Isolated bronchial hemangioma causing recurrent hemoptysis: A case report.

RATIONAL: The development of bronchial hemangioma in adults is rare, and massive hemoptysis due to diffuse vascular proliferation of bronchial hemangioma is fatal. PATIENT CONCERNS: A case of a 29-year-old woman kept massive hemoptysis even after being underwent repeated interventional embolization for recurrent massive hemoptysis. Eventually, the patient was performed the operation of right upper lung lobectomy and bronchial hemangioma with extracorporeal membrane oxygenation support and was followed up for 4 years without recurrent hemoptysis. DIAGNOSES: Bronchial hemangioma. CONCLUSION: For patients with bronchial angiomas bonded with bronchial artery-pulmonary arteriovenous fistulae, the early surgical resection is recommended if bronchial artery embolization (BAE) is considered ineffective.

Evaluación del tracto de salida del ventrículo derecho mediante resonancia magnética con flujo 4D en pacientes sometidos a recambio valvular pulmonar transcatéter / 4D flow magnetic resonance imaging to assess right ventricular outflow tract in patients undergoing transcatheter pulmonary valve replacement

Introducción y objetivos: La resonancia magnética (RM) con flujo 4D suele utilizarse para evaluar el ventrículo derecho y las arterias pulmonares antes del implante percutáneo de la válvula pulmonar (IPVP). Como el IPVP está limitado por el tamaño del tracto de salida del ventrículo derecho (TSVD), se necesita medirlo con precisión para planificar la intervención. El objetivo del presente estudio es comparar diferentes modalidades de RM con la medición invasiva del TSVD con balón.MétodosEstudio unicéntrico prospectivo de pacientes sometidos a IPVP por insuficiencia pulmonar aislada evaluada mediante RM con flujo 4D, precesión libre en estado estacionario/gradiente eco 3D (3D SSFP/GRE) y angiografía por RM con contraste. La medición con balón se consideró la referencia.ResultadosSe incluyó a 23 adultos (media de edad, 38,4±12,5 años). En 18 pacientes el IPVP fue exitoso. El diámetro medio del TSVD más pequeño fue de 25,4±4,3 mm medido con balón y 25,6±3,8 mm y 21,8±3,6 mm por RM con flujo 4D en sístole y diástole respectivamente. Comparados con los medidos con balón, los diámetros de TSVD se correlacionaron mejor cuando se estimaron mediante RM con flujo 4D sistólico (r=0,89; p<0,001) que mediante RM con flujo 4D diastólico (r=0,71; p<0,001), angiografía por R; 3D con contraste (r=0,73; p<0,001) o 3D SSFP/GRE (r=0,50; p=0,04), y no se correlacionaron de manera significativa cuando se realizaron en 2D en diástole o sístole. La diferencia media entre la RM con flujo 4D sistólico y la medición con balón fue de 0,2 mm (IC95%, –3,5 a 3,9 mm), en tanto que con las otras técnicas fue mayor.ConclusionesAdemás de la cuantificación de la insuficiencia de la válvula pulmonar, la RM con flujo 4D permite una estimación fiable de los diámetros del TSVD, sobre todo en sístole, lo que es fundamental antes de planificar el IPVP. (AU)

