Journal of Otolaryngology and Ophthalmology of Shandong University ›› 2022, Vol. 36 ›› Issue (4): 55-59.doi: 10.6040/j.issn.1673-3770.0.2021.410

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Newborn with Kasabach-Merritt phenomenon in parapharyngeal space:a case report and literature review

ZHU Jing, ZHANG Rui, ZHAO Yuan, LI Yang, ZHAO Yu   

  1. Department of Otorhinolaryngology & Head and Neck Surgery, Xi'an Childrens's Hospital, Xi'an 710003, Shaanxi, China
  • Published:2022-07-11

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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

CLC Number: 

  • R766.9
[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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