A case of bronchial Dieulafoy disease and literature review
Journal of Cardiothoracic Surgery volume 18, Article number: 197 (2023)
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.
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.
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).
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.
Dieulafoy's disease was first reported by French doctor Georges Dieulafoy  in 1898. It often occurs in the gastrointestinal tract, also known as gastric submucosal aneurysm or Dieulafoy's ulcer. Dieulafoy disease of bronchus is rare, which is characterized by the presence of dysplastic arteries in the submucosa of bronchus, leading to artery expansion, distortion and rupture, and is prone to fatal bleeding. Since Sweets  first reported it in 1995, less than 100 cases of BDD have been reported. In order to improve the understanding of BDD and avoid fatal hemoptysis, this article describes the clinical characteristics, diagnosis and treatment of BDD.
A 68 year old male smoking for 30 pack-year was admitted to the hospital on December 5, 2018 due to "left lung shadow was found for 4 months, and cough with left subcostal pain for 2 months". After admission, chest enhanced CT showed that there was a leaf like solid density increase shadow in the lower left lung with obvious enhancement, and the left lower lobe bronchus was partially occluded (Fig. 1). Tracheoscopy showed that there were nodular processes at the opening of the basal segment of the left lower lobe, with smooth surface and complete mucosa (Fig. 2). Local bleeding was seen on the surface of the node at the opening of the basal segment of the left lower lobe touched by the biopsy forceps, with the amount of 100 ml. After immediate intratracheal injection of hemocoagulase 2 IU, adrenaline 1 IU and intravenous drip of pituitrin 6 IU, the bleeding gradually decreased. The next day, only a small amount of dark red blood filaments were found in the sputum. The bronchial artery CTA showed that the anterior wall of the thoracic aorta sent out the left and right bronchial arteries from the level of the 5th and 6th thoracic vertebrae respectively. The branches of the left bronchial artery were tortuous and dilated (Figs. 3, 4, 5, 6, 7, 8 and 9). However, two days later, the patient coughed violently at night and had massive hemoptysis. The total amount of hemoptysis was about 500 ml within half an hour. After hemostasis with hemocoagulase 2 IU and pituitrin 12 IU, the amount of hemoptysis gradually decreased. After consultation with the thoracic surgeon, it was decided that because of the large amount of bleeding and the clear cause of the disease, the possibility of massive bleeding again after medical treatment was large, the emergency left lower lobe resection was performed immediately. The operation chosen to enter the chest cavity through the posterolateral incision of the left chest. Extensive adhesion was found in the chest cavity, which was carefully separated by ultrasonic knife The exploration found that the left lower lobe of the lung was consolidated and could not be re-expanded. The left lower pulmonary vein was thin. There were tortuous and thickened arterial vessels on the surface of the left main bronchus and the lower lobe bronchus. The abnormal vessels originated from the aortic arch (Fig. 10), and were cut off at the root of the vessels with linear cut stapler. First, cut off the lower lobe pulmonary vein with linear cut stapler, fully expose the lower lobe bronchus, and cut off with linear cut stapler. Continue to dissociate the lower lobe pulmonary artery and cut it with linear cut stapler. Cut along the gap between the upper and lower lobes with linear cut stapler, and remove the surgical specimen. The procedure went smoothly, approximately 400 ml of blood was lost, and no transfusion was given. The operation lasted three and a half hours. The patient was discharged after 1 week in hospital. Postoperative pathology: alveolar atrophy in the "lower lobe of left lung", bronchiole dilatation and bleeding, muscular vascular wall uneven thickness, congestion, interstitial fibrous tissue hyperplasia, chronic inflammatory cell infiltration; One lymph node in "Group 5", seven lymph nodes under "carina" showed reactive hyperplasia, and "bronchial artery in mediastinum" showed thick walled vessels with uneven wall thickness. No new acute hemoptysis was observed after discharge, and the patient was still being followed up at the time of writing this report.
