多西环素注射治疗儿童大囊型和混合型囊性淋巴管畸形的疗效与安全性探讨

Efficacy and safety of doxycycline injection for marcocystic and mixed-cystic lymphatic malformations in children

目的探讨多西环素注射治疗儿童大囊型和混合型囊性淋巴管畸形的有效性及安全性。方法回顾性分析2018年9月至2020年12月上海市儿童医院/上海交通大学附属儿童医院普外科收治,并进行多西环素注射治疗的13例大囊型和混合型囊性淋巴管畸形患儿临床资料。患儿均经术前超声及MRI明确诊断和分类。其中男6例,女7例;年龄(3.9±3.6)岁(3个月至13岁)。均全身麻醉后在超声/腹腔镜引导下穿刺、引流囊液,并注射多西环素。术后透视评估囊腔范围,并确认药物无溢出。术后4~6周进行初次随访。收集术前、术后影像学资料评价疗效,将疗效分为治愈(体积减小>90%)、有效(体积减小>50%)、部分有效(体积减小<50%)、无效(体积减小<10%或体积无减小)。如第一次注射后疗效未达到有效标准,则予重复注射。若再次注射后仍疗效欠佳,则予手术治疗。结果13例中,病变位于头颈部7例,腋下1例,上臂1例,胸壁1例,腹膜后2例,腘窝1例;其中1例有头颈部手术史。随访时间(9±6.17)个月(4~27个月)。总有效率为100%,治愈率为69.2%。部分患儿术后出现肿胀、疼痛或呕吐,均自行缓解或经对症治疗后缓解;1例出现囊内出血,予止血治疗后好转。13例均未出现四环素牙或神经损伤。结论多西环素注射治疗大囊型和混合型囊性淋巴管畸形效果满意,不良反应较少。

Objective To explore the efficacy and safety of doxycycline injection for macrocystic and mixed-cystic lymphatic malformations in children.Methods From September 2018 to December 2020,retrospective review of clinical data was performed for 13 children hospitalized with marcocystic and mixed-cystic lymphatic malformations receiving doxycycline injection.There were 6 boys and 7 girls with an average age of(3.9±3.6)(0.25-13)years.Preoperative ultrasound and magnetic resonance imaging(MRI)confirmed the diagnosis and classification.After general anesthesia,cyst was punctured,fluid drained and drug injected under ultrasound/laparoscopic guidance.Fluoroscopy was performed after injection to further evaluate the range of cysts and confirm that there was no drug overflow.During an initial follow-up period of 4-6 weeks after injection,physical and imaging examinations were performed.Based upon a comparison of preoperative and postoperative imaging data,the clinical responses were characterized as cure(volume reduction>90%or surface invisible),effective(volume reduction>50%),partially effective(volume reduction<50%)and no response.If the efficacy failed to reach the effective standard(no response or no change in appearance),protocol was repeated.Surgery was performed if re-injection remained ineffective.Results The lesions were located in head and neck(n=7),armpit(n=1),upper arm(n=1),chest wall(n=1),retroperitoneum(n=2)and popliteal fossa(n=1).One child had an operative history of head and neck.There was an average of 19(1.46±0.77)injections.The average follow-up period was(9±6.17)(4-27)months.The overall effective and cure rate was 100%and 69.2%respectively.Swelling,pain and vomiting after injection were relieved spontaneously or symptomatically.One case of hemorrhage in cyst improved after hospitalization for hemostasis.None of them had tetracycline tooth or nerve damage.Conclusion Doxycycline injection is effective for macrocystic and mixed-cystic lymphatic malformations with few adverse reactions.