新生儿咽旁间隙卡梅现象1例并文献复习

doi:10.6040/j.issn.1673-3770.0.2021.410

摘要/Abstract

摘要： 目的学习和掌握新生儿咽旁间隙卡梅现象(KMP)的临床特点、诊断及治疗方案的选择。方法回顾总结1例新生儿咽旁间隙KMP的诊疗过程,并复习相关文献。结果患儿先期治疗采用泼尼松和普萘洛尔联合口服效果不佳,后改用博来霉素、地塞米松混合液瘤体内灌注,分二期完成治疗,间隔1个月,瘤体消失,实验室检查正常。随访2年,患儿恢复良好,无不良反应出现及后遗症发生。结论 KMP是一种对婴幼儿危害较大的罕见疾病,发生于咽旁间隙尤为少见,目前尚无统一治疗方案,需个体化制定治疗措施。病例提示对于难以手术切除和全身用药治疗效果不佳的患儿,博来霉素、地塞米松局部注射是安全有效的治疗手段。

关键词: 卡梅现象, 新生儿, 咽旁间隙, 博来霉素, 地塞米松

Abstract: Objective We aimed to evaluate - the clinical characteristics,the diagnosis,and treatment of Kasabach-Merritt phenomenon(KMP)in newborns. Methods We retrospectively analyzed a case of new born with KMP in parapharyngeal space,along with a review of the related literature. Results In this case, oral administration of Prednisone and Propranolol was ineffective.As such,a combined solution of bleomycin and dexamethasone was injected into the tumor, and the treatment was completed in two stages, with an interval of one month.The tumor eventually resolved,and laboratory examinations were found to be normal. The patient was followed up for 2 years who showed good recovery without adverse reactions or sequelae. Conclusion KMP is a rare but greatharmful disease among in fants, especially rare in the parapharyngeal space. As there is no unified treatment at presen,there is a need for effective individualized management. This particular case shows that local injection of bleomycin and dexame thasone may be a safe and effective treatment in neonates with difficult surgical resection who are resistant to systemic medication.

Key words: Kasabach-Merritt phenomenon, Neonatal, Parapharyngeal space, Bleomycin, Dexamethasone

中图分类号:

R766.9

朱晶,张睿,赵媛,李炀,赵昱. 新生儿咽旁间隙卡梅现象1例并文献复习[J]. 山东大学耳鼻喉眼学报, 2022, 36(4): 55-59.

ZHU Jing, ZHANG Rui, ZHAO Yuan, LI Yang, ZHAO Yu. Newborn with Kasabach-Merritt phenomenon in parapharyngeal space:a case report and literature review[J]. Journal of Otolaryngology and Ophthalmology of Shandong University, 2022, 36(4): 55-59.