Massive hemoptysis is a common critical disease in respiratory medicine. The common causes include bronchiectasis, pulmonary tuberculosis or lung tumor. BDD is one of the rare causes of massive hemoptysis. Since Sweerts  and others first reported BDD in 1995, nearly 100 cases have been reported, mostly cases. We reported a case of BDD. In addition, we searched the databases of Pubmed, Web of Science, CNKI and Wanfang for "bronchial Dieulafoy disease". 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, and 88 cases of BDD [2,3,4,5,6,7,8,9,10,11,12,13,14,15,16,17,18,19,20,21,22,23,24,25,26,27,28,29,30,31,32,33,34,35,36,37,38,39,40,41,42,43,44,45,46,47,48,49,50,51,52,53,54,55,56,57,58,59,60,61] were reported (Table 1).
There are 54 males and 34 females in 88 BDD patients, with a male to female ratio of 1.6:1. The minimum age of the patients is 9 months + 3 days, the maximum age is 85 years old, and the average age is 46.1 years old. All age groups have diseases, and about 1/3 of the patients are aged 60–75 years.
The etiology and pathogenesis of BDD remain to be clarified. At present, the possible mechanisms include: (1) congenital bronchial artery dysplasia ; (2) Chronic airway inflammation or injury ; (3) It is related to long-term heavy smoking ; (4) Acquired disease or normal vascular variation , which is easily misdiagnosed as bronchiectasis and other diseases in clinic. The 68 year old male patient we reported had a clear smoking history. In addition, 43.1% of the 88 patients were previously associated with other respiratory diseases. BDD may be related to chronic airway inflammatory injury or bronchial artery stretching and dilation caused by pulmonary tuberculosis [3, 6,7,8, 14, 20, 29, 33, 39, 42, 48], pneumonia [5, 12, 23, 24] and bronchiectasis [5, 16, 23, 27, 52, 57].
The most common symptom of BDD is repeated hemoptysis. It is reported that the maximum hemoptysis volume is 2000 ml . However, the clinical manifestations of the disease are not specific, and patients may seek medical advice due to cough [23, 29, 45, 57], infection [16, 24, 45] or respiratory failure [3, 41, 43, 46]. Liu Yanhong and others once reported that a BDD patient only presented with chest pain but no hemoptysis. Later, hemoptysis occurred after bronchoscopic biopsy and was definitely diagnosed by bronchial angiography . The patient we reported only presented with cough and left chest pain without hemoptysis. Therefore, if the patient has repeated massive hemoptysis of unknown causes, BDD should be considered; we also need to focus on patients with recurrent respiratory symptoms.
Auxiliary examinations of BDD include chest X-ray and computed tomography (CT), bronchoscopy and bronchial angiography. 36 patients received chest X-ray [2, 3, 6,7,8, 10,11,12,13,14,15,16,17, 19, 24, 30, 32, 33, 36, 39, 41,42,43, 49, 56, 60]. 78 patients received chest CT [2, 4, 5, 7,8,9,10,11,12, 14,15,16,17,18,19,20, 22,23,24,25,26,27,28,29,30,31,32,33,34,35,36,37, 39,40,41,42,43,44,45,46,47,48,49, 51,52,53,54,55,56,57,58,59,60]. The imaging manifestations are mainly inflammatory changes and ground glass shadows. Others include atelectasis, consolidation, bronchiectasis, nodules or masses or cavity in the bronchial, etc. (Table 2). The imaging findings of this patient reported here are mainly lung consolidation and atelectasis; Due to lack of specificity and sensitivity, it is not the first choice for diagnosis of BDD. 82 patients underwent bronchoscopy, of which 26 patients underwent bronchoscopic biopsy [3, 5, 8, 10,11,12, 15,16,17,18, 20, 23, 24, 29, 36,37,38, 45, 48, 49, 52, 57, 61], and 18 patients (22%) had severe hemoptysis complications [3, 5, 10, 16,17,18, 20, 23, 24, 29, 36, 37, 45, 48, 49, 52, 57, 61], and 10 patients died (12.2%) (Table 3) [3, 5, 16, 23, 24, 29, 45, 49, 52, 61]; Microscopically, the main manifestations were small (about 2–7 mm in diameter), no pulsatile, smooth nodules or mucosal protrusions, white caps on the surface, and some nodules may appear with active bleeding and blood clots in the bronchus; In some cases, the abnormal blood vessels in the submucosa present twisting and earthworm like dilation, sometimes presenting purple nodules . Because of intra