Introduction and objectives: Magnetic resonance imaging (MRI) including 4D flow is used before percutaneous pulmonary valve implantation (PPVI). As PPVI is limited by the size of the right ventricular outflow tract (RVOT), accurate sizing is needed to plan the intervention. The aim of this study was to compare different MRI modalities and invasive angiogram to balloon sizing of RVOT.MethodsSingle-centre prospective study of patients who underwent PPVI for isolated pulmonary regurgitation assessed by 4D flow MRI, 3D steady-state free precession/gradient echo (3D SSFP/GRE) and contrast magnetic resonance angiogram. Balloon sizing was considered as the reference.ResultsA total of 23 adults were included (mean age, 38.4±12.5 years). Eighteen patients underwent successful primary PPVI. The average of the narrowest RVOT diameter was 25.4±4.3mm by balloon sizing. Compared to balloon sizing, RVOT diameters were better correlated when estimated by systolic 4D flow MRI (r=0.89, P<0.001) than by diastolic 4D flow MRI (r=0.71, P<0.001), 3D contrast magnetic resonance angiography (r=0.73; P<.001) and 3D SSFP/GRE (r=0.50; P=.04) and not significantly correlated when estimated by 2D in diastole and systole. The mean difference between systolic 4D flow MRI and balloon sizing was 0.2mm (95%CI, –3.5 to 3.9 mm), whereas it was wider with other techniques.ConclusionsBeyond the quantification of pulmonary valve regurgitation, 4D flow allows accurate estimation of RVOT diameters, especially in systole, which is fundamental before planning PPVI. (AU)

A case of bronchial Dieulafoy disease and literature review.

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.

[Endovascular Treatment of a Ruptured Mediastinal Bronchial Artery Aneurysm].

Case 1:A 73-year-old man was transported to our hospital for evaluation of sudden onset of chest pain, back pain, and dyspnea. We initially misdiagnosed him with advanced esophageal cancer accompanied by mediastinal metastasis;however, subsequent multi-detector row computed tomography (MDR-CT) confirmed the diagnosis. We performed coil embolization of a bronchial artery aneurysm and thoracic endovascular aortic repair( TEVAR) to seal the root of the bronchial artery. Case 2:An 81-year-old woman with a one-week history of fever and cervical pain was diagnosed with a ruptured infected thoracic aneurysm. She underwent the same treatment as described in Case 1. Physicians should consider it as a differential diagnosis of mediastinal hematoma.

Two Cases of Bronchial Artery Racemose Hemangioma Successfully Treated with Bronchial Artery Embolization.

Rupture of a racemose hemangioma causing dilatation and tortuosity of the bronchial artery can result in massive bleeding and respiratory failure. Bronchial artery embolization (BAE) can treat this life-threatening condition, as we show in two cases. The first case was of an 89-year-old female complaining of sudden-onset chest and back pain. Bronchial artery angiography demonstrated a racemose hemangioma with a 2 cm aneurysm. The second case was of a 50-year-old male with hemoptysis and dyspnea, eventually requiring intubation. Bronchial arteriography showed a racemose hemangioma and a bronchial artery-pulmonary arterial fistula. BAE was successfully performed in both cases, with no recurrent hemorrhage. Therapeutic interventions in bronchial artery racemose hemangiomas include lobectomy or segmentectomy, bronchial arterial ligation, and BAE. BAE should be considered as first-line therapy for bleeding racemose hemangiomas of the bronchial artery because of its low risk of adverse effects on respiratory status, minimal invasiveness, and faster patient recovery.

[Racemose Hemangioma of the Bronchial Artery with Pulmonary Artery Fistula].

A 55-year-old woman was suspected of having hilar lymph node enlargement on a routine examination of the chest computed tomography( CT) scan at our hospital. On further examination, thoracic contrast CT and bronchial arteriography showed prominent dilation and meandering of the right bronchial artery with an aneurysm which formed a fistula to the pulmonary artery A7 at its distal end. Diagnosed as racemose hemangioma of bronchial artery with pulmonary artery fistula, we performed a surgical resection by open thoracotomy on the second day following bronchial artery embolization( BAE). No recurrence was observed in the five years of follow-up period after surgery. Therefore, surgical resection for asymptomatic racemose hemangioma of bronchial artery with bronchial artery-pulmonary artery fistula can be considered an effective method for the long-term prevention of bleeding.

[The bronchial arteries: a small but vital contribution to lung perfusion after lung transplantation]. / Die Bronchialarterien ­ ein kleiner, aber lebenswichtiger Beitrag zur Lungenperfusion nach Lungentransplantation.

