Luo Yongfeng, Chen Hui, Ren Siying, Li Nan, Mishina Yuji, Shi Wei
Saban Research Institute, Children's Hospital Los Angeles, Los Angeles, California; Department of Surgery, Keck School of Medicine, University of Southern California, Los Angeles, California;
Saban Research Institute, Children's Hospital Los Angeles, Los Angeles, California; Department of Respiratory Medicine, The Second Xiangya Hospital, Central-South University, Changsha, Hunan, People's Republic of China;
Am J Physiol Lung Cell Mol Physiol. 2016 Jul 1;311(1):L29-38. doi: 10.1152/ajplung.00391.2015. Epub 2016 May 17.
Deficiency in pulmonary surfactant results in neonatal respiratory distress, and the known genetic mutations in key components of surfactant only account for a small number of cases. Therefore, determining the regulatory mechanisms of surfactant production and secretion, particularly during the transition from prenatal to neonatal stages, is essential for better understanding of the pathogenesis of human neonatal respiratory distress. We have observed significant increase of bone morphogenetic protein (BMP) signaling in neonatal mouse lungs immediately after birth. Using genetically manipulated mice, we then studied the relationship between BMP signaling and surfactant production in neonates. Blockade of endogenous BMP signaling by deleting Bmpr1a (Alk3) or Smad1 in embryonic day 18.5 in perinatal lung epithelial cells resulted in severe neonatal respiratory distress and death, accompanied by atelectasis in histopathology and significant reductions of surfactant protein B and C, as well as Abca3, whereas prenatal lung development was not significantly affected. We then identified a new BMP-Smad1 downstream target, Nfatc3, which is known as an important transcription activator for surfactant proteins and Abca3. Furthermore, activation of BMP signaling in cultured lung epithelial cells was able to promote endogenous Nfatc3 expression and also stimulate the activity of an Nfatc3 promoter that contains a Smad1-binding site. Therefore, our study suggests that the BMP-Alk3-Smad1-Nfatc3 regulatory loop plays an important role in enhancing surfactant production in neonates, possibly helping neonatal respiratory adaptation from prenatal amniotic fluid environment to neonatal air breathing.
肺表面活性物质缺乏会导致新生儿呼吸窘迫,而表面活性物质关键成分中已知的基因突变仅占少数病例。因此,确定表面活性物质产生和分泌的调节机制,特别是在从产前到新生儿阶段的转变过程中,对于更好地理解人类新生儿呼吸窘迫的发病机制至关重要。我们观察到出生后立即在新生小鼠肺中骨形态发生蛋白(BMP)信号显著增加。然后,我们使用基因操作小鼠研究了BMP信号与新生儿表面活性物质产生之间的关系。在围产期肺上皮细胞中于胚胎第18.5天删除Bmpr1a(Alk3)或Smad1来阻断内源性BMP信号,导致严重的新生儿呼吸窘迫和死亡,组织病理学显示伴有肺不张,表面活性物质蛋白B和C以及Abca3显著减少,而产前肺发育未受到显著影响。然后,我们确定了一个新的BMP-Smad1下游靶点Nfatc3,它是已知的表面活性物质蛋白和Abca3的重要转录激活因子。此外,在培养的肺上皮细胞中激活BMP信号能够促进内源性Nfatc3表达,并刺激包含Smad1结合位点的Nfatc3启动子的活性。因此,我们的研究表明,BMP-Alk3-Smad1-Nfatc3调节环在增强新生儿表面活性物质产生中起重要作用,可能有助于新生儿从产前羊水环境到新生儿空气呼吸的呼吸适应。