Role of computed tomography angiography in the evaluation of haemoptysis in children: Decoding the abnormal vessels.

Background & objectives: Haemoptysis in children is potentially life-threatening. In most cases, the bleeding arises from the systemic circulation, and in 5-10 per cent of cases, it arises from the pulmonary circulation. The role of computed tomography angiography (CTA) in this setting is important. This study was undertaken (i) to study the role of single-phase split-bolus dual energy contrast-enhanced multidetector row CTA (DECTA) in the evaluation of haemoptysis in children; (ii) to analyze the patterns of abnormal vascular supply in the various aetiologies encountered. Methods: A retrospective study of 86 patients who underwent split bolus DECTA for the evaluation of haemoptysis was performed. Final diagnoses were categorized as normal computed tomography, active tuberculosis (TB), post-infectious sequelae, non-TB active infection, cystic fibrosis (CF), non-CF bronchiectasis, congenital heart disease (CHD), interstitial lung disease, vasculitis, pulmonary thromboembolism and idiopathic pulmonary haemosiderosis. Abnormal bronchial arteries (BAs) and non-bronchial systemic collateral arteries (NBSCs) were assessed for number and site and their correlation with underlying aetiologies. Results: A total of 86 patients (45 males, age from 0.3 to 18 yr, mean 13.88 yr) were included in the study; among these only two patients were less than five years of age. The most common cause of haemoptysis was active infection (n=30), followed by bronchiectasis (n=18), post-infectious sequelae (n=17) and CHD (n=7). One hundred and sixty five abnormal arteries were identified (108 BA and 57 NBSC), and were more marked in bronchiectasis group. Interpretation & conclusions: Active infections and bronchiectasis are the most common causes of haemoptysis in children. While post-infectious sequelae are less common, in patients with haemoptysis, the presence of any abnormal arteries correlates with a more frequent diagnosis of bronchiectasis. NBSCs are more common in post-infectious sequelae and CHD.

Cryptogenic massive hemoptysis caused by bronchial artery-pulmonary artery fistula in a 12-year-old boy: A case report and literature review.

BACKGROUND: Hemoptysis is a frequently encountered symptom of the respiratory system in adult but is rare in children. Bronchial artery-pulmonary artery fistula (BPF) is one of the most important and life-threatening cause in pediatric hemoptysis patients. Although the severity of BPF has been proved in previous studies, details about clinical diagnosis and treatment of BPF in children have been rarely reported. CASE PREPARATION: A 12-year-old boy presented to the hospital with hematemesis after coughing, without any other symptoms. After admission, he had repeated hemoptysis, 20-30 ml each time, and on the 11th night of admission a massive hemoptysis (about 100 ml bright red blood) occurred suddenly. Chest computed tomography demonstrated patchy ground glass opacities in the right lung, suggestive of pulmonary hemorrhage. Bronchial arteriography showed an apparent BPF in the right lobe bronchial artery. Therefore, bronchial artery embolization was performed, following which a thrombus in the bronchial lumen was removed by bronchoscopy. After these interventions, the patient recovered quickly and no recurrence was noted in the following year. CONCLUSION: We believe that this case should raise awareness of cryptogenic massive hemoptysis caused by BPF. In the event of hemoptysis in a child, it is important to clarify the source of the bleeding. If common etiologies have been excluded, the presence of pulmonary and bronchial vascular malformations should be considered. Moreover, multidisciplinary collaboration is crucial in the diagnosis and management of cryptogenic hemoptysis.

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).

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

OBJECTIVE: This paper presents a case of a bronchial Dieulafoy's lesion in a pediatric patient with recurrent hemoptysis. CASE REPORT: A 11-year old female presented multiple times with dry cough and hemoptysis to an outside hospital, each time leading to a diagnosis of epistaxis and subsequent discharge. When she arrived to our tertiary center with heavy hemoptysis and no evidence of epistaxis, the patient was urgently taken to the operating room by both the otolaryngology and pediatric pulmonology services. Active bleeding from a Dieulafoy's lesion on the right lower bronchus was found and selective embolization of two tortuous arteries was subsequently performed. The patient was discharged in stable condition without recurrence of hemoptysis over the last two months. CONCLUSION: While rare, especially in pediatric patients, bronchial Dieulafoy's lesions may cause severe hemoptysis and should be considered in the differential diagnosis when the etiology for hemoptysis is unclear.

Massive hemoptysis due to anastomosis between the left phrenic artery and pulmonary arteries/veins.

Massive hemoptysis is a serious medical condition or emergency which needs immediate treatment. It typically appears in the bronchial arteries and can be caused by a wide range of pulmonary diseases. This report is based on a very rare case of a patient bleeding from an anastomosis between the left phrenic artery and pulmonary arteries/veins. In this case, the chest computed tomography had detected changes which required doctors to perform a successful embolization.