Blood supply to the lungs is carried out by the pulmonary and bronchial-arterial system. The bronchial-arterial vessels are involved in supplying the small airways all the way up to the terminal bronchioles. The bronchial-arterial system is also necessary for the regulation of airway temperature, humidity and mucociliary clearance. Chronic ischaemia of the small airways due to damage or injury to bronchial arterial supply increases the risk of fibrosis of the small airways (bronchiolitis obliteration), especially in lung transplantation (LTx). Although survival after LTx has improved over time, it is, with a 5-year survival rate of only 50 to 60%, still significantly worse than that of other organ transplants. It is likely that bronchial arterial revascularisation at the time of LTx plays an important transplant-preserving function.

A Surgical Case of Bronchial Artery Aneurysm Connecting to a Pulmonary Artery and Vein Complicated by Racemose Hemangioma.

We report a surgical case of bronchial artery aneurysm (BAA) that directly connected to a pulmonary artery and a pulmonary vein through an abnormal vessel. It was complicated by racemose hemangioma. This is a rare vascular malformation. An 82-year-old female had a large BAA that was found incidentally. First, we consider treating the BAA with embolization by interventional radiology (IVR). However, because of strong meandering of the bronchial artery, we could not advance a microcatheter into the BAA. Therefore, a surgical operation was performed through a standard posterior lateral thoracotomy. The BAA was located between the upper and lower lobes and directly connected to the pulmonary artery. Some bronchial artery branches that provided inflow to the aneurysm were ligated, and the abnormal vessel that connected the BAA to the upper pulmonary vein was ligated easily. A fistula between the BAA and pulmonary artery was sutured by the cardiovascular surgeon using an artificial cardiopulmonary device, with permissive stenosis of A2b (ascending A2).

Effects of preserving the bronchial artery on cough after thoracoscopic lobectomy.

BACKGROUND: The purpose of this prospective study was to explore the influence of both preoperative three-dimensional (3D) reconstruction and intraoperative preservation of the bronchial artery (BA) on postoperative cough after thoracoscopic lobectomy. METHODS: A total of 60 patients who had received a combination of thoracoscopic lobectomy and systematic lymph node dissection were included in this study. They were divided into two groups, namely the BA preservation group (Group A), and conventional surgical treatment group (Group B). In group A, we used Exoview software for 3D reconstruction of the BA before the operation and the BA was preserved during the operation. 3D reconstruction of the BA was not performed before surgery in group B. The incidence of postoperative cough, the Mandarin Chinese version of the Leicester cough questionnaire (LCQ-MC), physiological, psychological and social dimensions and total score of the two groups were compared and analyzed. RESULTS: The scores and total scores of LCQ-MC in group B were lower than those in group A one and two months after surgery. There were significant differences between the two groups in physiological and psychological dimensions and total scores (p < 0.05), but there was no significant difference in social dimension between the two groups (p > 0.05). The incidence of postoperative cough in group A (16.7%) was lower than that in group B (30%), while the difference was not statistically significant (p = 0.222). CONCLUSIONS: Preoperative 3D reconstruction and intraoperative preservation of the BA can reduce the severity of postoperative cough.

Transarterial management of advance lung cancer.

Previous reports on transarterial treatment for lung cancer were reviewed. The bronchial arterial infusion therapy has a long history since 1964. Better local control with less doses of anti-neoplastic agents was warranted by trying transarterial administration to lung and mediastinal tumors. It is reported that both primary and metastatic tumors are fed by bronchial or other systemic arteries. The bronchial arterial embolization for hemoptysis has been introduced for clinical practice since 1973. Hemoptysis by not only benign but also malignant diseases has been well controlled by embolization. In recent decades, the technical elements for transarterial treatments have markedly improved. They make it possible to carry out precise procedures of selective catheter insertion to the tumor relating arteries. Current concepts of transarterial treatment, technical aspects and treatment outcomes are summarized. Tentative result from chemo-embolization for advanced lung cancer using recent catheter techniques was also described. It provides favorable local control and survival merits. It is considered that a population of lung cancer patients can benefit from transarterial management using small doses of anti-neoplastic agents, with less complications and less medical costs.

