis of cystic fibrosis and (ii) modulate the associated comorbidities. We profiled blood samples in CF patients and healthy controls and analyzed RNA-seq data with Weighted Gene Correlation Network Analysis (WGCNA). Interestingly, lung function, body mass index, the presence of diabetes, and chronic P. aeruginosa infections correlated with four modules of co-expressed genes. Detailed inspection of networks and hub genes pointed to cell adhesion, leukocyte trafficking and production of reactive oxygen species as central mechanisms in lung function decline and cystic fibrosis-related diabetes. Of note, we showed that blood is an informative surrogate tissue to study the contribution of inflammation to lung disease and diabetes in CF patients. Finally, we provided evidence that WGCNA is useful to analyze-omic datasets in rare genetic diseases as patient cohorts are inevitably small.
囊性纤维化(CF)是一种罕见的遗传疾病,影响呼吸系统和消化系统。CF 患者的肺部疾病各不相同,与合并症和慢性感染的发展有关。肺功能恶化的速度不仅取决于 CFTR(致病基因)突变的类型,还取决于修饰基因。在本研究中,我们旨在鉴定(i)有助于囊性纤维化发病机制和(ii)调节相关合并症的基因和途径。我们对 CF 患者和健康对照者的血液样本进行了分析,并使用加权基因相关网络分析(WGCNA)对 RNA-seq 数据进行了分析。有趣的是,肺功能、体重指数、糖尿病的存在和慢性绿脓杆菌感染与四个共表达基因模块相关。对网络和枢纽基因的详细检查表明,细胞黏附、白细胞迁移和活性氧的产生是肺功能下降和 CF 相关糖尿病的中心机制。值得注意的是,我们表明血液是一种信息丰富的替代组织,可用于研究炎症对 CF 患者肺部疾病和糖尿病的贡献。最后,我们提供了证据表明,WGCNA 可用于分析罕见遗传疾病的组学数据集,因为患者队列不可避免地较小。