Bronchial Dieulafoy's Disease in Children: A Case Report and Review of Literature

Yeh, Yi-Ting; Ramaswamy, Madhavan; Shin, Jinnie; McIntyre, Denise; McIntosh, Neil; Racicot, Jean-Nicolas; Chippington, Samantha; Stuart, Sam; Muthialu, Nagarajan

doi:10.3389/fped.2020.00273

Cited by 7 publications

(7 citation statements)

References 7 publications

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“…However, it may not be superior to bronchial arteriography due to the lower amount of contrast agents injected into the bronchial arteries, which is more evident in children with smaller vessel diameters. Abnormal bronchial arteries were unmasked by CTA in only 5 out of 10 pediatric patients in the literature ( 10 , 15 , 16 , 18 , 19 ). As noted in case 2, bronchial arteriography was strongly recommended based on bronchoscopic findings, although CTA failed to demonstrate any vascular malformations.…”

Section: Discussionmentioning

confidence: 99%

“…In this context, it may be easily misdiagnosed as a neoplasm, and a subsequent biopsy may lead to a life-threatening hemorrhage (2,11). Bronchial arteriography is the most reliable approach to uncover abnormal bronchial arteries (10,(15)(16)(17). It helps to distinguish BDD from a carcinoid tumor which is also hypervascularized.…”

Section: Discussionmentioning

confidence: 99%

“…The rarity and insufficient understanding of this “adult disease” may lead to delayed diagnosis and treatment in children with hemoptysis, which in turn results in life-threatening hemorrhage and a high mortality rate. Nevertheless, pediatric case reports are increasing annually with the advancements in diagnostic technology ( 10 ). Overall, the clinical features of BDD are broadly consistent between children and adults, with a male preponderance, hemoptysis as the first symptom, and predominant right bronchus involvement ( 11 ).…”

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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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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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“…Bronchial arterial angiography and embolization should be attempted in all cases, as the source of bleeding is systemic in more than 80% of cases. 17 Surgical resection of the affected lobe is considered the treatment of choice in recurrent cases. 18,19 Diagnosing BDD requires a high index of suspicion in patients presenting with moderate to massive haemoptysis and normal CT findings without structural lung damage (Figure 9).…”

Section: Discussionmentioning

confidence: 99%

Bronchial Dieulafoy's disease: A series of seven cases with review of the literature

Nair,

James,

Vimala

et al. 2024

Respirology Case Reports

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Bronchial Dieulafoy's disease (BDD), remains poorly understood, with only 88 cases reported globally. Herein, we present the largest case series (n = 7) from a single centre, between 2017 and 2023, retrospectively reviewed, detailing clinical presentations, diagnoses, management and up to 4‐year follow‐up outcomes. Diagnosis relied on characteristic lesions detected through white light bronchoscopy with or without endobronchial ultrasound (EBUS) or narrow band imaging (NBI), along with computed tomography (CT) scans or bronchial angiography. Identification of aberrant vessels beneath lesions and bronchoscopy details were documented. Treatment modalities and follow‐up outcomes until December 2023 were noted. All patients were non‐smokers. Review of imaging findings by an experienced radiologist was crucial in suspected cases due to risk of bleeding and often unconclusive results from biopsy. Management of BDD varied, with six patients undergoing bronchial artery embolization (BAE) and one requiring lobectomy; four patients received additional endobronchial therapy, one died due to malignancy, none experienced recurrence of haemoptysis. Identifying patients with large volume haemoptysis disproportionate to parenchymal disease in CT scans is important. A bronchoscopic surveillance is crucial to avoid biopsy; it can be confirmed using EBUS of NBI. While no established guidelines exist, BAE and endobronchial therapy emerge as valuable interventions, with surgical resection reserved for recurrent cases.

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“…Dieulafoy's disease occurs frequently but is not limited to the gastrointestinal tract. There are also many reports of bronchial Dieulafoy's disease in children, which is characterized by arterial rupture and haemorrhage caused by malformation of bronchial submucosa (21)(22)(23)(24)(25). The diagnosis and treatment of bronchial Dieulafoy's disease have provided us with many experiences and lessons for the diagnosis and treatment of the digestive tract Dieulafoy's disease.…”

Section: Discussionmentioning

confidence: 99%

Massive gastrointestinal haemorrhage caused by pancreatic pseudocyst complicated with Dieulafoy's disease in a child: A case report and review of the literature

et al. 2022

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BackgroundPancreatic pseudocyst (PPC) with massive gastrointestinal bleeding is rare, especially in children. Inadvertent intraoperative examination and damage to the gastric mucosa and malformed blood vessels by the fluid content of PPC can lead to massive bleeding, which may endanger the patient's life.Case presentationHere, we present a case of an 8-year-old boy who was diagnosed with a massive gastrointestinal haemorrhage caused by PPC complicated with Dieulafoy's disease. At his first admission, his complaint was being hit to the stomach by the handlebar while riding bicycle 24 h before admission. After being hospitalized, he was diagnosed with pancreatic injury by abdominal CT. Conservative treatment lasted for 1 month in the Department of Pediatric Surgery. Then, a pancreatic pseudocyst was formed. Under the guidance of ultrasonic endoscopy, cyst puncture and drainage of pseudocysts through the gastric wall were performed. Unexplained hematemesis occurred 8 days after surgery. Emergency gastroscopy was performed, and abnormal submucosal vascular haemorrhage was found at the gastric fundus. Gastric Dieulafoy's disease was diagnosed. The boy underwent gastroscopic titanium clipping of abnormal arteries. He had no complications during the 3-month follow-up. Then, the patient returned to the hospital, and the stent was removed under endoscopy. No bleeding was found, and the patient was discharged. The patient recovered smoothly and was followed up for half a year without any complications, and hematological indicators were normal.ConclusionEndoscopic ultrasonography-guided gastric puncture and internal drainage of cysts is a safe and effective surgical method for the treatment of pancreatic pseudocysts. However, at the same time, it is necessary to thoroughly and carefully explore the stomach cavity to prevent adverse consequences caused by a missed diagnosis of gastric Dieulafoy's disease or other abnormal abnormalities.

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Bronchial Dieulafoy's Disease in Children: A Case Report and Review of Literature

Cited by 7 publications

References 7 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

Bronchial Dieulafoy's disease: A series of seven cases with review of the literature

Massive gastrointestinal haemorrhage caused by pancreatic pseudocyst complicated with Dieulafoy's disease in a child: A case report and review of the literature

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