bronchial hemorrhage and blood clots, it is difficult to find small mucosal protrusions, or the mucosal protrusions are located below the subsegmental bronchus and cannot be found through conventional bronchoscopy. At this time, bronchial angiography can show the rich blood supply of the corresponding part of the lesion , the distortion, expansion, deformation of the bronchial artery and the bronchopulmonary fistula, which is helpful for the diagnosis of BDD (Fig. 3). In this case, the patient suffered from bleeding without clamping under fiberoptic bronchoscope, indicating obvious vascular defects. Endobronchial ultrasound (EBUS) is a new diagnostic method, which can determine the nature of bronchial mucosal protrusion, mainly manifested as an echo free area of submucosal lesions. Doppler mode can detect blood flow signals . Nevertheless, the diagnosis still depends on pathology. The main pathological features of the disease are submucous bronchial artery expansion or abnormal artery rupture and bleeding . Twisted, dilated and deformed arteries form small nodules with a diameter of several millimeters that protrude from the lumen of the bronchi and cover the bronchial mucosa; In some cases, the diseased bronchi are surrounded by abundant blood vessels, and part of them invade the bronchial wall and directly reach the submucosa . The diagnosis of BDD is mainly based on the bronchoscopy, bronchial angiography and pathological findings of surgical or autopsy specimens. 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].
Summarizing the lesions of BDD, we found that there were 62 cases of right bronchus (3 cases of right main bronchus, 15 cases of right upper lobe, 20 cases of right middle lobe, 24 cases of right lower lobe, 6 cases of right middle bronchus); 25 cases of left bronchus (5 cases of left main bronchus, 10 cases of left upper lobe, 10 cases of left lower lobe); There were 5 cases of bilateral bronchi and 1 case near the carina (Table 4). To sum up, BDD usually occurs in the right bronchus, accounting for about two thirds of the total cases. It is more likely to occur in the right bronchus, which may be related to its anatomical structure. The risk of abnormality of the right bronchial artery is higher due to the diversity of embryonic development of the right bronchial artery, which is a congenital etiology . Therefore, for patients with hemoptysis of unknown cause, if bronchoscopy shows lesions similar to BDD and the lesions are located in the right bronchus, it is necessary to doubt whether there is abnormal development of bronchial artery, and biopsy should be avoided or careful to prevent massive hemoptysis. Most of the abnormal arteries originate from the bronchial artery system and a small part from the pulmonary artery .
The main treatment methods of BDD include conservative medical treatment, selective bronchial artery embolization (SBAE), lobectomy and argon plasma coagulation via bronchoscope. At present, SBAE is the preferred surgical method. For SABE or recurrent hemoptysis after embolization, lobectomy of the diseased lung is used. Eighty eight patients with BDD were summarized, including: (1) conservative drug therapy (9 cases); (2) Endobronchial intervention (7 cases); (3) Bronchoscopic arterial embolization only (30 cases); (4) Pulmonary lobectomy only (15 cases); (5) Bronchial artery embolism + pulmonary lobectomy (24 cases) (Table 5). Two patients were successfully treated with argon plasma coagulation through bronchoscope [28, 39]. One patient failed to receive cryotherapy, and then placed silica gel stent . 54 patients tried first-line selective embolization, and 24 patients received lobectomy due to unsuccessful embolization or prevention of bleeding. 66 patients were followed up for improvement without hemoptysis; Ten patients died (all of them were caused by massive hemorrhage during bronchoscopic biopsy). SBAE is usually used as the first-line method to treat hemoptysis, which is effective for some patients; The reported cases of intercostal artery embolism failure partially confirmed that the abnormal blood vessels originated from the pulmonary artery, and pulmonary lobectomy was necessary to control the bleeding.
