2种病变内平阳霉素注射方法治疗68例儿童唇面部微囊型淋巴管畸形疗效分析

doi:10.19438/j.cjoms.2022.03.011

摘要/Abstract

摘要： 目的: 总结儿童唇面部微囊型淋巴管畸形（lymphatic malformation,LM）2种病变内平阳霉素注射方法治疗的远期疗效和安全性。方法: 回顾分析1990年1月—2013年1月临沂市肿瘤医院收治的68例儿童唇面部弥漫性微囊型LM病例。32例患儿（A组）采用同期、全层病变内平阳霉素注射治疗,即经黏膜、黏膜下层病变内注射平阳霉素并同期行唇面部全层病变内平阳霉素注射治疗;36例患儿（B组）行分期、分层次病变内平阳霉素注射治疗,即先期行经黏膜、黏膜下层病变内平阳霉素注射治疗,黏膜面病变基本消失后再行经皮、面颊部浅层病变内平阳霉素注射治疗。疗效评价采用Achauer 4级标准。记录疗程,平阳霉素总剂量,观察远期局部凹陷萎缩并发症。采用SPSS 18.0软件包对数据进行统计学分析。结果: 随访5~25年,A组病灶评价疗效Ⅰ级0例,Ⅱ级0例,Ⅲ级4例,Ⅳ级28例;B组病灶评价疗效Ⅰ级0例,Ⅱ级0例,Ⅲ级5例,Ⅳ级31例。2组Ⅳ级疗效比较无统计学差异（P>0.05）。A组疗程（5.9±1.4）次,B组疗程（6.4±1.3）次,2组比较无统计学差异（P>0.05）。A组注射平阳霉素总剂量（44.1±8.5）mg,B组注射平阳霉素总剂量（48.2±10.6）mg,2组比较无统计学差异（P>0.05）。A组20例（62.5%）出现局部凹陷萎缩,B组1例（2.8%）出现萎缩凹陷,2组差异有显著性（P<0.05）。结论: 采用分期、分层病变内平阳霉素注射治疗,与同期、全层病变内平阳霉素注射治疗儿童唇面部微囊型淋巴管畸形相比,其疗效、疗程和平阳霉素总剂量相当,但远期局部组织萎缩凹陷并发症发生率显著降低,治疗后面部外形好,患者满意度高。

关键词: 淋巴管畸形, 微囊型, 颌面部, 平阳霉素

Abstract: PURPOSE: To summarize the long-term clinical experience of two methods of intralesional injection of pingyangmycin for the treatment of microcystic lymphatic malformation (LM) in children's lip and face. METHODS: From January 1990 to January 2013, a total of 68 children with diffuse microcystic LM were admitted and treated. All patients received intralesional injection of pingyangmycin, and a total of 68 cases were followed up to the age of 18 years. Among 68 patients, 37 were male and 31 were female. The age at consultation ranged from 10 months to 11 years, and the median age was 3.5 years. From January 1990 to December 1998, 32 children (group A) were treated with full-layer intralesional injection of pingyangmycin at one time. From January 1999 to January 2013, 36 children (group B) were treated with in-layers intralesional injection of pingyangmycin at different times, first mucosal, and submucosa injection followed by percutaneous injection of face and lip after mucosal lesions disappeared. The therapeutic effect was evaluated according to a 4-grade system. The course of treatment, total dose of pingyangmycin were recorded, and long-term local complications were observed. SPSS 18.0 software package was used for statistical analysis. RESULTS: The patients were followed up for 5 to 25 years, there were 4 cases of grade Ⅲ and 28 cases of grade Ⅳ in group A, and 5 cases of grade Ⅲ and 31 cases of grade Ⅳ in group B. By Fisher's exact test, there was no significant difference in the efficacy rate of grade Ⅳ between group A and group B (P>0.05). The treatment course of group A was(5.9±1.4) times, and that of group B was (6.4±1.3) times. The total dose of pingyangmycin was (44.1±8.5) mg in group A and (48.2±10.6) mg in group B, respectively. In group A, 20 cases (62.5%) showed local atrophy and depression, and in group B, 1 case (2.8%) showed atrophy and depression. CONCLUSIONS: Compared with full-layer intralesional injection of pingyangmycin at one time in the treatment of microcystic lymphatic malformation, the incidence of complications of local atrophy and depression in the long term was significantly reduced when using in-layer injection at different times. After treatment, the facial appearance was good and the patients’ satisfaction was high.

Key words: Lymphatic malformation, Microcystic type, Maxillofacial region, Pingyangmycin

中图分类号:

R654.7

邰茂众, 陈涛, 李克雷, 葛春晓, 徐振国, 秦中平. 2种病变内平阳霉素注射方法治疗68例儿童唇面部微囊型淋巴管畸形疗效分析[J]. 中国口腔颌面外科杂志, 2022, 20(3): 268-272.

TAI Mao-zhong, CHEN Tao, LI Ke-lei, GE Chun-xiao, XU Zhen-guo, QIN Zhong-ping. Comparison of two methods of intralesional injection of pingyangmycin for the treatment of microcystic lymphatic malformations in children's lip, face[J]. China J Oral Maxillofac Surg, 2022, 20(3): 268-272.

