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人类博卡病毒感染的同卵双胞胎同时发生肺不张:是塑型性支气管炎吗?

Simultaneous atelectasis in human bocavirus infected monozygotic twins: was it plastic bronchitis?

机构信息

Neonatal and Pediatric Intensive Care Unit, Graubuenden Cantonal Hospital, Chur, Switzerland.

出版信息

BMC Pediatr. 2013 Dec 18;13:209. doi: 10.1186/1471-2431-13-209.

Abstract

BACKGROUND

Plastic bronchitis is an extremely rare disease characterized by the formation of tracheobronchial airway casts, which are composed of a fibrinous exudate with rubber-like consistency and cause respiratory distress as a result of severe airflow obstruction. Bronchial casts may be associated with congenital and acquired cardiopathies, bronchopulmonary diseases leading to mucus hypersecretion, and pulmonary lymphatic abnormalities. In recent years, however, there is growing evidence that plastic bronchitis can also be triggered by common respiratory tract infections and thereby cause atelectasis even in otherwise healthy children.

CASE PRESENTATION

We report on 22-month-old monozygotic twins presenting with atelectasis triggered by a simple respiratory tract infection. The clinical, laboratory, and radiographic findings given, bronchial cast formation was suspected in both infants but could only be confirmed after bronchoscopy in the first case. Real-time polymerase chain reaction of the removed cast as well as nasal lavage fluid of both infants demonstrated strong positivity for human bocavirus.

CONCLUSION

Our case report is the first to describe two simultaneously affected monozygotic twins and substantiates the hypothesis of a contributing genetic factor in the pathophysiology of this disease. In this second report related to human bocavirus, we show additional evidence that this condition can be triggered by a simple respiratory tract infection in previously healthy infants.

摘要

背景

塑型性支气管炎是一种极为罕见的疾病,其特征是形成气管支气管气道铸型,这些铸型由具有橡胶状一致性的纤维渗出物组成,并由于严重的气流阻塞而导致呼吸窘迫。支气管铸型可能与先天性和获得性心脏病、导致黏液过度分泌的支气管肺部疾病以及肺淋巴异常有关。然而,近年来越来越多的证据表明,塑型性支气管炎也可能由常见的呼吸道感染引发,从而导致即使在其他方面健康的儿童中也发生肺不张。

病例介绍

我们报告了一对 22 个月大的同卵双胞胎,因简单的呼吸道感染引发肺不张。根据临床表现、实验室检查和影像学结果,怀疑两名婴儿均出现支气管铸型形成,但仅在第一例婴儿中进行支气管镜检查后才能确诊。对取出的铸型以及两名婴儿的鼻洗液进行实时聚合酶链反应检测,均显示人博卡病毒呈强阳性。

结论

我们的病例报告是首例描述同时受影响的同卵双胞胎的报告,并证实了遗传因素在这种疾病发病机制中的作用。在与人类博卡病毒相关的第二份报告中,我们提供了更多证据表明,这种情况可能由先前健康的婴儿的简单呼吸道感染引发。

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