Case ReportTwo cases of bilateral bronchial artery varices: One with and one without bilateral coronary-to-pulmonary artery fistulas. Review and characterization of the clinical features of bronchial artery varices reported in Japan
Introduction
Babo et al.1 first reported bronchial artery varices (BAVs), so-called racemose arteriovenous angiomas, in 1976, which were defined as morphological abnormalities of the bronchial arteries, such as dilatation and tortuosity, with or without anastomoses to either pulmonary arteries or veins. In the United States and Europe, diverse terminology has been used to describe this anomaly, including angioma racemosum arteriovenosum,2 angioma arteriovenosum racemosum, pulmonary or bronchial arteriovenous malformation,3 or systemic to pulmonary vascular malformation, which occasionally includes fistulas of pulmonary arteries or veins. This diversity has led to difficulty in characterizing the clinical signs and symptoms of BAVs. Conversely, in Japan, since 1976, a new clinical entity called BAVs (racemose arteriovenous angioma) has been widely recognized, and 47 cases have been reported to date (Table 1), which enables its characteristic clinical findings to be identified. Herein, two cases of BAVs, one of which had bilateral coronary artery fistulas (CAFs) and the other of which had only BAV, are presented.
Section snippets
Case 1
A 63-year-old woman was referred to the emergency department because of sudden onset of haemosputum, haemoptysis, and dyspnoea. She had a history of surgery for right breast cancer 5 years earlier and had been treated for asthma and hypertension for the last 3 years. She had no clinical findings of recurrence. Her vital signs were normal except for hypoxaemia (94% with oxygen at 3 l/min via a nasal cannula). The admission chest radiograph (Fig 1a) showed a faint, abnormal shadow in the right
Discussion
An intensive examination of the 47 reported cases of BAVs in Japan (Table 1)6, 7, 8, 9, 10, 11, 12, 13, 14, 15, 16, 17, 18, 19 showed several characteristics.16, 17, 18, 19 The chief complaints were haemosputum and/or haemoptysis (n = 39, 83%). The incidence of B–P shunt was 70% (n = 28), with the pulmonary artery (n = 28, 70%) and both the artery and vein (n = 1, 0.02%). Endoscopic findings were a polypoid and/or bulging lesion (n = 35, 77.8%) and blood clots/bleeding (n = 8, 17.8%). The
Acknowledgements
The authors thank Mr Koyanagi M., Radiological Technologist (Department of radiology, Kyorin University hospital, Tokyo, Japan), for his useful advice on radiological assessment.
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