Hamvas Aaron, Cole F Sessions, Nogee Lawrence M
Edward Mallinckrodt Department of Pediatrics, Washington University and St. Louis Children's Hospital, St. Louis, MO 63110, USA.
Neonatology. 2007;91(4):311-7. doi: 10.1159/000101347. Epub 2007 Jun 7.
Inherited disorders of pulmonary surfactant-associated proteins are rare but provide important insights into unique mechanisms of surfactant dysfunction. Recessive loss-of-function mutations in the surfactant protein-B and the ATP-binding cassette family member A3 (ABCA3) genes present as lethal surfactant deficiency in the newborn, whereas other recessive mutations in ABCA3 and dominant mutations in the surfactant protein-C gene result in interstitial lung disease in older infants and children. The molecular basis and the genetic and tissue-based approaches to the evaluation of children suspected of having one of these disorders are discussed.
遗传性肺表面活性物质相关蛋白疾病较为罕见,但能为深入了解表面活性物质功能障碍的独特机制提供重要线索。表面活性蛋白B和ATP结合盒家族成员A3(ABCA3)基因的隐性功能丧失突变在新生儿中表现为致死性表面活性物质缺乏,而ABCA3基因的其他隐性突变以及表面活性蛋白C基因的显性突变则会在大龄婴幼儿和儿童中导致间质性肺病。本文将讨论疑似患有这些疾病之一的儿童评估的分子基础以及基于基因和组织的方法。