Rui He, Nan Zhang, Wu Guo, Lin Ma, Zigang Xu, Bin Zhang
{"title":"Unilateral naevoid telangiectasia: A retrospective study of 11 paediatric cases.","authors":"Rui He, Nan Zhang, Wu Guo, Lin Ma, Zigang Xu, Bin Zhang","doi":"10.25259/IJDVL_13_2024","DOIUrl":null,"url":null,"abstract":"<p><p>Background Unilateral naevoid telangiectasia (UNT) is a rare disease with only sporadic cases reported. The pathogenesis remains elusive and especially in paediatric patients, effective and safe treatment is still uncertain. Objectives The purpose of this study was to summarise the clinical characteristics of UNT, explore the possible pathogenesis and evaluate the efficacy and safety of pulsed dye laser (PDL) therapy. Materials and Methods The epidemiological data, clinical manifestations, laboratory tests and pathological features of paediatric patients with UNT were retrospectively reviewed. PDL treatment was done on some of the patients. Clinical documents and patient images before and after treatment were assessed to evaluate efficacy and adverse events. Results Most of the cases (9/11) presented with unilateral lesions. The laboratory results of all the 11 cases were normal. Histological examination in six cases revealed multiple, dilated veins in the reticular dermis. Vascular endothelial growth factor (VEGF) staining was positive, whereas oestrogen receptor staining was negative. Nine cases were treated with PDL which was shown to be effective and safe. Conclusion UNT has typical clinical manifestations. The pathogenesis of this disease could be linked to VEGF; however, more research and confirmation are needed. PDL is an effective and safe treatment for UNT.</p>","PeriodicalId":50376,"journal":{"name":"Indian Journal of Dermatology Venereology & Leprology","volume":" ","pages":"1-6"},"PeriodicalIF":3.2000,"publicationDate":"2024-07-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Indian Journal of Dermatology Venereology & Leprology","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.25259/IJDVL_13_2024","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q2","JCRName":"DERMATOLOGY","Score":null,"Total":0}
引用次数: 0
Abstract
Background Unilateral naevoid telangiectasia (UNT) is a rare disease with only sporadic cases reported. The pathogenesis remains elusive and especially in paediatric patients, effective and safe treatment is still uncertain. Objectives The purpose of this study was to summarise the clinical characteristics of UNT, explore the possible pathogenesis and evaluate the efficacy and safety of pulsed dye laser (PDL) therapy. Materials and Methods The epidemiological data, clinical manifestations, laboratory tests and pathological features of paediatric patients with UNT were retrospectively reviewed. PDL treatment was done on some of the patients. Clinical documents and patient images before and after treatment were assessed to evaluate efficacy and adverse events. Results Most of the cases (9/11) presented with unilateral lesions. The laboratory results of all the 11 cases were normal. Histological examination in six cases revealed multiple, dilated veins in the reticular dermis. Vascular endothelial growth factor (VEGF) staining was positive, whereas oestrogen receptor staining was negative. Nine cases were treated with PDL which was shown to be effective and safe. Conclusion UNT has typical clinical manifestations. The pathogenesis of this disease could be linked to VEGF; however, more research and confirmation are needed. PDL is an effective and safe treatment for UNT.
背景单侧无色素性毛细血管扩张症(UNT)是一种罕见疾病,仅有散发性病例报道。该病的发病机制至今仍难以确定,尤其是对儿童患者而言,有效而安全的治疗方法仍不确定。本研究的目的是总结 UNT 的临床特征,探讨可能的发病机制,并评估脉冲染料激光疗法(PDL)的有效性和安全性。材料和方法 回顾性研究了儿童 UNT 患者的流行病学数据、临床表现、实验室检查和病理特征。对部分患者进行了 PDL 治疗。对治疗前后的临床文件和患者图像进行评估,以评价疗效和不良反应。结果 大多数病例(9/11)为单侧病变。所有 11 例患者的化验结果均正常。6例病例的组织学检查显示,网状真皮层中存在多条扩张的静脉。血管内皮生长因子(VEGF)染色呈阳性,而雌激素受体染色呈阴性。九例患者接受了 PDL 治疗,结果显示该疗法有效且安全。结论 UNT 具有典型的临床表现。该病的发病机制可能与血管内皮生长因子有关,但还需要更多的研究和证实。PDL 是治疗 UNT 有效而安全的方法。
期刊介绍:
The Indian Association of Dermatologists, Venereologists & Leprologists (IADVL) is the national association of Indian medical specialists who manage patients with skin disorders, sexually transmitted infections (STIs) or leprosy. The current member strength of the association is about 3800. The association works for the betterment of the specialty by holding academic meetings, printing a journal and publishing a textbook. The IADVL has several state branches, each with their own office bearers, which function independently within the constitution of the IADVL.
Established in 1940, the Indian Journal of Dermatology, Venereology and Leprology (IJDVL, ISSN 0378-6323) is the official publication of the IADVL (Indian Association of Dermatologists, Venereologists and Leprologists).