Journal of Clinical Pediatrics ›› 2025, Vol. 43 ›› Issue (2): 135-140.doi: 10.12372/jcp.2025.24e0996
• Clinical Report • Previous Articles Next Articles
LIN Lihua, ZHANG Ning, CHEN Qihong, CHEN Lili, CHEN Lixian, YANG Yungang(
)
Received:2024-09-19
Accepted:2024-11-26
Published:2025-02-15
Online:2025-02-12
LIN Lihua, ZHANG Ning, CHEN Qihong, CHEN Lili, CHEN Lixian, YANG Yungang. Bronchial dieulafoy's disease in children: a case report and review of literature[J].Journal of Clinical Pediatrics, 2025, 43(2): 135-140.
Table 1
Clinical characteristics of 17 patients"
| 患儿 | 性别 | 年龄 | 病变位置 | 支气管镜 | 肺部CT血管造影 | BAE |
|---|---|---|---|---|---|---|
| 1[ | 男 | 13岁 | 右肺下叶 | 右下叶黏膜结节样突起 | 右支气管动脉在T5水平从胸主动脉的前壁出现 | PVA治疗3个月后再反复大咯血,失败 |
| 2[ | 女 | 16岁 | 右肺下叶 | 右下叶开口黏膜结节样突起 | NA | NA |
| 3[ | 女 | 5岁 | 右肺上叶 | 右主支气管间脊处见小结节增生,直径约为0.5 cm | 仅仅做肺部增强CT未见异常强化灶 | PVA颗粒栓塞2次失败 |
| 4[ | 女 | 18岁 | 右肺下叶 | 4年前右下后基底段开口一结节,活检出血300 mL,4年后复查未见结节 | NA | 介入封堵,材料不详,成功 |
| 5[ | 女 | 7岁 | 右中间支气管 | 右中间支气管结节样突起 | 右中间支气管有异常血管 | NA |
| 6[ | 男 | 8岁 | 右主支气管 | 右主支气管血块堵塞 | 右支气管动脉异常扩张 | NBCA栓塞,成功 |
| 7[ | 男 | 11岁 | 右肺下叶 | NA | 右支气管动脉扩张弯曲,右肺下叶弥漫性磨玻璃 | 弹簧圈封堵,成功 |
| 8[ | 男 | 8岁 | 右肺下叶 | 右下叶出现粉红色丘疹样病变及搏动性出血 | NA | 弹簧圈栓塞3处,成功 |
| 9[ | 男 | 15岁 | 右肺下叶 | 右肺下叶活动性出血 | 右肺下叶模糊影,无肺栓塞迹象 | PVA颗粒栓塞,成功 |
| 10[ | 男 | 9月 | 右肺下叶 | 右肺下叶光滑的黏膜突起,接触后出血明显 | NA | PVA颗粒和GS栓塞,成功 |
| 11[ | 男 | 9月 | 右肺下叶开口 | 右下基底段可见一向官腔内隆起的新生物 | 心脏CTA未见明显异常 | PVA颗粒栓塞1次,成功 |
| 12[ | 男 | 10岁 | 右肺背段 | 右肺中叶与下叶交汇处可见红色圆形光滑凸起 | 右肺背段模糊密度增高影,可见迂曲小血管两肺内斑片状、大片状密度增高影 | PVA栓塞颗粒及弹簧圈封堵,成功 |
| 13[ | 男 | 9岁 | 右下前基底段 | 右肺下叶前基底段可见一新生物,管腔内出血 | 右肺下叶模糊片影,左肺散在慢性炎症 | PVA栓塞微球和GS栓塞 |
| 14[ | 女 | 6岁 | 右中下叶 | 右中下叶支气管侧壁帽状突起,高度约4 mm,可见迂曲、无搏动 | 没有显示血管畸形,但在右支气管处发现了一个具有对比度增强的小突起 | PVA颗粒栓塞,成功 |
| 15[ | 男 | 8岁 | 右肺下叶前基底段 | 右下肺前基底段2个2~4 mm白色结节 | 增粗支气管动脉向下延伸至右下叶,起源于胸主动脉,支气管与肺动脉分流 | PVA颗粒栓塞,成功 |
| 16[ | 男 | 4岁 | 右下叶基底段 | 右中间支气管侧壁结节性突起 | 右下肺另一条弯曲扩张的供血动脉,是膈下动脉(IPA)的异常上行支 | PVA封堵后1个月咯血复发,再次封堵 |
| 本文 | 女 | 11岁 | 右中下叶 | 右下叶各基底段可见鲜血渗出,右下叶开口处黏膜可见突起,表面光滑 | 气管隆突下方增粗迂曲血管影,考虑增粗的支气管动脉 | GS栓塞,成功 |
| [1] | Senger JL, Kanthan R. The evolution of Dieulafoy's lesion since 1897: then and now—a journey through the lens of a pediatric lesion with literature review[J]. Gastroenterol Res Pract, 2012, 2012: 432517. |
| [2] | Qian X, Du Q, Wei N, et al. Bronchial Dieulafoy's disease: a retrospective analysis of 73 cases[J]. BMC Pulm Med, 2019, 19(1): 104. |
| [3] |
Fang Y, Wu Q, Wang B. Dieulafoy’s disease of the bronchus: report of a case and review of the literature[J]. J Cardiothorac Surg, 2014, 9: 191.
