临床儿科杂志 ›› 2025, Vol. 43 ›› Issue (2): 135-140.doi: 10.12372/jcp.2025.24e0996

• 临床报道 • 上一篇    下一篇

儿童支气管Dieulafoy病1例并文献复习

林丽华, 张宁, 陈奇洪, 陈莉莉, 陈丽羡, 杨运刚()   

  1. 厦门大学附属第一医院儿科 厦门市儿科重点实验室 厦门大学医学院儿童医学研究所 (福建厦门 361003)
  • 收稿日期:2024-09-19 录用日期:2024-11-26 出版日期:2025-02-15 发布日期:2025-02-12
  • 通讯作者: 杨运刚 电子信箱:xmyyg@sina.com
  • 基金资助:
    国家重点研发计划资助项目编号(2022YFC2704803);厦门市医疗卫生重点项目(YDZX20193502000003)

Bronchial dieulafoy's disease in children: a case report and review of literature

LIN Lihua, ZHANG Ning, CHEN Qihong, CHEN Lili, CHEN Lixian, YANG Yungang()   

  1. Department of Pediatrics, Pediatric Key Laboratory of Xiamen, Institute of Pediatrics, The First Affiliated Hospital of Xiamen University, School of Medicine, Xiamen University, Xiamen 361003, Fujian, China
  • Received:2024-09-19 Accepted:2024-11-26 Published:2025-02-15 Online:2025-02-12

摘要:

目的 总结儿童支气管Dieulafoy病的临床诊治过程,探讨该疾病的临床特点及治疗方案。方法 对1例儿童支气管Dieulafoy病的临床资料进行回顾性分析,并复习文献进行总结。结果 患者为11岁女性,咳嗽伴咯血,电子支气管镜检查显示右中下叶开口处结节样隆起,结合支气管造影检查考虑为支气管Dieulafoy病。经过支气管动脉栓塞治疗后未再出现出血。共检索到病例报告14篇,包含本例在内共17例患者,发病年龄范围为8个月至18岁,多见于男童。所有病例均表现为咯血,病变均发生在右肺。经支气管动脉栓塞和/或肺叶切除治疗的16例患者均未出现复发,仅有1例患儿在先后接受两次支气管动脉栓塞和右肺上叶切除后再次出现咯血,但在内科保守随访1年内未再复发。结论 儿童支气管Dieulafoy病较为罕见,支气管镜检查中出现的结节样隆起需注意鉴别,以避免盲目活检。支气管动脉造影在诊断该病方面具有重要帮助。支气管动脉栓塞术是儿童支气管Dieulafoy病主要治疗方法,如仍反复咯血可行支气管肺叶切除术。

关键词: 支气管Dieulafoy病, 咯血, 支气管镜, 儿童

Abstract:

Objective To summarize the clinical diagnosis and treatment process of Dieulafoy's disease in children, and explore the clinical characteristics and treatment options of the disease. Methods A retrospective analysis was conducted on the clinical data of a child with bronchial Dieulafoy disease, and relevant literature reports were summarized. Results The patient was an 11-year-old female with acute onset, presenting with cough and hemoptysis, without extrapulmonary symptoms. Bronchoscopy showed a smooth mucosal protrusion at the opening of the right middle and lower lobes, and bronchial artery embolization was considered after bronchial angiography. There was no recurrence of bleeding after bronchial artery embolization treatment. A total of 14 case reports were retrieved, including 17 children, with age of onset ranging from 8 months to 18 years, with a predominance in males. All cases presented with hemoptysis, all the lesions occurred in the right lung. Among the 16 children treated with bronchial artery embolization and/or lobectomy, there were no recurrences, except for 1 child who had hemoptysis again after two bronchial artery embolizations and right upper lobe resection, but did not have a recurrence within 1 year of conservative follow-up. Conclusion Bronchial Dieulafoy disease in childhood is rare. For children with unexplained hemoptysis, nodular elevations seen on bronchoscopy need to be carefully differentiated to avoid blind biopsy. Bronchial artery angiography plays an important role in diagnosing this disease. Bronchial artery embolization is the main treatment for children with bronchial Dieulafoy disease, and if the hemoptysis persists, a lobectomy of the bronchus and lung may be performed.

Key words: bronchial Dieulafoy, hemoptysis, bronchoscopy, child