Bronchial Dieulafoy’s disease: a retrospective analysis of 73 cases

Abstract

Background Bronchial Dieulafoy’s disease (BDD) is a rare disease that is known to be a cause of hemorrhage. The characteristics of this disease are still unknown. The present study describes the disorder based on a review of the world’s literature, emphasizing the diagnostic and therapeutic views. Methods A comprehensive research of BDD of the PubMed, Google Scholar, and Web of Science databases was performed. The following data were collected: patient characteristics; chest imaging, bronchoscopy, vascular angiography, and histopathologic examination findings; and treatment rendered. Results 73 cases of BDD have been reported from 1995 to 2019. Most of the cases occurred in Asia (52.1%), followed by Europe (31.5%). Chest imaging findings were non-specific. The main bronchoscopy finding was a nodular or protruding lesion (60.9%). 19 patients underwent bronchoscopic biopsies, 17 had bleeding, and 6 died. Four patients were successfully shown to have vascular malformations under mucosal protrusion by endobronchial ultrasound scan (EBUS). Vascular angiography mainly showed tortuous, dilated bronchial arteries. Vascular angiography mainly showed tortuous, dilated bronchial arteries. The arterial supply was mainly provided by bronchial arteries (48 cases) and the pulmonary circulation (4 cases). The lesions were mainly located in the right bronchus (53 cases). Selective bronchial artery embolization (BAE) was attempted in 38 patients and 20 patients underwent lobectomies. Emergency resection was performed in 15 patients, all of whom survived and had no recurrent hemoptysis. Conclusions Massive hemoptysis was the common manifestation of BDD. Vascular angiography and EBUS is a very useful examination before biopsy. BAE may be used in stable patients, or patients who cannot tolerate surgery, while surgical resection should be considered in patients who are unstable, patients with uncontrolled hemoptysis, or following BAE failure. Electronic supplementary material The online version of this article (10.1186/s12890-019-0863-1) contains supplementary material, which is available to authorized users.

Recurrent Hemoptysis: A Bronchial Dieulafoy’s Lesion in a Pediatric Patient

“…Angiography can be useful in revealing the course of the dilated, tortuous artery. 5 Bronchial DL should be considered in the setting of hemoptysis with normal CT or X-ray findings, especially when bleeding is localized to one lung. 9,10…”

A rare cause of massive hemoptysis in a child: Bronchial Dieulafoy's disease - the first report of transcatheter treatment in pediatric age

“…The mean age of diagnosis is 47 years, and the right lower lobe is the most affected. [ 3 4 5 ] Chronic inflammation of the airways, respiratory tract injury, and long-term smoking are considered triggers for the bleeding events. The diagnostic “gold standard” is the histopathological examination, which may demonstrate the arterial malformation in the bronchial submucosa.…”

“…[ 5 ] The angio-CT scan and the angiography are useful imaging tools for diagnosis with typical signs of disease: tortuous and dilated bronchial arteries with endobronchial bleeding. [ 3 4 ]…”

“…This “bronchial” Dieulafoy's disease is usually an adult disease exacerbated by smoking and hypertension. [ 3 4 ] We report a very rare case of bronchial Dieulafoy's disease in a child with recurrent and severe hemoptysis treated with a transcatheter embolization of the bleeding bronchial artery.…”

Dieulafoy’s Lesion of the Nasal Mucosa: A Case of Recurrent Epistaxis From Submucosal Arterial Malformation

“…1 While most cases present in the stomach or along the GI tract, there are reports of the lesion in extra-gastrointestinal locations such as the bronchus and head and neck. 2 In the head and neck, the pathology is conserved but is referred to as either cirsoid aneurysm (on the scalp) or submucosal arterial malformation. 3 DLs are a source of profound bleeding, and to our knowledge, this is the first report of a DL as a source of recurrent epistaxis.…”