使用博莱霉素硬化剂治疗腹股沟淋巴管瘤:病例报告

IF 0.2 Q4 PEDIATRICS
Hien Duy Pham , Duy Le Dinh Tran , Thom Hoang Dang , Son Hoang Nguyen , Vy Huynh Khanh Nguyen , Quang Thanh Nguyen
{"title":"使用博莱霉素硬化剂治疗腹股沟淋巴管瘤:病例报告","authors":"Hien Duy Pham ,&nbsp;Duy Le Dinh Tran ,&nbsp;Thom Hoang Dang ,&nbsp;Son Hoang Nguyen ,&nbsp;Vy Huynh Khanh Nguyen ,&nbsp;Quang Thanh Nguyen","doi":"10.1016/j.epsc.2024.102832","DOIUrl":null,"url":null,"abstract":"<div><h3>Introduction</h3><p>Lymphatic malformations (LMs) or lymphangiomas are rare congenital anomalies affecting the lymphatic system. They are predominantly found in the head and neck, with less common occurrences in abdominal cavity or scrotum. Bleomycin is among the sclerotic agents used, capable of inducing a non-specific inflammatory process leading to thrombosis and fibrosis of the cyst. However, there is limited literature regarding the efficacy of Bleomycin as a standalone treatment for large and mixed types of cystic LMs.</p></div><div><h3>Case presentation</h3><p>A 17-year-old male presented with swelling in the right inguinal and scrotal regions, without tenderness. Ultrasound and contrast-enhanced computed tomography scan (CT scan) revealed a large, well-defined multiloculated cystic lesion measuring 7x8x8cm in the pelvic region, extending into the left inguinoscrotal region (15x6x8cm), consistent with LMs. Treatment comprised aspiration of cystic fluid followed by Bleomycin injection, administered at a maximum dosage of 0.5 IU/kg body weight under general anesthesia. A sequence of five sclerotherapy sessions of Bleomycin injections was carried out, with each session spaced one month apart. Following 15 months of observation, no intra-abdominal cystic masses were identified, and scrotal ultrasound displayed near-complete resolution. Only a fibrotic tissue measuring 1.5 × 1cm in size was identified in the scrotum, and it remained stable across three consecutive scans, indicating that no further intervention was required.</p></div><div><h3>Conclusion</h3><p>Based on this case study, sclerotherapy using intralesional Bleomycin injection for large, complex LMs in children appears to be safe and effective. This scarless approach seems to provide good cosmetic outcomes and poses a minimal risk of neurovascular injury.</p></div>","PeriodicalId":45641,"journal":{"name":"Journal of Pediatric Surgery Case Reports","volume":"106 ","pages":"Article 102832"},"PeriodicalIF":0.2000,"publicationDate":"2024-05-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2213576624000605/pdfft?md5=eb8f37271477e9945c703bcaf843e2ec&pid=1-s2.0-S2213576624000605-main.pdf","citationCount":"0","resultStr":"{\"title\":\"Abdominoscrotal lymphangioma treated with bleomycin sclerotherapy: A case report\",\"authors\":\"Hien Duy Pham ,&nbsp;Duy Le Dinh Tran ,&nbsp;Thom Hoang Dang ,&nbsp;Son Hoang Nguyen ,&nbsp;Vy Huynh Khanh Nguyen ,&nbsp;Quang Thanh Nguyen\",\"doi\":\"10.1016/j.epsc.2024.102832\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<div><h3>Introduction</h3><p>Lymphatic malformations (LMs) or lymphangiomas are rare congenital anomalies affecting the lymphatic system. They are predominantly found in the head and neck, with less common occurrences in abdominal cavity or scrotum. Bleomycin is among the sclerotic agents used, capable of inducing a non-specific inflammatory process leading to thrombosis and fibrosis of the cyst. However, there is limited literature regarding the efficacy of Bleomycin as a standalone treatment for large and mixed types of cystic LMs.</p></div><div><h3>Case presentation</h3><p>A 17-year-old male presented with swelling in the right inguinal and scrotal regions, without tenderness. Ultrasound and contrast-enhanced computed tomography scan (CT scan) revealed a large, well-defined multiloculated cystic lesion measuring 7x8x8cm in the pelvic region, extending into the left inguinoscrotal region (15x6x8cm), consistent with LMs. Treatment comprised aspiration of cystic fluid followed by Bleomycin injection, administered at a maximum dosage of 0.5 IU/kg body weight under general anesthesia. A sequence of five sclerotherapy sessions of Bleomycin injections was carried out, with each session spaced one month apart. Following 15 months of observation, no intra-abdominal cystic masses were identified, and scrotal ultrasound displayed near-complete resolution. Only a fibrotic tissue measuring 1.5 × 1cm in size was identified in the scrotum, and it remained stable across three consecutive scans, indicating that no further intervention was required.</p></div><div><h3>Conclusion</h3><p>Based on this case study, sclerotherapy using intralesional Bleomycin injection for large, complex LMs in children appears to be safe and effective. This scarless approach seems to provide good cosmetic outcomes and poses a minimal risk of neurovascular injury.</p></div>\",\"PeriodicalId\":45641,\"journal\":{\"name\":\"Journal of Pediatric Surgery Case Reports\",\"volume\":\"106 \",\"pages\":\"Article 102832\"},\"PeriodicalIF\":0.2000,\"publicationDate\":\"2024-05-28\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.sciencedirect.com/science/article/pii/S2213576624000605/pdfft?md5=eb8f37271477e9945c703bcaf843e2ec&pid=1-s2.0-S2213576624000605-main.pdf\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Journal of Pediatric Surgery Case Reports\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://www.sciencedirect.com/science/article/pii/S2213576624000605\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q4\",\"JCRName\":\"PEDIATRICS\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Journal of Pediatric Surgery Case Reports","FirstCategoryId":"1085","ListUrlMain":"https://www.sciencedirect.com/science/article/pii/S2213576624000605","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q4","JCRName":"PEDIATRICS","Score":null,"Total":0}
引用次数: 0