参考文献

[1] 中华医学会整形外科分会血管瘤和脉管畸形学组.血管瘤和脉管畸形的诊断及治疗指南(2019版)[J].组织工程与重建外科杂志, 2019, 5:277-317. doi:10.3969/j.issn.1673-0364.2019.05.001.
[2] Kasabach HH, Merritt KK. Capillary hemangioma with extensive purpura[J]. Am J Dis Child, 1940, 59: 1063-70. doi:10.1001/archpedi.1940.01990160135009.
[3] Enjolras O, Wassef M, Mazoyer E, et al. Infants with Kasabach-Merritt syndrome do not have “true” hemangiomas [J]. J Pediatr, 1997, 130(4):631-640. doi: 10.1016/s0022-3476(97)70249-x.
[4] Sarkar M, Mulliken JB, Kozakewich HP, et al. Thrombocytopenic coagulopathy(Kasabach-Merritt phenomenon)is associated with Kaposiform hemangioendothelioma and not with common infantile hemangioma [J]. Plast Reconstr Surg, 1997, 100( 6):1377-1386. doi: 10.1097/00006534-199711000-00001.
[5] Roteau SE, Kozakewich HP, Perez-Atayde AR, et al. Kaposiform lymphangiomatosis: a distinct aggressive lymphatic anomaly[J]. J Pediatr, 2014, 164(2): 383-388. doi:10.1016/j.jpeds.2013.10.013.
[6] Croteau SE, Liang MG, Kozakewich HP, et al. Kaposiform hemangioendothelioma: atypical features and risks of Kasabach- Merrittphenomenon in 107 referrals[J]. J Pediatr, 2013,162(1):142-147. doi:10.1016/j.jpeds.2012.06.044.
[7] Seo SK, Suh JC, Na GY, et al. Kasabach-Merritt syndrome: identification of platelet trapping in a tufted angioma by immunohistochemistry technique using monoclonal antibody to CD61[J]. Pediatr Dermatol, 1999, 16(5): 392-394. doi: 10.1046/j.1525-1470.1999.00103.x.
[8] Chinello M, di Carlo D, Olivieri F, et al. Successful management of kaposiform hemangioendothelioma with long-term sirolimus treatment: a case report and review of the literature[J]. Mediterr J Hematol Infect Dis, 2018, 10(1): e2018043. doi:10.4084/MJHID.2018.043.
[9] 王英豪, 戴立英,王丽丽, 等. 16例新生儿卡梅综合征临床分析[J]. 中国当代儿科杂志, 2021, 23(7): 696-701. doi:10.7499/j.issn.1008-8830.2103146. WANG Yinghao, DAI Liying, WANG Lili, et al. Clinical features of Kasabach-Merritt syndrome: an analysis of 16 neonates[J]. ChineseJournal of Contemporary Pediatrics, 2021, 23(7): 696-701. doi:10.7499/j.issn.1008-8830.2103146.
[10] 树叶, 黄文雅,罗勇奇, 等. 10例婴幼儿卡梅综合征临床特征及治疗经验总结[J]. 临床小儿外科杂志, 2019, 18(9): 775-778. doi:10.3969/j.issn.1671-6353.2019.09.014. SHU Ye, HUANG Wenya, LUO Yongqi, et al. Summary of clinical features and treatment protocols of 10 infants with Kasabach-Merritt syndrome[J]. Journal of Clinical Pediatric Surgery, 2019, 18(9): 775-778. doi:10.3969/j.issn.1671-6353.2019.09.014.
[11] 袁斯明, 沈卫民, 王修坤, 等. 11例中国儿童Kasabach-Merritt现象的临床研究[J]. 组织工程与重建外科杂志, 2010, 6(3): 148-152. doi:10.3969/j.issn.1673-0364.2010.03.007. YUAN Siming, SHEN Weimin, WANG Xiukun,et al. Clinical study on 11 cases of kasabach-Merritt phenomenon in Chinese children[J]. Journal of Tissue Engineering and Reconstructive Surgery, 2010, 6(3): 148-152. doi:10.3969/j.issn.1673-0364.2010.03.007.
[12] 李克雷, 姚伟, 秦中平, 等. Kasabach-Merritt现象诊断与治疗中国专家共识[J]. 中国口腔颌面外科杂志, 2019, 17(2): 97-105. doi:10.19438/j.cjoms.2019.02.001. LI Kelei, YAO Wei, QIN Zhongping, et al. Chinese expert consensus on diagnosis and treatment of Kasabach-Merritt phenomenonFull text replacement[J]. China Journal of Oral and Maxillofacial Surgery, 2019, 17(2): 97-105. doi:10.19438/j.cjoms.2019.02.001.