doi: 10.1186/s13019-014-0191-8 pmid: 25438694 |
| [4] |
Wadji MB, Farahzadi A. Dieulafoy's disease of the bronchial tree: a case report[J]. Sao Paulo Med J, 2017, 135(4): 396-400.
doi: S1516-31802017000400396 pmid: 28562735 |
| [5] | Niu HL, Yi P, Wang H, et al. Infantile Dieulafoy's disease of bronchus: report of a case[J]. Zhonghua Binglixue Zazhi, 2017, 46(10): 731-732. |
| [6] |
Chen W, Chen P, Li X, Gao X, Li J. Clinical characteristics and treatments for bronchial Dieulafoy's disease[J]. Respir Med Case Rep, 2019, 26: 229-235.
doi: 10.1016/j.rmcr.2019.01.004 pmid: 30740302 |
| [7] | Yeh YT, Ramaswamy M, Shin J, et al. Bronchial Dieulafoy’s disease in children: a case report and review of literature[J]. Front Pediatr, 2020, 8: 273. |
| [8] |
Woodhull S, Bush A, Tang AL, et al. Massive paediatric pulmonary haemorrhage in Dieulafoy’s disease: roles of CT angiography, embolisation and bronchoscopy[J]. Paediatr Respir Rev, 2020, 36: 100-105.
doi: 10.1016/j.prrv.2020.06.001 pmid: 32680823 |
| [9] |
Giordano M, Bigazzi MC, Palladino MT, et al. A rare cause of massive hemoptysis in a child: Bronchial Dieulafoy's disease—the first report of transcatheter treatment in pediatric age[J]. Ann Thorac Med, 2020, 15(4): 244-246.
doi: 10.4103/atm.ATM_163_20 pmid: 33381240 |
| [10] | Ruthberg JS, Abrol A, Howard NS, et al. Recurrent hemoptysis: a bronchial Dieulafoy’s lesion in a pediatric patient[J]. Ann Otol Rhinol Laryngol, 2021, 130(5): 528-531. |
| [11] |
Vijayasekaran D, Sivabalan S. Bronchial Dieulafoy disease with recurrent life-threatening hemoptysis[J]. Indian Pediatr, 2021, 58(3): 287-288.
pmid: 33713072 |
| [12] | Chen Y, Mao Y, Cheng X, et al. Case report: a case of infant bronchial Dieulafoy's disease and article review[J]. Front Pediatr, 2021, 9: 674509. |
| [13] | Han F, Du Q, Lu XX, et al. A case of Dieulafoy disease of children bronchus[J]. Chinese Journal of Practical Pediatrics, 2021, 36(8): 1191-1193. |
| [14] | Wang YJ, Wu L, Hu J, et al. Endovascular embolization for bronchial Dieulafoy's disease: a report of 2 cases[J]. Chinese Journal of Evidence-Based Pediatrics, 2023, 18(4): 155-158. |
| [15] | Ling YL, Yong L, X ZM, et al. Pediatric bronchial Dieulafoy’s disease with bronchial artery embolization: two case reports[J]. Transl Pediatr. 2023, 12(1): 79-85. |
| [16] |
Fang W, Jiao T, Mou P, et al. Recurrent hemoptysis in pediatric bronchial Dieulafoy’s disease with inferior phrenic artery supply: a case report[J]. World J Clin Cases. 2023, 11(26): 6268-6273.