Detection of Shunting Into Pulmonary Artery on Multidetector Row Computed Tomography Arteriography Before Bronchial Arterial Embolization: A Preliminary Study.

OBJECTIVE: The aim of this study was to investigate the diagnostic performance of detecting systemic arterial pulmonary circulation shunts on multidetector row computed tomography arteriography (MDCTA). METHODS: Thirty-five consecutive bronchial artery embolization sessions with preprocedural MDCTA were performed for 32 patients and 35 sessions. The MDCTA studies with computed tomography value of pulmonary trunk visually lower than that of ascending aorta were defined as "diagnostic MDCTA." Angiographic studies and "diagnostic MDCTA" were evaluated, respectively, for shunting into pulmonary artery. Based on the results of angiographic studies, diagnostic performance of "diagnostic MDCTA" was evaluated. RESULTS: The rate of diagnostic MDCTA was 63% (23 of 35). On "diagnostic MDCTA," sensitivity, specificity, and positive and negative predictive values for detecting shunts were 83% 100%, 100%, 94%, respectively. CONCLUSIONS: Systemic arterial pulmonary circulation shunts were detected on "diagnostic MDCTA" with high sensitivity and specificity.

Value of multidetector computed tomography angiography before bronchial artery embolization in hemoptysis management and early recurrence prediction: a prospective study.

BACKGROUND: Multidetector computed tomography (MDCT) angiography is a useful examination to detect the source of the bleeding in patients with hemoptysis. The aim of the study was to prospectively evaluate the role and clinical efficacy of MDCT angiography before bronchial artery embolization (BAE) for the management of hemoptysis, and to investigate the predictors of early recurrence. METHODS: It is a double-center study which included 57 hemoptysis patients undergoing MDCT angiography prior to BAE from August 2019 to July 2020. A prospective analysis of culprit arteries detected by MDCT angiography allowed an evaluation of the role of this technique. A follow-up was done to assess the efficacy of BAE with preprocedural MDCT angiography and to explore the risk factors of early recurrent hemoptysis. RESULTS: The accuracy of MDCT angiography in the identification of culprit arteries was as high as 97.5%. The average number of total culprit arteries per patient was 2.75 ± 1.73. Among which, the average numbers of culprit ectopic bronchial arteries (BAs) and non-bronchial systemic arteries (NBSAs) per patient were 0.21 ± 0.41 and 1.04 ± 1.57, respectively. The immediate clinical success rate, total hemoptysis recurrence rate, and early hemoptysis recurrence rate of BAE following MDCT angiography were 94.7, 18.5, 16.7%, respectively. Aspergilloma (HR = 6.63, 95% CI: 1.31-33.60, p = 0.022) was associated with an increase in the risk of early recurrence. CONCLUSIONS: MDCT angiography should be performed before BAE for the management of hemoptysis. Aspergilloma was an independent predictor for early recurrence.

Massive paediatric pulmonary haemorrhage in Dieulafoy's disease: Roles of CT angiography, embolisation and bronchoscopy.

Acute, major pulmonary haemorrhage in children, is rare, may be life-threatening and at times presents atypically. Dieulafoy's disease of the bronchus presenting with recurrent or massive hemoptysis was first described in adults. Prior to reviewing the literature, we report an illustrative case of bronchial Dieulafoy's disease (BDD) in a child presenting unusually with massive apparent hematemesis. The source of bleeding is a bronchial artery that fails to taper as it terminates within the bronchial submucosa. A high index of suspicion is required to identify such lesions via radiological imaging and the role of bronchial artery embolisation is highlighted with video images of angiography included.

A rare and fatal respiratory disease: bronchial Dieulafoy's disease.

A 66-year-old woman had two severe episodes of massive hemoptysis without any premonitory symptoms, with approximately 400-500 ml blood each time. Bronchoscopic exam revealed a smooth and pulsatile protrusion that was approximately 8-10 mm in diameter found at the beginning of the right middle lobe bronchus in the bronchial lumen. The protrusion arose from the surface with absolutely normal mucosa. Selective bronchial arteriography showed that elongated, tortuous, and dilated branches of the bronchial artery in the region of the middle lobe bronchus. Further bronchial arterial embolization (BAE) is recommended, although the patient currently has no active bleeding. Bronchial Dieulafoy's disease (BDD) is a rare and life-threatening disease. Selective bronchial arteriography is a diagnostic tool to detect and locate abnormal arteries. There is no unified guideline or expert consensus on the treatment of BDD. Selective BAE or surgical resection is usually used as a first-line treatment to control hemoptysis. The reviews of this paper are available via the supplemental material section.

Massive Hemoptysis in Children.

