Abstract
BackgroundPlastic 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 presentationWe 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.ConclusionOur 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.
Highlights
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
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
In this article we describe two monozygotic twins without underlying conditions suffering from respiratory distress following a common, human bocavirus 1 (HBoV1) positive respiratory tract infection
Summary
The presented cases are the first to describe two simultaneously affected monozygotic twins and substantiate the hypothesis of a contributing genetic factor in the pathophysiology of this disease. In this second report related to HBoV1, we show additional evidence that this condition can be triggered by a simple respiratory tract infection in previously healthy infants. Competing interests The authors declare that they have no competing interests. Authors’ contributions CMR was responsible for literature review, conception and preparation of the manuscript. WB and PI participated in preparation and critical revision of the manuscript. All authors read and approved the final manuscript
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