doi: 10.12998/wjcc.v11.i26.6268 pmid: 37731559 |
| [17] |
Stoopen E, Baquera-Heredia J, Cortes D, et al. Dieulafoy's disease of the bronchus in association with a paravertebral neurilemoma[J]. Chest, 2001, 119: 292-294.
doi: 10.1378/chest.119.1.292 pmid: 11157620 |
| [18] | Yang RH, Li JF, Liu J, et al. Three cases of bronchial Dieulafoy disease and literature review[J]. Chinese Journal of Tuberculosis and Respiratory Diseases, 2013, 36(8): 577-580. |
| [19] | Huang ZC, Tang CM, Chen SL, et al. Diagnosis of bronchial Dieulafoy's by bronchial arteriography CT combined with bronchoscopy (with 5 case reports)[J]. Chinese Journal of Physicians, 2022, 24(11): 1697-1701. |
| [20] | Qian X, Du Q, Wei N, et al. Bronchial Dieulafoy's disease: a retrospective analysis of 73 cases[J]. BMC Pulm Med, 2019, 19(1): 104. |
| [21] | Padilla-Serrano A, Estrella-Palomares V, Martinez-Palacios B, et al. A case of massive hemoptysis related to a smoking-history: an acquired form of the Dieulafoy's disease?[J]. Rev Port Pneumol, 2015, 21(5): 275-279. |
| [22] | Yue TH, Yuan P, Wu S, et al. A controlled study of different bronchial artery embolization materials in the treatment of massive hemoptysis[J]. Journal of Practical Radiology, 2021, 37(1): 124-127. |
| [23] | Xia XD, Ye LP, Zhang WX, et al. Massive cryptogenic hemoptysis undergoing pulmonary resection: clinical and pathological characteristics and management[J]. Int J Clin Exp Med, 2015, 8(10): 18130-18136. |
| [24] | Xing XQ, Liu J, XU S, et al. Research advances in Dieulafoy's disease of the bronchus (Review)[J]. Exp Ther Med, 2022, 23(1): 100. |
| [1] | LIN Jinfeng, CHEN Yi, DENG Yan, FENG Ai, HUANG Juan, CHEN Lizhu, XIE Jingqi, NIE Xiaojing. A comparative study on the detection of anti-Nephrin antibodies in children's nephrotic syndrome by chemiluminescence method and cell-based assay [J]. Journal of Clinical Pediatrics, 2026, 44(8): 682-689. |
| [2] | ZHENG Fangyuan, WANG Miao, DING Mingming, LU Aidong, JIA Yueping, ZENG Huimin, ZHANG Leping. Next-generation sequencing identified gene variants and their prognostic impact in pediatric Philadelphia chromosome positive acute lymphoblastic leukemia [J]. Journal of Clinical Pediatrics, 2026, 44(8): 690-697. |
| [3] | ZHANG Xiao, FANG Hongjun, KUANG Xiaojun, WANG Lijuan, WU Zhao, JIANG Sha, LI Pei, WU Liwen. Analysis of the clinical and electrophysiological characteristics of atonic seizures in children [J]. Journal of Clinical Pediatrics, 2026, 44(8): 698-704. |
| [4] | WANG Mengqian, YANG Hui. Case series analysis of 13 intestinal Behcet’s syndrome in children [J]. Journal of Clinical Pediatrics, 2026, 44(8): 705-711. |
| [5] | WU Xirong, GAO Qi, GUO Siyuan, YIN Qingqin, QIN Qiang, YIN Ju, JIAO Anxia, CHEN Chenghao, WU Di, WANG Bei, DUAN Xiaomin, PENG Yun, XU Baoping, GAO Liwei. Clinical characteristics and prognosis of pulmonary mucormycosis in children with diabetes mellitus [J]. Journal of Clinical Pediatrics, 2026, 44(8): 712-720. |
| [6] | HAN Yanan, GE Lanlan, CUI Jieyuan. A case report of branchio-oto-renal syndrome caused by EYA1 gene variant with a 6-year follow-up [J]. Journal of Clinical Pediatrics, 2026, 44(8): 721-726. |