Thoracoscopic Bronchial Artery Resection for Multiple Bronchial Artery Aneurysms.

We describe a 36-year-old asymptomatic female with multiple bronchial artery aneurysms (BAAs) and a bronchial artery (BA) to pulmonary artery (PA) fistula. She was treated with thoracoscopic BA resection without lobectomy in lieu of catheter embolization as first-line treatment. The configuration of the BA and the location of the BAAs were clearly visualized using three-dimensional computed tomography (3DCT); therefore, the segment of the BA to resect was assessed preoperatively and complete resection of all BAAs was performed. Preoperative BA angiography delineated the BA to PA fistula, and guided surgical decision-making.

A Novel Technique for the Management of Massive Hemoptysis: The Customized Endobronchial Silicone Blocker.

OBJECTIVE: Massive hemoptysis (MH) has a high mortality rate. Therapeutic options include bronchoscopy for endobronchial lesions, bronchial artery embolization (BAE), and emergency surgery. Scant options exist for patients who are not candidates for these modalities. Culprit bronchial segment occlusion is an option to prevent "spillover flooding → hypoxia." Applying this concept, we describe a case series of MH control using a novel bronchoscopically inserted customized endobronchial silicone blocker (CESB). We analyzed the safety and efficacy of CESBs in a select subset of patients with MH. METHODS: Inclusion criteria were patients with MH who were unstable for surgery/BAE, failed BAE, or relatively contraindicated/refused BAE. CESBs were manufactured on-site by modifying silicone stents, inserted using rigid bronchoscopy and reinforced with glue. The CESB was removed after 6 weeks when possible. A successful outcome was defined as immediate bleeding control with no recurrence after removal. RESULTS: Over 4.5 years, 13 episodes of MH in 12 patients meeting the criteria specified earlier were treated with CESBs. Seven of 12 patients had tuberculosis, 4 active and 3 inactive. One patient had mucormycosis, 1 post-lobectomy, 1 endobronchial renal cell carcinoma, 1 fibrosing mediastinitis, and 1 patient had metastatic laryngeal Ca. Eight of 12 patients were taken for primary-CESB placement. Four of 12 patients were sent for BAE, which was unsuccessful, and rescue-CESB was done for definitive management. The success rate, as defined earlier, was 92.3%, with no deaths from MH. CONCLUSIONS: Innovative bronchoscopically inserted CESBs are an effective strategy in MH when patients are unstable or fail conventional management.

Open surgical correction of multiple bronchial artery aneurysms.

BACKGROUND: Bronchial artery aneurysms (BAAs) are a rare vascular entity. They can have various presentations ranging from an incidental finding on radiological examination to life-threatening hemoptysis. MATERIAL AND METHODS: We report the case of a 60-year-old woman with three posterior mediastinal BAAs who presented with unilateral periscapular pain, shortness of breath, hoarseness, and dysphagia. The BAAs were removed successfully via thoracotomy, with excellent recovery and relief of the periscapular pain. DISCUSSION AND CONCLUSION: We use this case as a platform to discuss the treatment options for BAAs.

A Ruptured Mediastinal Bronchial Artery Aneurysm Treated with Urgent Thoracic Endovascular Aortic Repair.

Bronchial artery aneurysms (BAA) are a rare but potentially life-threatening complications because of the massive hemothorax or hemoptysis that occurs with ruptures. A 79-year-old woman was transferred to our hospital because of the sudden onset of back pain, syncope, and subsequent hypotension. Computed tomography showed a left BAA with bilateral hemothorax and hemomediastinum. Transcatheter bronchial artery embolization failed because of the anatomical location, and she went into cardiopulmonary arrest. Cardiopulmonary resuscitation was performed with successful revival. Urgent thoracic endovascular aortic repair to cover the root of the left bronchial artery was successful, and she survived without any neurological deficits.

Localized Bronchial Hyperemia in Cases of Iatrogenic Hemoptysis: Clinical Presentations and Pathophysiological Mechanisms.