Rationale. Hemoptysis is a rare but often life-threatening condition in pediatric patients. Massive hemoptysis can easily lead to asphyxia, respiratory failure, shock, and even death. The most common causes of severe hemoptysis are lower respiratory tract infection, vascular malformation, and bronchial foreign body. We present an unusual case of massive hemoptysis caused by malformation of the bronchial artery, which includes bronchial artery hypertrophy, bronchial-pulmonary artery fistula, and ectopic bronchial artery. Patient. An 11-year-old boy was admitted to the hospital with mild hemoptysis lasting for the two preceding days. He did not report any discomfort, such as fever or chest pain. His complete blood count and coagulation function were normal. Chest X-ray documented lower right pneumonia. Massive hemoptysis occurred on the night of the admission. Diagnosis. Bronchial arteriography revealed that the right lower bronchial artery and the ectopic bronchial artery from the renal artery were the responsible vessels for hemoptysis. Interventions. The boy underwent a successful bronchial artery embolization and bronchoscopy to remove the blood clot from the airway. Outcomes. After bronchial artery embolization and bronchoscopy, the boy recovered without complications. Hemoptysis and chest pain disappeared, and chest radiographs returned to normal. Lessons. Bronchial arterial bleeding often presents as life-threatening massive hemoptysis. Patients should immediately receive hemostatic treatment and undergo chest CTA, bronchial arteriography, BAE, and bronchoscopy according to their condition. Rapid identification of the etiology and symptomatic treatment are critical to saving the lives of children.

Ectopic origin of bronchial arteries: still a potential pitfall in embolization.

PURPOSE: To evaluate the influence of ectopic origin of bronchial arteries (BAs) on bronchial artery embolization (BAE) for hemoptysis. METHODS: CT and angiography images of 50 consecutive sessions in 39 patients (aged 26-93 years; mean, 70.6 years) who underwent BAE for hemoptysis from April 2010 to December 2019 were reviewed. We defined ectopic BA as a systemic artery originating from other than the T5-T6 vertebral level of the descending aorta with course along the major bronchi. The background of patients, number of BAs, culprit arteries, and treatment outcomes were compared between the cases with and without ectopic BAs. RESULTS: Seventeen patients (43.6%) demonstrated 19 ectopic BAs, originating from the subclavian artery (n = 7), aortic arch above the T5-T6 level (n = 6), internal mammary artery (n = 3), brachiocephalic trunk (n = 2) or lower descending thoracic aorta (n = 1). Total number of BAs in the cases with ectopic BA was significantly greater than those in cases without ectopic BA (p = 0.0062). Required sessions of embolization were similar in the two groups. No procedure-related significant complications were noted; however, four ectopic BAs caused unexpected filling of contrast media or migration of the embolic material from the orthotopic BA to ectopic BA originating from the arch vessels via tiny communication. CONCLUSION: Although BAE under the presence of ectopic BA is feasible and safe, detection of BAs with ectopic origin, even of small diameter, is needed to avoid risk of non-target coursing of embolic materials.

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.

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.

Multidetector computed tomography angiography prior to bronchial artery embolization helps detect culprit ectopic bronchial arteries and non-bronchial systemic arteries originating from subclavian and internal mammary arteries and improve hemoptysis-free early survival rate in patients with hemoptysis.

OBJECTIVES: To compare the average number of culprit arteries per patient, clinical success rate, and hemoptysis-free survival rate between hemoptysis patients with multidetector computed tomography (MDCT) angiography prior to bronchial artery embolization (BAE) and those without preprocedural MDCT angiography METHODS: This retrospective study was approved by the institutional review board with waiver of patient informed consent. From September 2012 to March 2017, 157 consecutive hemoptysis patients had been undergoing BAE. Among them, 106 patients received preprocedural MDCT angiography (MDCT group), while 51 patients did not receive preprocedural MDCT angiography (control group). The average number of culprit arteries per patient, clinical success rate, and hemoptysis-free survival rate were compared between the two groups. RESULTS: The average number of culprit ectopic bronchial arteries and that of non-bronchial systemic arteries originating from the subclavian and internal mammary arteries per patient in the MDCT group were both significantly higher than those in the control group (0.15 ± 0.51 vs 0.04 ± 0.20, p = 0.022, and 0.17 ± 0.56 vs 0.08 ± 0.39, p = 0.040, respectively). The clinical success rate of BAE with preprocedural MDCT angiography tended to be higher than that without MDCT angiography (97.2 vs 88.2%, p = 0.057). Importantly, patients in the MDCT group had a significantly higher hemoptysis-free early survival rate compared to those in the control group (96.1 vs 86.7%, p = 0.031). CONCLUSIONS: Preprocedural MDCT angiography helps detect culprit ectopic bronchial arteries and non-bronchial systemic arteries originating from subclavian and internal mammary arteries during BAE, and can improve the hemoptysis-free early survival rate, which could be recommended as a regular examination prior to BAE in patients with hemoptysis. KEY POINTS: • Preprocedural MDCT angiography helps detect culprit ectopic bronchial arteries and NBSAs originating from subclavian and internal mammary arteries during BAE. • Conducting MDCT angiography prior to BAE can improve hemoptysis-free early survival rate in hemoptysis patients.