摘要

导言淋巴畸形(LMs)或淋巴管瘤是影响淋巴系统的罕见先天性畸形。淋巴畸形主要发生在头颈部,腹腔或阴囊较少见。博莱霉素是常用的硬化剂之一,能诱发非特异性炎症过程,导致囊肿血栓形成和纤维化。然而,有关博莱霉素单独治疗大型和混合型囊性 LMs 的疗效的文献却很有限。病例介绍 一位 17 岁的男性出现右侧腹股沟和阴囊区域肿胀,无压痛。超声波和造影剂增强计算机断层扫描(CT 扫描)显示,骨盆区域有一个 7x8x8 厘米大、轮廓清晰的多发性囊性病变,并延伸至左侧腹股沟区域(15x6x8 厘米),与 LM 相吻合。治疗包括抽吸囊液,然后注射博莱霉素,在全身麻醉的情况下,最大剂量为每公斤体重 0.5 IU。博莱霉素注射硬化剂治疗共进行了五次,每次间隔一个月。经过 15 个月的观察,没有发现腹腔内囊性肿块,阴囊超声波显示几乎完全消退。结论根据本病例研究,使用博莱霉素鞘内注射进行硬化剂注射治疗儿童巨大、复杂的 LM 似乎是安全有效的。这种无疤痕的方法似乎能带来良好的美容效果,而且造成神经血管损伤的风险极低。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Abdominoscrotal lymphangioma treated with bleomycin sclerotherapy: A case report

Introduction

Lymphatic malformations (LMs) or lymphangiomas are rare congenital anomalies affecting the lymphatic system. They are predominantly found in the head and neck, with less common occurrences in abdominal cavity or scrotum. Bleomycin is among the sclerotic agents used, capable of inducing a non-specific inflammatory process leading to thrombosis and fibrosis of the cyst. However, there is limited literature regarding the efficacy of Bleomycin as a standalone treatment for large and mixed types of cystic LMs.

Case presentation

A 17-year-old male presented with swelling in the right inguinal and scrotal regions, without tenderness. Ultrasound and contrast-enhanced computed tomography scan (CT scan) revealed a large, well-defined multiloculated cystic lesion measuring 7x8x8cm in the pelvic region, extending into the left inguinoscrotal region (15x6x8cm), consistent with LMs. Treatment comprised aspiration of cystic fluid followed by Bleomycin injection, administered at a maximum dosage of 0.5 IU/kg body weight under general anesthesia. A sequence of five sclerotherapy sessions of Bleomycin injections was carried out, with each session spaced one month apart. Following 15 months of observation, no intra-abdominal cystic masses were identified, and scrotal ultrasound displayed near-complete resolution. Only a fibrotic tissue measuring 1.5 × 1cm in size was identified in the scrotum, and it remained stable across three consecutive scans, indicating that no further intervention was required.

Conclusion

Based on this case study, sclerotherapy using intralesional Bleomycin injection for large, complex LMs in children appears to be safe and effective. This scarless approach seems to provide good cosmetic outcomes and poses a minimal risk of neurovascular injury.

求助全文
通过发布文献求助,成功后即可免费获取论文全文。 去求助
来源期刊
CiteScore
0.60
自引率
25.00%
发文量
348
审稿时长
15 days
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
确定
请完成安全验证×
copy
已复制链接
快去分享给好友吧!
我知道了
右上角分享
点击右上角分享
0
联系我们:info@booksci.cn Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。 Copyright © 2023 布克学术 All rights reserved.
京ICP备2023020795号-1
ghs 京公网安备 11010802042870号
Book学术文献互助
Book学术文献互助群
群 号:481959085
Book学术官方微信