11岁男性患儿，因间断发热15 d，咳嗽10 d，“咯血”7 d入院。患儿15 d前以发热咳嗽起病，伴左侧颈部疼痛，抗生素治疗有效。病程中突然出现大“咯血”致休克，后经纤维支气管镜检查发现左侧梨状窝内瘘口并持续渗血，结合颈部及血管影像学检查考虑为先天性梨状窝瘘感染并颈部脓肿致颈内静脉受损血栓形成，通过低温等离子消融梨状窝瘘封闭术治疗后好转，随访1年半未反复。检索文献未发现梨状窝瘘感染引起大出血休克报道，该文总结此例患儿的临床特点及诊治经过，为早期诊断该类疾病及其并发症防治提供参考依据。
Abstract
A boy, aged 11 years, was admitted due to intermittent fever for 15 days, cough for 10 days, and "hemoptysis" for 7 days. The boy had fever and cough with left neck pain 15 days ago, and antibiotic treatment was effective. During the course of disease, the boy developed massive "hemoptysis" which caused shock. Fiberoptic bronchoscopy revealed a left pyriform sinus fistula with continuous bleeding. In combination with neck and vascular imaging examination results, the boy was diagnosed with internal jugular vein injury and thrombosis due to congenital pyriform sinus fistula infection and neck abscess. The boy was improved after treatment with temperature-controlled radiofrequency ablation for the closure of pyriform sinus fistula, and no recurrence was observed during the follow-up for one year and six months. No reports of massive hemorrhage and shock due to pyriform sinus fistula infection were found in the searched literature, and this article summarizes the clinical features, diagnosis, and treatment of this boy, so as to provide a reference for the early diagnosis of such disease and the prevention and treatment of its complications.
关键词
梨状窝瘘 /
颈内静脉损伤 /
大出血 /
休克 /
儿童
Key words
Pyriformis sinus fistula /
Internal jugular vein injury /
Massive hemorrhage /
Shock /
Child
{{custom_sec.title}}
{{custom_sec.title}}
{{custom_sec.content}}
参考文献
1 中华医学会儿科学分会呼吸学组, 《中华实用儿科临床杂志》编辑委员会. 儿童咯血诊断与治疗专家共识[J]. 中华实用儿科临床杂志, 2016, 31(20): 1525-1530. DOI: 10.3760/cma.j.issn.2095-428X.2016.20.002.
2 蒋廷, 李渠北. 儿童弥漫性肺泡出血[J]. 中国当代儿科杂志, 2019, 21(9): 949-954. PMID: 31506159. PMCID: PMC7390247. DOI: 10.7499/j.issn.1008-8830.2019.09.020.
3 陈伟, 陈佳瑞, 陈芳, 等. 内镜下射频消融治疗儿童急性感染期梨状窝瘘[J]. 中国耳鼻咽喉头颈外科, 2021, 28(2): 109-111. DOI: 10.16066/j.1672-7002.2021.02.011.
4 Sandborn WD, Shafer AD. A branchial cleft cyst of fourth pouch origin[J]. J Pediatr Surg, 1972, 7(1): 82. PMID: 5013141. DOI: 10.1016/0022-3468(72)90421-6.
5 中华医学会外科学分会血管外科学组. 深静脉血栓形成的诊断和治疗指南(第三版)[J]. 中华普通外科杂志, 2017, 32(9): 807-812. DOI: 10.3760/cma.j.issn.1007-631X.2017.09.032.
6 Audu CO, Wakefield TW, Coleman DM. Pediatric deep venous thrombosis[J]. J Vasc Surg Venous Lymphat Disord, 2019, 7(3): 452-462. PMID: 30853559. DOI: 10.1016/j.jvsv.2018.12.012.
7 James A, Stewart C, Warrick P, et al. Branchial sinus of the piriform fossa: reappraisal of third and fourth branchial anomalies[J]. Laryngoscope, 2007, 117(11): 1920-1924. PMID: 17878800. DOI: 10.1097/MLG.0b013e31813437fc.
8 马静, 明澄, 娄凡, 等. 儿童先天性梨状窝瘘的误诊原因分析及治疗方法探讨[J]. 中华耳鼻咽喉头颈外科杂志, 2018, 53(5): 381-384. PMID: 29764021. DOI: 10.3760/cma.j.issn.1673-0860.2018.05.010.
9 Madana J, Yolmo D, Gopalakrishnan S, et al. Cervical infection secondary to pyriform sinus fistula of branchial origin[J]. Congenit Anom (Kyoto), 2009, 49(4): 276-278. PMID: 20021489. DOI: 10.1111/j.1741-4520.2009.00249.x.
10 董锦锦, 田秀芬. 先天性梨状窝瘘的诊断与治疗经验探讨[J]. 中华耳鼻咽喉头颈外科杂志, 2018, 53(6): 444-447. PMID: 29902850. DOI: 10.3760/cma.j.issn.1673-0860.2018.06.011.
11 王奥楠, 邬海博. 鳃裂瘘管的CT及MRI表现[J]. 实用放射学杂志, 2018, 34(4): 515-517. DOI: 10.3969/j.issn.1002-1671.2018.04.007.
12 Wang HK, Tiu CM, Chou YH, et al. Imaging studies of pyriform sinus fistula[J]. Pediatr Radiol, 2003, 33(5): 328-333. PMID: 12695866. DOI: 10.1007/s00247-003-0887-8.
13 Davidson K, Shojaee S. Managing massive hemoptysis[J]. Chest, 2020, 157(1): 77-88. PMID: 31374211. DOI: 10.1016/j.chest.2019.07.012.
14 梁璐, 陈柳勤, 陈良嗣, 等. 先天性梨状窝瘘继发颈深部感染的病原学分析及治疗[J]. 临床耳鼻咽喉头颈外科杂志, 2018, 32(7): 514-518. PMID: 29798081. DOI: 10.13201/j.issn.1001-1781.2018.07.009.
15 Ahn D, Sohn JH, Kim H, et al. Clinical and microbiological differences between pyriform sinus fistulae in pediatric and non-pediatric patients[J]. Auris Nasus Larynx, 2015, 42(1): 34-38. PMID: 25183403. DOI: 10.1016/j.anl.2014.06.003.
16 Watson GJ, Nichani JR, Rothera MP, et al. Case series: endoscopic management of fourth branchial arch anomalies[J]. Int J Pediatr Otorhinolaryngol, 2013, 77(5): 766-769. PMID: 23478017. DOI: 10.1016/j.ijporl.2013.02.007.
17 窦倩雯, 田秀芬. 低温等离子射频消融治疗216例先天性梨状窝瘘的临床分析[J]. 山东大学耳鼻喉眼学报, 2021, 35(2): 43-49. DOI: 10.6040/j.issn.1673-3770.1.2020.092.
基金
湖南省儿童呼吸疾病重点实验室项目（2019TP1043）。
PDF(810 KB)