[13] Mazoyer E, Enjolras O, Laurian C, et al. Coagulation abnormalities associated with extensive venous malformations of the limbs: differentiation from Kasabach -Merritt syndrome [J].Clin Lab Haematol, 2002, 24(4):243-251. doi:10.1046/j.1365-2257.2002.00447.x.
[14] 刘磊,陈文嘉,王燕妮,等.16例婴幼儿卡梅现象临床疗效分析[J].现代生物医学进展,2021,21(3):450-454. doi:10.13241/j.cnki.pmb.2021.03.009. LIU Lei, CHEN Wenjia, WANG Yanni, et al. Clinical Analysis of 16 Infants with Kasabach-Merritt Phenomenon[J].Progress in Modern Biomedicine, 2021, 21(3):450-454. doi:10.13241/j.cnki.pmb.2021.03.009.
[15] 赵亚梅. 卡梅现象的诊断与治疗进展[J]. 国际儿科学杂志, 2018, 45(3): 224-227. doi:10.3760/cma.j.issn.1673-4408.2018.03.017. ZHAO Yamei. Recent advances in the diagnosis and treatment of Kasabach-Merritt phenomenon[J]. International Journal of Pediatrics, 2018, 45(3): 224-227. doi:10.3760/cma.j.issn.1673-4408.2018.03.017.
[16] Yao W, Li KL, Qin ZP, et al. Standards of care for Kasabach?傆bMerritt phenomenon in China[J]. World J Pediatr, 2021, 17(2): 123-130. doi:10.1007/s12519-020-00379-9.
[17] Yao W, Li K, Wang Z, et al. Comparison of Corticosteroid and Vincristine in Treating Kaposiform Hemangioendothelioma and Tufted Angioma: A Systematic Review and Meta-Analysis[J]. Eur J Pediatr Surg, 2019, 29(5):401-407. doi:10.1055/s-0038-1673708.
[18] Wang Z, Li K, Yao W, et al. Steroid-resistant kaposiform heman-gioendothelioma: a retrospective study of 37 patients treated withvincristine and long-term follow-up[J]. Pediatr Blood Cancer, 2015, 62(4):577-580. doi:10.1002/pbc.25296.
[19] Adams DM, Trenor CC 3rd, Hammill AM, et al. Efficacy andsafety of sirolimus in the treatment of complicated vascular anomalies[J]. Pediatrics, 2016, 137(2):e20153257. doi:10.1542/peds.2015-3257.
[20] Sakata N, Suenobu SI, Okano M, et al. Impact of sirolimus treatment for refractory kaposiform hemangioendothelioma with exacerbation of the disease 10 years after initial diagnosis[J]. RareTumors, 2018, 10:2036361318776185. doi:10.1177/2036361318776185.
[21] Wang Y, Kong L, Sun B, et al. Sirolimus for Kaposiform Hemangioendothelioma With Kasabach-Merritt Phenomenon in Two Infants[J]. J Craniofac Surg, 2020, 31(4):1074-1077.
[22] Zhang G, Chen H, Gao Y, et al. Sirolimus for treatment of Kaposiform haemangioendothelioma with Kasabach-Merritt phenomenon: a retrospective cohort study[J]. Br J Dermatol, 2018,178(5):1213-1214. doi:10.1111/bjd.16400.
[23] 李英,徐晓琳,刘莹,等.西罗莫司致儿童不良反应的文献分析[J].临床药物治疗杂志,2021,19(9):74-78. doi:10.3969/j.issn.1672-3384.2021.09.015. LI Ying, XU Xiaolin, LIU Ying, et al. Sirolimus-induced adverse reactions in children:a literature analysis[J]. Clinical Medication Journal, 2021, 19(9):74-78. doi:10.3969/j.issn.1672-3384.2021.09.015.
[24] 赵昱,陈晓栋,石力,等. 平阳霉素联合地塞米松注射治疗咽喉血管瘤的疗效观察[J].临床耳鼻咽喉头颈外科杂志, 2014, 18(18):1367-1369. doi:10.13201/j.issn.1001-1781.2014.18.001. ZHAO Yu, CHEN Xiaodong, SHI Li, et al. Clinical observation of Pingyangmycin plus Dexamethasone local injection treatment for pharynx and larynx hemangioma[J]. Journal of Clinical Otorhinolaryngology Head and Neck Surgery, 2014, 18(18):1367-1369. doi:10.13201/j.issn.1001-1781.2014.18.001.

