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膝关节进行性双侧树枝状脂肪瘤病合并幼年脊柱关节病:病例报告及文献复习。

Progressive bilateral lipoma arborescens of the knee complicated by juvenile spondyloarthropathy: a case report and review of the literature.

机构信息

Department of Rheumatology, Second Affiliated Hospital, Zhejiang University, School of Medicine, Hangzhou, P.R. China.

出版信息

Semin Arthritis Rheum. 2013 Oct;43(2):259-63. doi: 10.1016/j.semarthrit.2012.12.022. Epub 2013 Jan 24.

DOI:10.1016/j.semarthrit.2012.12.022
PMID:23352250
Abstract

OBJECTIVES

To report an unusual case of lipoma aborescens (LA) presented in a patient with treatment-responsive juvenile spondyloarthropathy (JSPA) and to summarize the clinical manifestations, therapy and prognosis of LA by literature review.

METHODS

We report an atypical case of a 17-year-old patient with an initial presentation of juvenile spondyloarthropathy, whose inflammatory condition was improved successfully by traditional anti-rheumatic drugs and an anti-TNF alpha agent but developed progressive swelling of bilateral knees. Lipoma arborescens were diagnosed in each knee by synovial biopsy obtained by arthroscopic surgery. Fifty-one cases of LA have been reported and are reviewed in detail.

RESULTS

Clinically, LA could present as monoarthritis or oligoarthritis. The lateral compartment of the knee is the most common site of involvement. Several cases were reported as a comorbidity of inflammatory diseases, but were not improved by anti-inflammatory therapy. Most patients were diagnosed by classic MRI and biopsy findings. The lesions can be managed by open or arthroscopic surgery, but a minority of the cases may have reoccurrence in the same or opposite joint.

CONCLUSIONS

LA is a very rare lesion of the synovial and bursal tissue with an unknown etiology. It is considered to be a benign proliferation of the synovial fat associated with trauma, degenerative or inflammatory conditions. LA should be considered as a secondary or comorbid condition in inflammatory arthropathies if other joints respond well to intensive therapy and one or more do not.

摘要

目的

报告一例发生于对治疗有反应的青少年脊柱关节病(JSPA)患者的罕见增生性脂膜炎(LA)病例,并通过文献复习总结 LA 的临床表现、治疗和预后。

方法

我们报告了一例 17 岁患者的不典型病例,该患者最初表现为青少年脊柱关节病,其炎症状况通过传统抗风湿药物和抗 TNF-α 药物成功改善,但双侧膝关节逐渐肿胀。通过关节镜手术获得的滑膜活检诊断为每个膝关节均存在增生性脂膜炎。详细回顾并分析了 51 例 LA 病例。

结果

临床上,LA 可表现为单关节炎或寡关节炎。膝关节外侧间室是最常见的受累部位。有几例报告为炎症性疾病的合并症,但抗炎治疗无效。大多数患者通过典型 MRI 和活检发现诊断为 LA。病变可通过开放性或关节镜手术治疗,但少数病例在同一或对侧关节可能会复发。

结论

LA 是一种非常罕见的滑膜和滑囊组织病变,病因不明。它被认为是与创伤、退行性或炎症性疾病相关的滑膜脂肪良性增生。如果其他关节对强化治疗反应良好而一个或多个关节没有反应,则应将 LA 视为炎症性关节病的继发或合并症。

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