In conclusion, the case we reported and the 88 cases we summarized support that BDD should be considered when recurrent and unexplained hemoptysis occurs and the lesion is limited to the right bronchus. Bronchial biopsy should be avoided and bronchial arteriography should be performed as early as possible. SBAE can be used for stable or intolerant patients to reduce the risk of life-threatening hemoptysis. Although it can retain some functions of the diseased lung, it is easy to recur after treatment. For uncontrollable cases or cases with poor prognosis, lobectomy can be the best choice to save lives. Although it will affect the quality of life of patients, it can eliminate the possibility of recurrence and obtain histopathological confirmation.
Availability of data and materials
The data that support the findings of this study are available from the corresponding author, Bing Li & Xuewei Zhao, upon reasonable request.
Dieulafoy G. Exulceratio simplex. L’intervention chirurgicale dans les hématémèses foudroyantes consécutives à l’exulcération simple del’estomac. Bull Acad Méd. 1898;39:49–84.
Sweerts M, Nicholson AG, Goldstraw P, et al. Dieulafoy’s disease of the bronchus. Thorax. 1995;50:697–8.
van der Werf TS, Timmer A, Zijlstra JG. Fatal haemorrhage from Dieulafoy’s disease of the bronchus. Thorax. 1999;54:184–5.
Stoopen E, Baquera-Heredia J, Cortes D, et al. Dieulafoy’s disease of the bronchus in association with a paravertebral neuri-lemoma. Chest. 2001;119:292–4.
Maxeiner H. Lethal hemoptysis caused by biopsy injury of an abnormal bronchial artery. Chest. 2001;119:1612–5.
Hope-Gill B, Prathibha BV. Bronchoscopic and angiographic findings in Dieulafoy’s disease of the bronchus. Hosp Med. 2002;63:178–9.
Bhatia P, Hendy MS, Li-Kam-Wa E, et al. Recurrent embolotherapy in Dieulafoy’s disease of the bronchus. Can Respir J. 2003;10:331–3.
Kuzucu A, Gürses I, Soysal O, et al. Dieulafoy’s disease: a cause of massive hemoptysis that is probably underdiagnosed. Ann Thorac Surg. 2005;80:1126–8.
Pomplun S, Sheaff MT. Dieulafoy’s disease of the bronchus: An uncommon entity. Histopathology. 2005;46:598–9.
Löschhorn C, Nierhoff N, Mayer R, et al. Dieulafoy’s disease of the lung: a potential disaster for the bronchoscopist. Respiration. 2006;73:562–5.
Xie BS, Chen YS, Lin MF, et al. Dieulafoy’s disease of the bronchus: a case report and review of the literature. Zhonghua Jie He He Hu Xi Za Zhi. 2006;29:801–3.
Rennert D, Gharagozloo F, Schwartz AM, et al. Dieulafoy’s lesion of the bronchus: report of a case and review of the literature. Pathol Case Rev. 2007;12:93–5.
Fields EL, De Keratry DR. Dieulafoy disease of the bronchus: case report and presentation of a novel therapeutic modality. J Bronchol Interv Pulmonol. 2008;15:107–9.
Parrot A, Antoine M, Khalil A, et al. Approach to diagnosis and pathological exami-nation in bronchial Dieulafoy disease: a case series. Respir Res. 2008;9:58.
Zhu JY, Chen G, Yin YP, et al. A case of bronchial Dieulafoy’s disease with massive hemoptysis. J Yunyang Med Coll. 2009;28:86–7.
D’Souza F, Sharma R. Dieulafoy’s disease of the bronchus. Pathology. 2010;42:683–4.
Ding D, Lu L, Shuai ZC, et al. A case of massive hemoptysis and asphyxia caused by Dieulafoy disease of bronchus biopsy and literature review. Int J Respir. 2010;30:1495–7.
Gurioli C, Casoni GL, Gurioli C, et al. Endobronchial ultrasound in Dieulafoy’s disease of the bronchus: an additional application of EBUS. Monaldi Arch Chest Dis. 2010;73:166–8.
Hu HY, Xin H, Xu EB, et al. A case of massive hemoptysis caused by bronchialDieulafoy disease. Chin J Respir Crit Care. 2010;9:544–5.
Wan W, Xia Y, Huang HD, et al. A case of Dieulafoy disease of bronchus and literature review. Int J Respir. 2011;31:919–22.
