A rare and fatal respiratory disease: bronchial Dieulafoy’s disease

Liao, Shi-Xia; Sun, Pengpeng; Li, Bangguo; He, Shuang-Fei; Liu, Maomao; Ouyang, Yao

doi:10.1177/1753466620929236

Cited by 7 publications

(5 citation statements)

References 16 publications

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“…This vascular malformation was first reported in the gastrointestinal tract, which is also the most affected site, and the involvement of the bronchus is rare ( 2 ). The etiology and pathogenesis of BDD remain inconclusive and may be of a congenital nature ( 8 ). In addition, smoking, chronic airway inflammation, and iatrogenic injury also serve as possible contributory factors for hemorrhage ( 9 ).…”

Section: Discussionmentioning

confidence: 99%

Pediatric bronchial Dieulafoy’s disease with bronchial artery embolization: two case reports

Lai¹,

Lu²,

Xi³

et al. 2023

Transl Pediatr

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Background: Bronchial Dieulafoy's disease (BDD) is a vascular malformation characterized by the presence of a dysplastic artery in the bronchial submucosa. It is very rare in children but potentially fatal due to life-threatening hemoptysis.Case Description: An 8-year-old boy and a 6-year-old girl were referred to our hospital with recurrent moderate to massive hemoptysis. Intraluminal protrusions with a tendency to bleed were found by bronchoscopy in both patients. Computed tomography angiography (CTA) revealed an abnormal bronchial artery in one patient and a small intraluminal nodule with contrast enhancement in the other. An enlarged bronchial artery and bronchial-to-pulmonary fistulae corresponding to the lesion site were detected by bronchial arteriography in both patients. Based on the radiological findings, the diagnosis of BDD was established. Subsequent bronchial artery embolization (BAE) was successful, and no recurrence of hemoptysis was observed during the 15-to 18-month follow-up.Conclusions: Our cases highlighted the importance of considering BDD in the context of hemoptysis and endobronchial protrusion in children. Bronchial arteriography plays a critical role in diagnosis, especially in cases where CTA does not reveal vascular malformations. Early identification is essential as biopsy is contraindicated. BAE may be an appropriate treatment to improve the prognosis of children with BDD.

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Section: Discussionmentioning

confidence: 99%

Pediatric bronchial Dieulafoy’s disease with bronchial artery embolization: two case reports

Lai¹,

Lu²,

Xi³

et al. 2023

Transl Pediatr

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“…However, due to the risk of fatal bleeding, the necessity of pathological diagnosis remains controversial. In some cases, the diagnosis is based on the findings of bronchoscopy and bronchial angiography [ 6 , 21 , 22 , 27 , 33 , 37 , 42 , 47 , 51 , 53 ].…”

