目的:单性眼睑幼年黄色肉芽肿(JXG)极为罕见,关于其临床特征和治疗结果的文献有限.这里,我们提供了一个病例系列和文献的综合综述。
方法:我们从我们机构的孤立眼睑JXG病例记录中系统地提取数据,并利用PubMed从文献中搜索其他病例,万方,和中国国家知识基础设施(CNKI)数据库。对JXG患者的年龄进行了分析,性别,临床表现,治疗,和结果。进行组比较。
结果:确定了32例患者(包括我们机构的13例和以前出版物的19例)。当前患者首次就诊时的中位年龄高于已发表病例的患者(中位年龄为9岁,范围为1.2至47.0年;中位数为2年,0.5个月到46.0年,分别,P=0.014)。在具有已知特征的患者中,两组在性别方面没有观察到显著差异,受影响的眼睛,眼睑部位,皮肤受累的类型,或症状持续时间(均P>0.05)。17例(54.8%)患者为男性。最常见的病变位置是上眼睑(n=10,62.5%)。二十四(75.0%)个皮肤病变有全层皮肤受累;8(25.0%)个皮下肿块有类似的外观。组织学上,JXG肿块的特征是Touton巨细胞和炎性细胞。此外,两组治疗方式差异无统计学意义(P=0.072),24例(75.0%)患者接受手术切除。当前患者的总无复发生存期为3.6至52.8个月(中位数27.0)。对于有可用随访信息的已发表病例,10例无复发,1例无好转,中位随访时间为9.5个月。
结论:孤立性眼睑JXG是一种罕见的临床实体,应包括在所有年龄段患者的眼睑肿块病变的鉴别诊断中。通常选择手术切除以进行有效治疗并获得切除活检。
OBJECTIVE: Solitary eyelid juvenile xanthogranuloma (JXG) is extremely rare, and there is limited literature on its clinical features and treatment outcomes. Here, we present a case series and comprehensive
review of the literature on patients with isolated eyelid JXG.
METHODS: We systematically extracted data from our institution\'s records of isolated eyelid JXG cases and conducted a search for additional cases from the literature utilising the PubMed, Wanfang, and Chinese National Knowledge Infrastructure (CNKI) databases. Patients with JXG were analysed with respect to age, sex, clinical presentation, therapy, and outcome. Group comparisons were performed.
RESULTS: Thirty-two patients (including 13 at our institution and 19 from prior publications) were identified. The median age at first presentation was higher in current patients than in the patients from the published cases (median 9 years, range 1.2 to 47.0 years; median 2 years, range 0.5 months to 46.0 years, respectively, P = 0.014). Of the patients who had known characteristics, no significant differences were observed between the two groups in terms of sex, affected eye, eyelid site, type of cutaneous involvement, or duration of symptoms (each P > 0.05). Seventeen (54.8%) patients were male. The most common lesion location was the upper eyelid (n = 10, 62.5%). Twenty-four (75.0%) cutaneous lesions had full-thickness skin involvement; 8 (25.0%) subcutaneous masses had a chalazion-like appearance. Histologically, the JXG masses were characterised by Touton giant cells with inflammatory cells. Additionally, there was no significant difference in treatment modalities between the two groups (P = 0.072), and 24 (75.0%) patients underwent surgical excision. The overall recurrence-free survival was 3.6 to 52.8 (median 27.0) months in the current patients. For published cases with available follow-up information, there was no recurrence in 10 cases and improvement in 1 case, with a median follow-up of 9.5 months.
CONCLUSIONS: Solitary eyelid JXG is a rare clinical entity and should be included in the differential diagnosis of eyelid mass lesions in patients of all age groups. Surgical excision is often selected for efficient treatment and to obtain an excisional biopsy.