Barisione EE, Ferretti GG, Ravera SS, et al. Dieulafoy’s disease of the bronchus: a possible mistake. Multidiscip Respir Med. 2012;7:40.
Kolb T, Gilbert C, Fishman EK, et al. Pearse D, Feller-Kopman D and Yarmus L: Dieulafoy’s disease of the bronchus. Am J Respir Crit Care Med. 2012;186:1191.
Chen PP, Fang NX, Chen ZX. Bronchial mucosal biopsy leads to massive hemoptysis: a case report of bronchial artery abnormalities. Chin J Pract Intern Med. 2013;33:749–50.
Yang RH, Li JF, Liu J, et al. Dieulafoy disease of the bronchus: 3 cases report with literature review. Zhonghua Jie He He Hu Xi Za Zhi. 2013;36:577–80.
Fang Y, Wu QC, Wang B. Dieulafoy’s disease of the bronchus: report of a case and review of the literature. J Cardiothorac Surg. 2014;9:191.
Smith B, Hart D, Alam N. Dieulafoy’s disease of the bronchus: a rare cause of massive hemoptysis. Respirol Case Rep. 2014;2:55–6.
Liu YH, Li YL, Xing XQ, Yang YJ, Wu XW, et al. Diagnosis and treatment of Dieulafoy’s disease of the bronchus. China J Endosc. 2014;20:795–9.
Dalar L, Sökücü SN, Özdemir C. Endobronchial argon plasma coagulation for treatment of Dieulafoy disease. Respir Care. 2015;60:e11–3.
Wang WJ, Chang XH. A case of massive hemoptysis death caused by bronchial Dieulafoy disease bronchoscopy biopsy. Clin Misdiagn Misther. 2015;28:57–9.
Xia XD, Ye LP, Zhang WX, et al. Massive cryptogenic hemoptysis undergoing pulmonary resection: clinical and pathological characteristics and management. Int J Clin Exp Med. 2015;8:18130–6.
Ganganah O, LiangGuo S, Chiniah M, et al. Endobronchial ultrasound and bronchial artery embolization for Dieulafoy’s disease of the bronchus in a teenager: a case report. Respir Med Case Rep. 2015;16:20–3.
Padilla-Serrano A, Estrella-Palomares V, Martinez-Palacios B, et al. A case of massive hemoptysis related toa smoking-history: an acquired form of the Dieulafoy’s disease? Rev Port Pneumol. 2015;21:276–9.
Leng JW, Wang JG, Liu SF. A case of bronchial Dieulafoy disease. J Clin Pulmon Med. 2015;20:765–6.
Wang YF, Zeng YM. A case report of bronchial Dieulafoy disease and literature review. Int J Respir. 2015;35:1719–22.
Zhang JF, Ye J, Chen H, et al. A case of bronchial Dieulafoy disease: diagnosed by airway ultrasound. Chin J Rural Med Pharm. 2015;11:70–4.
Viola P, Villegas IA, Dusmet M, et al. Dieulafoy’s disease of the airways: a comprehensive review of a rare entity. Histopathology. 2016;69(5):886–90.
Ge T, Wu HC, Wang GA, et al. Two cases of bronchial Dieulafoy diseaseand literature review. Zhejiang Pract Med. 2016;21:453–6.
Hadjiphilippou S, Shah PL, Rice A, et al. Bronchial dieulafoy lesion. A 20-year history of unexplained hemoptysis. Am J Respir Crit Care Med. 2017;195:397.
Madan K, Dhungana A, Hadda V, et al. Flexible bronchoscopic argon plasma coagulation for management of massive hemoptysis in bronchial Dieulafoy’s disease. Lung India. 2017;34:99–101.
Niu HL, Yi P, Wang H, et al. Infantile Dieulafoy’s disease of bronchus: report of acase. Zhonghua Bing Li Xue ZaZhi. 2017;46:731–2.
Wadji MB, Farahzadi A. Dieulafoy’s disease of the bronchial tree: a case report. Sao Paulo Med J. 2017;135:396–400.
Yang D, Rong C, Gu J, et al. Dieulafoy disease of the trachea with recurrent episodes of massive hemoptysis: a case report. Medicine. 2017;96:e5855.
