山东大学耳鼻喉眼学报 ›› 2022, Vol. 36 ›› Issue (4): 55-59.doi: 10.6040/j.issn.1673-3770.0.2021.410
朱晶,张睿,赵媛,李炀,赵昱
ZHU Jing, ZHANG Rui, ZHAO Yuan, LI Yang, ZHAO Yu
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。
中图分类号:
| [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. |
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