| [7] | SHI Jiayi, GAO Chunlin, JIA Lili, LU Yunyun, SUN Tao, ZHU Xiaodong, XIA Zhengkun, ZHANG Pei. Phenotypic characteristics and prognostic analysis of a pedigree with childhood isolated nephrotic syndrome caused by compound heterozygous mutations in the COQ2 gene [J]. Journal of Clinical Pediatrics, 2026, 44(8): 727-732. |
| [8] | Rachel Rosen, Osvaldo Borrelli, Christophe Faure, Katja Karrento, Usha Krishnan, Samuel Nurko, Nathalie Rommel, Alan Silverman, Michiel van Wijk, Marc Benninga (Author), Chinese Medical Association Parenteral and Enteral Nutrition Branch Pediatrics Group, The Subspecialty Group of Gastroenterology, The Society of Pediatric, Chinese Medical Association, Gastroenterology Group, Pediatrician Branch, Chinese Medical Doctor Association (Translator). Core interpretation and explanation of Rome Ⅴ diagnostic criteria: upper gastrointestinal disorders of gut-brain interaction in children and adolescents [J]. Journal of Clinical Pediatrics, 2026, 44(7): 595-608. |
| [9] | Carlo Di Lorenzo, Miguel Saps, Bruno P. Chumpitazi, Shaman Rajindrajith, Annamaria Staiano, Nikhil Thapar, Miranda van Tilburg, Carlos Velasco-Benítez, Arine Vlieger (Author), Chinese Medical Association Parenteral and Enteral Nutrition Branch Pediatrics Group, The Subspecialty Group of Gastroenterology, The Society of Pediatric, Chinese Medical Association, Gastroenterology Group, Pediatrician Branch, Chinese Medical Doctor Association (Translator). Core interpretation and explanation of Rome Ⅴ diagnostic criteria: lower and biliary disorders of gut-brain interaction in children and adolescents [J]. Journal of Clinical Pediatrics, 2026, 44(7): 609-621. |
| [10] | LI Xufeng, TANG Haoxun, LI Xingjun, ZHANG Lize, GAO Chenqing, ZHAO Shunying, LIU Jianhua. Clinical characteristics, chest CT imaging phenotypes, and their correlation with prognosis in 76 children with Chlamydia pneumoniae pneumonia [J]. Journal of Clinical Pediatrics, 2026, 44(7): 622-627. |
| [11] | WANG Zhen, SHAO Jingbo, ZHANG Na, XIA Min, ZHU Jiashi, DU Chengkan, LI Hong. Factors influencing long-term recurrence in pediatric acute lymphoblastic leukemia patients with low-risk disease at initial diagnosis: a five-year follow-up analysis of 254 cases from a single center [J]. Journal of Clinical Pediatrics, 2026, 44(7): 628-635. |
| [12] | GUO Xuemei, XUE Yao, WANG Yongren, RONG Liucheng, FANG Yongjun, LIN Rufeng. Clinical features and prognosis of childhood acute myeloid leukemia with CBFA2T3::GLIS2 fusion [J]. Journal of Clinical Pediatrics, 2026, 44(7): 636-643. |
| [13] | YANG Lijun, FU Dongxia, CUI Yan, YANG Junmei, ZHANG Liming. Clinical manifestation and genetic analysis of CHARGE syndrome caused by CHD7 gene variation [J]. Journal of Clinical Pediatrics, 2026, 44(7): 644-650. |
| [14] | HU Yan, YANG Fan, LIANG Aimin, MAO Meng. Identification and management of faltering growth in children: insights from the 13th International Conference on Nutrition and Growth [J]. Journal of Clinical Pediatrics, 2026, 44(6): 485-488. |
| [15] | LIU Ziyao, WANG Congying, WANG Hongmao, ZHANG Mingming, XU Yingjie, LAI Jianming, NIU Wenquan, LI Xiaohui. Early differentiation of Kawasaki disease with joint symptoms and systemic juvenile idiopathic arthritis with Kawasaki disease-like symptoms: a single-center retrospective study [J]. Journal of Clinical Pediatrics, 2026, 44(6): 489-498. |
|
||