参考文献

[1] Matsudera S, Sato-Yazawa H, Terada M, et al.Histopathological evaluation of the effectiveness of oral Eppikajutsuto treatment for lymphatic malformation[J]. J Pediatr Surg, 2021, 56(9): 1668-1672.
[2] Gupta A, Sardana K, Arora P, et al.Microcystic lymphatic malformation in a child and his mother[J]. Pediatr Dermatol, 2019, 36(6): 967-969.
[3] McCormack L, Jones K, Huang JT. Micro- and macrocystic lymphatic malformation[J]. J Pediatr, 2020, 219: 275-276.
[4] 刘文英. 淋巴管畸形的诊治进展[J]. 中华妇幼临床医学杂志(电子版), 2012, 8(6): 573-575.
Liu WY.Progress in diagnosis and treatment of lymphatic malformation[J]. Chinese Journal of Obstetrics & Gynecology and Pediatrics(Electronic Edition), 2012, 8(6): 573-575.
[5] Upadhyaya VD, Bhatnagar A, Kumar B, et al.Is multiple session of intralesional bleomycin mandatory for complete resolution of macrocystic lymphatic malformation?[J]. Indian J Plast Surg, 2018, 51(1): 60-65.
[6] Achauer BM, Chang CJ, Vander Kam VM.Management of hemangioma of infancy: review of 245 patients[J]. Plast Reconstr Surg, 1997, 99(5): 1301-1308.
[7] Li J, Zhong W, Geng X, et al.Ultrasonographic diagnosis, classification, and treatment of cervical lymphatic malformation in paediatric patients: a retrospective study[J]. BMC Pediatr, 2020, 20(1): 441-447.
[8] Anggreyni G, Agustriani N, Agustriani N, et al.Comparison of different therapeutic approaches for children with common lymphatic malformation[J]. Med J Malaysia, 2020, 75(Suppl 1): 32-36.
[9] Kim CM, Vahabzadeh-Hagh AM, Luu QC.Failure of multiple imaging modalities to diagnose lymphatic malformation of the tongue[J]. Ear Nose Throat J, 2020, 99(5): 309-310.
[10] Ueno S, Fujino A, Morikawa Y, et al.Indications for tracheostomy in children with head and neck lymphatic malformation: analysis of a nationwide survey in Japan[J]. Surg Today, 2019, 49(5): 410-419.
[11] Parashar G, Shankar G, Sahadev R, et al.Intralesional sclerotherapy with bleomycin in lymphatic malformation of tongue an institutional experience and outcomes[J]. J Indian Assoc Pediatr Surg, 2020, 25(2): 80-84.
[12] Honnorat M, Viremouneix L, Ayari S, et al.Early adjuvant medication with the mTOR inhibitor sirolimus in a preterm neonate with compressive cystic lymphatic malformation[J]. Front Pediatr, 2020, 8: 418-422.
[13] Giacalone G, Yamamoto T, Belva F, et al.The application of virtual reality for preoperative planning of lymphovenous anastomosis in a patient with a complex lymphatic malformation[J]. J Clin Med, 2019, 8(3): 371-377.
[14] Laviv A, Maly A, Abu-Tair J.Large facial lymphatic malformation treatment using sclerosing agent followed by surgical resection: clinical and pathology reportetal[J]. Oral Maxillofac Surg, 2017, 21(3): 363-369.
[15] Savas JA, Ledon J, Franca K, er al. Carbon dioxide laser for the treatment of microcystic lymphatic malformations (lymphangioma circumscriptum): a systematic review[J]. Dermatol Surg, 2013, 39(8): 1147-1157.
[16] Lisan Q, Villepelet A, Parodi M, et al.Value of radiofrequency ablation in the management of retropharyngeal lymphatic malformation[J]. Int J Pediatr Otorhinolaryngol, 2016, 83(1): 37-40.
[17] Liviskie CJ, Brennan CC, McPherson CC, et al. Propranolol for the treatment of lymphatic malformations in a neonate - a case report and review of literature[J]. J Pediatr Pharmacol Ther, 2020, 25(2): 155-162.
[18] Ghariani Fetoui N, Boussofara L, Gammoudi R, et al.Efficacy of sirolimus in the treatment of microcystic lymphatic malformation of the tongue[J]. J Eur Acad Dermatol Venereol, 2019, 33(9): e336-e337.
[19] Basaran MM, Karatayli SÖ, Pampal A, et al.Successful treatment of a case with cervical lymphatic malformation: repeated bleomycin sclerotherapy[J]. Turk Arch Otorhinolaryngol, 2017, 55(4): 180-183.
[20] Bhatnagar A, Upadhyaya VD, Kumar B, et al.Aqueous intralesional bleomycin sclerotherapy in lymphatic malformation: our experience with children and adult[J]. Natl J Maxillofac Surg, 2017, 8(2): 130-135.
[21] 李克雷, 王玉平, 陈涛, 等. 舌微囊型淋巴管畸形分型及疗效的回顾性分析[J].中华口腔医学杂志, 2019, 54(5): 303-308.
Li KL, Wang YP, Chen T, et al.Retrospective analysis of classification and treatment of microcystic lymphatic malformations of tongue[J]. Chinese Journal of Stomatology, 2019, 54(5): 303-308.
[22] 景纯, 温玉明, 王大章, 等. 人体口腔颊、舌部淋巴管的结构特点及分布与癌转移[J]. 华西口腔医学杂志, 1995, 13(2): 125-129,146.
Jing C, Wen YM, Wang DZ, et al.Buccal and lingual cancer metastasis to structural features and distribution of the lymphatics[J]. West China Journal of Stomatology, 1995, 13(2): 125-129,146.