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

Ruthberg, Jeremy S; Abrol, Anish; Howard, N. Scott

doi:10.1177/0003489420962132

Cited by 6 publications

(4 citation statements)

References 14 publications

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“…No other study in literature comparing aetiology with the mean number of arteries in paediatric haemoptysis. Other vascular causes of haemoptysis reported in literature include pulmonary arteriovenous malformations, Dieulafoy’s lesions and fistulae; however, these are rare in the paediatric age group 21 22 and no such cases were identified in the present study.…”

Section: Discussioncontrasting

confidence: 50%

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

Naranje

Shera

Bhalla

et al. 2022

Indian Journal of Medical Research

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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.

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Section: Discussioncontrasting

confidence: 50%

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

Naranje

Shera

Bhalla

et al. 2022

Indian Journal of Medical Research

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“…Major complications of BAE include spinal ischemia, ischemic colitis, myocardial infarction, and stroke, occurring in less than 5% of cases (21). Proper choice of embolic material and identification of hazardous collaterals are crucial to avoid inadvertent embolization In contrast to earlier reports revealing an unsatisfactory recurrence rate (20/38) after BAE in adult cases (11), pediatric patients may have a better prognosis with a lower recurrence rate (2/10) at similar follow-up times (3)(4)(5)(6). The difference in prognosis may also be associated with the progress of BAE in the last decade.…”

Section: Discussionmentioning

confidence: 99%

“…Moreover, only five out of the 33 postoperative or autopsied histological specimens revealed classic pathological features. Among the ten documented cases of pediatric BDD in the literature (3)(4)(5)(6), none had a pathological biopsy performed. Considering the above, endobronchial biopsies were not considered in our patients.…”

Section: Discussionmentioning

confidence: 99%

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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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An unexpected bronchoscopic finding possibly induced by extracorporeal membrane oxygenation: A case report of bronchial Dieulafoy disease

Lv,

Hu,

et al. 2024

Medicine

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Rationale: Bronchial Dieulafoy disease (BDD), a rarely reported disease, comes from dilated or abnormal arteries under the bronchial mucosa. Patients with BDD are generally asymptomatic so this disease is frequently misdiagnosed. However, the submucosal arteries may dilate and rupture for various reasons, leading to recurrent respiratory tract bleeding and potentially life-threatening conditions. With the change of reversible factors such as intravascular pressure, the arteries may return to normal, allowing patients to recover to an asymptomatic state. This phenomenon has not been mentioned and concerned in previous studies, but it may have important implications for our correct understanding of this disease. Patient concerns: A 44-year-old female was admitted to intensive care unit with recurrent malignant arrhythmias. With the assistance of VA-extracorporeal membrane oxygenation (ECMO), both her vital signs and internal environment were all gradually stabilized. However, she had been experiencing recurrent respiratory tract bleeding. While removing the bloody secretion with a fiber bronchoscopy, a congested protruding granule on the wall of the patient’s left principal bronchus was found. Diagnosis: The patient was diagnosed with BDD and the granule was thought to be an abnormal artery of BDD. Interventions: For the patient’s condition, we did not implement any targeted interventions with the abnormal artery. Outcomes: After the weaning of VA-ECMO, the patient’s granule could not be found and the bleeding had also disappeared. She gradually weaned off the mechanical ventilation and was transferred to the Department of Cardiology. Then the patient was discharged after her condition stabilized. In more than half a year, the patient is in a normal physical condition. Lessons: The appearance and disappearance of abnormal artery is an interesting phenomena of BDD. The change of intravascular pressure due to various causes such as VA-ECMO may be the primary factor of it.

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

Cited by 6 publications

References 14 publications

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

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

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

An unexpected bronchoscopic finding possibly induced by extracorporeal membrane oxygenation: A case report of bronchial Dieulafoy disease

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