Section: Discussionmentioning

confidence: 99%

A case of bronchial Dieulafoy disease and literature review

Shi

Wang

et al. 2023

J Cardiothorac Surg

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Objective Bronchial Dieulafoy's disease (BDD) is a rare disease that causes massive hemoptysis. This paper reports a case of BDD treated surgically. At the same time, we summarize the data of BDD patients reported in domestic and foreign literature to improve the understanding, diagnosis and treatment of this disease. Methods A case of BDD with hemoptysis during bronchoscopy was reported. In addition, we searched for "bronchial Dieulafoy disease" through Pubmed, Web of Science, CNKI and Wanfang databases, covering the literature related to BDD that was definitely diagnosed or highly suspected from January 1995 to December 2021, and summarized the clinical characteristics, chest imaging, bronchoscopic manifestations, angiographic characteristics, pathological characteristics, treatment and outcome of patients. Results The patient was a 68 year old male. Tracheoscopy revealed nodular and mass like changes in the basal segment of the left lower lobe, which appeared massive hemorrhage when touching the surface. The computed tomography angiophy of the bronchial artery confirmed that the branches of the left bronchial artery were tortuous and dilated, and then the left lower lobe of the lung was resected. During the operation, 3 thick tortuous nutrient artery vessels were sent out from the descending aorta, and 1 thick tortuous nutrient artery was sent out from the autonomic arch. All of them were ligated and cut. The pathology after the operation was in accordance with BDD; The patient did not have hemoptysis after discharge and is still under follow-up. The database identified 65 articles from January 1995 to December 2021. After removing repeated reports, meetings, incomplete information and nursing literature, 60 articles were included to report 88 cases of BDD. BDD can occur at all ages, with a male to female ratio of about 1.6:1. It mainly starts with hemoptysis, and can also be seen due to cough, infection, and respiratory failure; Inflammatory changes such as pulmonary patch shadow, exudation shadow and ground glass shadow of pulmonary hemorrhage were more common in chest imaging; The diagnosis of BDD is mainly based on the bronchoscopy, bronchial angiography and pathological findings of surgical or autopsy specimens. Bronchoscopic findings were mostly non pulsating, smooth nodular or mucosal processes. Bronchial angiography mainly showed tortuous dilatation of bronchial artery, and the lesions were mainly located in the right bronchus, more from the bronchial artery; Diagnosis depends on pathology, showing submucosal expansion of bronchus or abnormal artery rupture and bleeding; 54 cases underwent selective bronchial artery embolization, 39 cases underwent pulmonary lobectomy, 66 cases improved, and 10 cases died (all of them were caused by massive hemorrhage during bronchoscopic biopsy). Conclusion BDD is rare, but may cause fatal massive hemoptysis. Bronchial angiography is considered to be an effective method to diagnose BDD. Since pathological biopsy may lead to fatal bleeding, the necessity of pathological diagnosis remains controversial. Interventional and surgical treatment plays an important role in patients with cough accompanied by massive hemoptysis.

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“…This classification refers to cryptogenic hemoptysis as hemoptysis without an underlying disease. This classification proposal requires further discussion, including the definition of cryptogenic hemoptysis bronchial Dieulafoy's disease [32][33][34] , which cannot be diagnosed without bronchoscopy, and bronchial aneurysm 35) , which cannot be diagnosed without CT angiography or angiography, which were listed independently.…”

Section: ) Major Classification Of Hemoptysis According To the Presen...mentioning

confidence: 99%

A Technical Manual of Bronchial Artery Embolization by Coil for Pulmonologists: An Expert's Opinion

Ishikawa,

Yamaguchi,

Nishihara

et al. 2023

Respiratory Endoscopy

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Hemoptysis is a symptom with a high mortality rate. The in-hospital mortality rate of hospitalized patients with hemoptysis is 5%-9%. Bronchial artery embolization was formerly positioned as an emergency treatment for massive hemoptysis. Nowadays, it has become a standard treatment for elective treatment for chronic recurrent hemoptysis. Despite the high mortality rate, only 2%-8% of hospitalized patients with hemoptysis underwent Bronchial Artery Embolization (BAE). One possible reason is the concern about spinal cord infarction, followed by the shortage of operators specializing in BAE. The incidence rate of spinal cord infarction was 0.18%, 0.71%, and 0.06% for gelatin sponge, n-butyl-2cyanoacrylate (NBCA), and coil, respectively, in a nationwide observational study in JAPAN. CIRSE (Cardiovascular and Interventional Radiological Society of Europe) guideline states that BAE by the coil is relatively safe and spinalprotective based on this study. BAE is primarily performed by radiologists and is a unique intervention targeting various arteries throughout the thorax, often smaller than 2 mm and, in some cases, less than 1 mm. The authors believe that it is essential to increase the number of pulmonologists subspecializing in BAE, like cardiologists and neurosurgeons who perform catheter treatment in their field. This paper aims to provide a technical and practical Expert Review of BAE by coil for pulmonologists based on our experience at a high-volume center managed by pulmonologists. The authors will also discuss a general narrative review on classifications of hemoptysis and BAE because we are concerned about the extreme international inconsistency in classifying and defining hemoptysis-related matters and will propose some challenging suggestions depending on our experience to rectify this situation.

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A rare and fatal respiratory disease: bronchial Dieulafoy’s disease

Cited by 7 publications

References 16 publications

Pediatric bronchial Dieulafoy’s disease with bronchial artery embolization: two case reports

Pediatric bronchial Dieulafoy’s disease with bronchial artery embolization: two case reports

A case of bronchial Dieulafoy disease and literature review

A Technical Manual of Bronchial Artery Embolization by Coil for Pulmonologists: An Expert's Opinion

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