Hemoptysis is a frequently encountered symptom in many clinical settings, and etiologic diagnosis can sometimes prove challenging. Bronchoscopy may not promptly reveal the source or the cause of bleeding and few reports have focused so far on the abnormalities of bronchial mucosa vasculature that may unveil the underlying pathophysiology. In this special feature article, we present a series of cases presenting with hemoptysis after angiographic interventions in the thoracic vessels. Localized hyperemia and vascular dilatations in the bronchial mucosa observed during bronchoscopy as unique findings became clues enabling the correct diagnosis and management. We suggest the relevant pathophysiological mechanisms and discuss the available published experience on similar clinical entities.

Combined Management of a Bronchial Artery Fistula After Lung Transplantation.

A 46-year-old female patient exhibited massive endobronchial bleeding after dilation of a left bronchial anastomotic stenosis after lung transplantation, consistent with a bronchopulmonary artery fistula (BPAF). The BPAF was treated with a bronchial covered self-expandable metallic stent and percutaneous transcatheter pulmonary artery stent placement. BPAF is rare and leads to death in most cases because of massive hemoptysis. We describe a case of successful combined management of BPAF using both bronchial and pulmonary stent placement.

Dieulafoy disease of the bronchus involving bilateral arteries: A case report and literature review.

RATIONALE: Dieulafoy disease of the bronchus is a rare vascular deformity. To the best of our knowledge, reports of these involving both lung vascular are hitherto absent. PATIENT CONCERNS: A 67-year-old male was admitted to our department due to agnogenic hemoptysis. DIAGNOSES: Bronchoscopy was performed and some smooth, pulsatile nodular lesions were found in the middle and lower lobes, Computed tomography angiography of the bronchial artery confirmed a left bronchial artery arising from the aortic arch at T4 level, and both bronchial arteries were dilated and tortuous. INTERVENTIONS: Bronchial artery embolization was performed successfully. OUTCOMES: The patient was discharged with no hemoptysis. In addition, patient is under follow-up until today without any further incidents. LESSONS: This case reminds us that Dieulafoy disease of the bronchus could be a potential etiology for unexplained hemoptysis. The clinician should be aware of this disease when bronchoscopy revealed multiple some smooth, pulsatile nodular lesions, thereafter, bronchoscope biopsy should be avoided, as it could lead to fatal hemoptysis.

Potential Factors Affected Safety and Efficacy of Transcatheter Plug Closure for Pediatric Hemoptysis with Anomalous Bronchial Arteries.

OBJECTIVE: To evaluate the safety and efficacy of interventional care in pediatric hemoptysis for anomalous bronchial arteries (BAs) and to identify the potential factors resulting in hemoptysis recurrence. METHODS: 20 children complained of hemoptysis were diagnosed with anomalous BAs. All patients received transcatheter plug occlusion in Department of Cardiology, Children's Hospital of Chongqing Medical University. The safety and efficacy were evaluated according to clinical symptoms and images monitoring of enrolled subjects grouped as recurrence group and nonrecurrence group. The potential factors causing hemoptysis recurrence were reviewed and summarized. RESULTS: No deaths were recorded in a follow-up. Otherwise, hemoptysis recurrence was found in 8 subjects for 14 times, accounting for about 40%. Compared with nonrecurrence group, it indicated a statistical significance in hemoglobin levels (P=0.049), mycoplasma pneumonia particle assays (MP-PA) titers (P=0.030), and number of anomalous BAs (P=0.020). Meanwhile, 50% recurrent scenarios were associated with a respiratory infection by microbiological assessment before transcatheter plug occlusion. The repeat occlusion was applied for unclosed BAs leading to visual recurrent hemoptysis, the average interval time of which was 5.4 ± 3.6 mon. CONCLUSION: The data from this retrospective study have shown that transcatheter plug occlusion is a relatively safe procedure with a low mortality. The number of abnormal BAs has been identified as a highly significant predictor of recurrence, and the role of MP and other potential factors should be verified in a multicenter, larger sample size, and randomized controlled trial.