Bonnefoy V, Garnier M, Tavolaro S, et al. Bronchial Dieulafoy’s disease: Visualization of embolization particles in bronchial aspirate. Am J Respir Crit Care Med. 2018;198:954–5.
Mincholé E, Penin RM, Rosell A. The utility of linear endobronchial ultrasound for the incidental finding of Dieulafoy disease of the bronchus. J Bronchol Interv Pulmonol. 2018;25:e48–50.
Pan F, Wang F, Liu Z, et al. The computed tomography angiography features of Dieulafoy disease of the bronchu]. Zhonghua Jie He He Hu Xi Za Zhi. 2018;41:949–53.
Sheth HS, Maldonado F, Lentz RJ. Two cases of Dieulafoy lesions of the bronchus with novel comorbid associations and endo- bronchial ablative management. Medicine. 2018;97:8.
Wang F, Kuang TG, Wang J, et al. A rare cause of recurrent fatal hemoptysis: Dieulafoy’s disease of the bronchus. Chin Med J. 2018;131:2758–9.
Zhou JH, Yan XW, Liu RJ, et al. A case of misdiagnosis of bronchial Dieulafoy disease. Clin Misdiagn Misther. 2018;31:32–3.
Chen W, Chen P, Li X, et al. Clinical characteristics and treatments for bronchial Dieulafoy’s disease. Respir Med Case Rep. 2019;26:229–35.
Tang P, Wu T, Li C, et al. Dieulafoy disease of the bronchus involving bilateral arteries: a case report and literature review. Medicine. 2019;98:e17798.
White C, Ottaviano P, Munn N, et al. Massive hemoptysis due to recurrence of bronchial to pulmonary vascular malformation: a case report. Respir Med Case Rep. 2019;26:248–50.
Zhou P, Yu W, Chen K, et al. A case report and review of literature of Dieulafoy’s disease of bronchus: a rare life-threatening pathologic vascular condition. Medicine. 2019;98:8.
Liao SX, Sun PP, Li BG, et al. A rare and fatal respiratory disease: bronchial Dieulafoy’s disease. Ther Adv Respir Dis. 2020;14:1–5.
Yeh YT, Ramaswamy M, Shin J, et al. Bronchial Dieulafoy’s disease in children: a case report and review of literature. Front Pediatr. 2020;28(8):273.
Giordano M, Bigazzi MC, Palladino MT, et al. A rare cause of massive hemoptysis in a child: bronchial Dieulafoy’s disease-the first report of transcatheter treatment in pediatric age. Ann Thorac Med. 2020;15(4):244–6.
Woodhull S, Bush A, Tang AL, et al. Massive paediatric pulmonary haemorrhage in Dieulafoy’s disease: roles of CT angiography, embolisation and bronchoscopy. Paediatr Respir Rev. 2020;36:100–5.
Li X, Chen J, Yang S, et al. Experience and literature review of 1 case of massive hemoptysis after bronchial dieufay disease biops. J Rare Uncommon Dis. 2020;27(6):12–3.
Vijayasekaran D, Sivabalan S. Bronchial Dieulafoy disease with recurrent life-threatening hemoptysis. Indian Pediatr. 2021;8(3):287–8.
Ruthberg JS, Abrol A, Howard NS. Recurrent hemoptysis: a bronchial Dieulafoy’s lesion in a pediatric patient. Ann Otol Rhinol Laryngol. 2021;130(5):528–31.
Chen Y, Mao Y, Cheng X, et al. Case report: a case of infant bronchial Dieulafoy’s disease and article review. Front Pediatr. 2021;15(9):674509.
Li JT, Li LF, He GQ, et al. A case of death from hemorrhagic shock caused by bronchial Dieulafoy disease. Chin J Forensic Med. 2021;36(1):108–9.
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Shi, X., Wang, M., Wang, Y. et al. A case of bronchial Dieulafoy disease and literature review. J Cardiothorac Surg 18, 197 (2023). https://doi.org/10.1186/s13019-023-02279-1
- Dieulafoy disease of bronchus
- Vascular malformation
- Bronchial angiography
- Case report