相关文章 15

[1]	张丰珍,王桂香,赵靖,王华,段清川,李宏彬,黑明燕,翁景文,张杰. 新生儿双侧声带麻痹临床特点及预后分析[J]. 山东大学耳鼻喉眼学报, 2022, 36(1): 86-90.
[2]	曾斌, 吕丹, 任佳, 胡娟娟, 于凌昱, 卢欢, 杨慧. 喉显微外科技术在严重新生儿上气道梗阻中的应用[J]. 山东大学耳鼻喉眼学报, 2022, 36(1): 95-99.
[3]	盘琳琳,孔令漪综述翟丰,陈洁审校. 新生儿听力障碍常见危险因素及听力筛查方法研究进展[J]. 山东大学耳鼻喉眼学报, 2022, 36(1): 131-137.
[4]	杨红,滕以书,李兰,潘宏光,梁振江,韩赛红. 新生儿梨状窝瘘3例并文献复习[J]. 山东大学耳鼻喉眼学报, 2021, 35(4): 40-44.
[5]	朱华明,关建,茆松,张维天,易红良. 低温等离子射频消融辅助咽旁间隙良性肿瘤切除的应用分析[J]. 山东大学耳鼻喉眼学报, 2021, 35(2): 28-33.
[6]	孙夏雨,陈建勇综述段茂利,杨军审校. 新生儿婴幼儿前庭功能发育和评估的研究进展[J]. 山东大学耳鼻喉眼学报, 2020, 34(5): 82-88.
[7]	梁刚,马蓉,张丰菊. SMILE术中角膜帽下地塞米松平衡液冲洗与否的早期临床观察[J]. 山东大学耳鼻喉眼学报, 2020, 34(2): 22-31.
[8]	孙臻峰,张佳,苏甜甜,董频. 急性扁桃体炎致甲状腺脓肿、咽旁间隙脓肿一例并文献复习[J]. 山东大学耳鼻喉眼学报, 2016, 30(6): 58-60.
[9]	王雅丛, 耿韶辉, 李艳涛, 王玉芳, 张红, 张婷婷, 张佳佳, 张炜. Retcam-3检查足月新生儿视网膜出血分级的初步探讨[J]. 山东大学耳鼻喉眼学报, 2015, 29(6): 78-81.
[10]	王国强, 赵春红, 霍红, 龚志云, 宋伟伟, 严丽霞, 李雪华, 王伟丽. 早产儿听力筛查结果及其听力障碍危险因素[J]. 山东大学耳鼻喉眼学报, 2015, 29(4): 7-10.
[11]	张宝莹, 卢永田, 李建兴. 口内径路咽旁间隙的内镜解剖结构[J]. 山东大学耳鼻喉眼学报, 2015, 29(4): 38-41.
[12]	江朝勇, 蔡永明, 殷亚磊. 耳后药物注射治疗突发性聋32例[J]. 山东大学耳鼻喉眼学报, 2014, 28(6): 18-20.
[13]	周晓娓, 王跃建, 段光荣, 虞幼军. 听力诊疗网络平台的开发[J]. 山东大学耳鼻喉眼学报, 2014, 28(6): 79-84.
[14]	胡娟娟, 洪育明. 咽旁间隙肿瘤的诊断和治疗[J]. 山东大学耳鼻喉眼学报, 2014, 28(5): 85-90.
[15]	周慧, 黄雪琴, 胡俊丽, 姚俊, 张月飞, 江枫. 黄芩苷对鼻咽癌化疗期间激素使用影响的实验研究[J]. 山东大学耳鼻喉眼学报, 2014, 28(4): 30-32.

多维度评价

Viewed

Full text

Abstract

Cited

Shared

Discussed