Kurahara David, Morie Marina, Yamane Maya, Lam Sarah, Matthews Wallace, Yee Keolamau, Yamamoto Kara
Department of Pediatrics, John A. Burns School of Medicine, University of Hawaii, Honolulu, HI, USA.
Case Rep Pediatr. 2014;2014:876195. doi: 10.1155/2014/876195. Epub 2014 Sep 17.
We describe a possible association between pulmonary hemosiderosis (PH) and a history of bronchopulmonary dysplasia (BPD). Both patients were born at 28-week gestation and presented with PH at ages 22 months and 6 years, respectively. Both initially presented with cough and tachypnea, and bronchoalveolar lavage showed evidence of hemosiderin-laden macrophages. Initial hemoglobin levels were < 4 g/dL and chest radiographs showed diffuse infiltrates that cleared dramatically within days after initiation of intravenous corticosteroids. In the first case, frank pulmonary blood was observed upon initial intubation, prompting the need for high frequency ventilation, immediate corticosteroids, and antibiotics. The mechanical ventilation wean was made possible by the addition of mycophenolate mofetil (MMF) and hydroxychloroquine. Slow tapering off of medications was accomplished over 6 years. These cases represent a possible correlation between prematurity-associated BPD and PH. We present a review of the literature regarding this possible association. In addition, MMF proved to be life-saving in one of the PH cases, as it has been in pulmonary hemorrhage related to systemic lupus erythematosus. Further studies are warranted to investigate the possible association between PH and prematurity-related BPD, as well as the use of MMF in the treatment of PH.
我们描述了肺含铁血黄素沉着症(PH)与支气管肺发育不良(BPD)病史之间可能存在的关联。两名患者均在孕28周时出生,分别在22个月和6岁时出现PH。两人最初均表现为咳嗽和呼吸急促,支气管肺泡灌洗显示有含铁血黄素巨噬细胞的证据。初始血红蛋白水平<4g/dL,胸部X线片显示弥漫性浸润,在开始静脉注射皮质类固醇后数天内显著消退。在第一例中,初次插管时观察到明显的肺出血,因此需要高频通气、立即使用皮质类固醇和抗生素。通过添加霉酚酸酯(MMF)和羟氯喹,成功实现了机械通气的撤机。药物缓慢减量过程持续了6年。这些病例代表了早产相关BPD与PH之间可能存在的相关性。我们对有关这种可能关联的文献进行了综述。此外,MMF在其中一例PH病例中被证明挽救了生命,就像其在与系统性红斑狼疮相关的肺出血中所起的作用一样。有必要进一步研究PH与早产相关BPD之间的可能关联,以及MMF在PH治疗中的应用。