Kattan Abdulhakeim K, Bulagannawar Prakash S, Malik Iftikhar H
Department of Pediatrics, King Faisal Specialist Hospital and Research Centre, Riyadh, Kingdom of Saudi Arabia.
Saudi Med J. 2004 Oct;25(10):1474-7.
Pulmonary alveolar proteinosis is recently described as a rare cause of lung dysfunction and respiratory distress in term neonates. In several cases, a deficiency or insufficiency of surfactant protein B SP-B has been caused by a frame shift mutation in the gene encoding SP-B. Three siblings with congenital pulmonary alveolar proteinosis showed clinical and radiological evidence. Histopathological and immunohistochemical studies in the last sibling revealed deficiency of SP-B, one of the group of 3 specific lipoproteins that reduce the surface tension between air and liquid interface within pulmonary alveoli, suggesting a gene associated illness.
肺泡蛋白沉积症最近被描述为足月儿肺功能障碍和呼吸窘迫的罕见原因。在一些病例中,表面活性物质蛋白B(SP-B)的缺乏或不足是由编码SP-B的基因发生移码突变所致。三名患有先天性肺泡蛋白沉积症的兄弟姐妹有临床和影像学证据。对最后一名兄弟姐妹进行的组织病理学和免疫组化研究显示SP-B缺乏,SP-B是一组能降低肺泡内气液界面表面张力的3种特定脂蛋白之一,提示这是一种基因相关疾病。