Pediatric bronchial Dieulafoy's disease with bronchial artery embolization: two case reports.
Bronchial Dieulafoy’s disease (BDD)
bronchial artery embolization (BAE)
case report
children
hemoptysis
Journal
Translational pediatrics
ISSN: 2224-4344
Titre abrégé: Transl Pediatr
Pays: China
ID NLM: 101649179
Informations de publication
Date de publication:
31 Jan 2023
31 Jan 2023
Historique:
received:
28
06
2022
accepted:
02
12
2022
entrez:
17
2
2023
pubmed:
18
2
2023
medline:
18
2
2023
Statut:
ppublish
Résumé
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. 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. 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.
Sections du résumé
Background
UNASSIGNED
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
UNASSIGNED
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
UNASSIGNED
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.
Identifiants
pubmed: 36798928
doi: 10.21037/tp-22-294
pii: tp-12-01-79
pmc: PMC9926130
doi:
Types de publication
Case Reports
Langues
eng
Pagination
79-85Informations de copyright
2023 Translational Pediatrics. All rights reserved.
Déclaration de conflit d'intérêts
Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://tp.amegroups.com/article/view/10.21037/tp-22-294/coif). The authors have no conflicts of interest to declare.
Références
Thorax. 1995 Jun;50(6):697-8
pubmed: 7638820
Ann Thorac Surg. 2005 Sep;80(3):1126-8
pubmed: 16122511
Exp Ther Med. 2022 Jan;23(1):100
pubmed: 34976142
Sao Paulo Med J. 2017 Jul-Aug;135(4):396-400
pubmed: 28562735
Indian Pediatr. 2021 Mar 15;58(3):287-288
pubmed: 33713072
Respir Med Case Rep. 2019 Feb 05;26:248-250
pubmed: 30788209
Front Pediatr. 2020 May 28;8:273
pubmed: 32670994
Respir Res. 2008 Aug 05;9:58
pubmed: 18681960
Ann Otol Rhinol Laryngol. 2021 May;130(5):528-531
pubmed: 33025790
Ther Adv Respir Dis. 2020 Jan-Dec;14:1753466620929236
pubmed: 32482143
Medicine (Baltimore). 2019 Feb;98(7):e14471
pubmed: 30762767
Am J Respir Crit Care Med. 2017 Feb 1;195(3):397
pubmed: 27925465
BMC Pulm Med. 2019 Jun 6;19(1):104
pubmed: 31170962
Ann Thorac Med. 2020 Oct-Dec;15(4):244-246
pubmed: 33381240
Semin Intervent Radiol. 2012 Sep;29(3):155-60
pubmed: 23997406
J Cardiothorac Surg. 2014 Dec 02;9:191
pubmed: 25438694
Respir Med Case Rep. 2019 Jan 07;26:229-235
pubmed: 30740302
Respir Med Case Rep. 2015 May 27;16:20-3
pubmed: 26744645
Paediatr Respir Rev. 2020 Nov;36:100-105
pubmed: 32680823
Paediatr Respir Rev. 2008 Jun;9(2):95-104
pubmed: 18513669
Can Respir J. 2003 Sep;10(6):331-3
pubmed: 14530826
Front Pediatr. 2021 Sep 15;9:674509
pubmed: 34604131