Amyloid arthropathy with joint flexion contracture as the initial manifestation: a report of 5 cases and literature review

Wan Yue-Hua; Yang Jin-Shui; Zhao Yu-Rong; Li Kun-Peng; Zhao Zheng; Jin Jing-Yu; Zhu Jian

doi:10.11855/j.issn.0577-7402.2252.2023.0818

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Amyloid arthropathy with joint flexion contracture as the initial manifestation: a report of 5 cases and literature review

Clinical Research | 更新时间：2024-01-04

- Amyloid arthropathy with joint flexion contracture as the initial manifestation: a report of 5 cases and literature review
- Medical Journal of Chinese People′s Liberation Army Vol. 48, Issue 12, Pages: 1451-1456(2023)
- 作者机构：
  
  解放军总医院第一医学中心风湿免疫科，北京　100853
- 作者简介：
  
  E-mail: jian_jzhu@126.com
- 基金信息：
- DOI：10.11855/j.issn.0577-7402.2252.2023.0818
  CLC：
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万月华, 杨金水, 赵玉荣, 等. 以关节屈曲挛缩为首发表现的淀粉样关节病5例并文献复习[J]. 解放军医学杂志, 2023, 48(12): 1451-1456.

Wan Yue-Hua,Yang Jin-Shui,Zhao Yu-Rong,et al.Amyloid arthropathy with joint flexion contracture as the initial manifestation: a report of 5 cases and literature review[J].Medical Journal of Chinese People′s Liberation Army,2023,48(12):1451-1456.
万月华, 杨金水, 赵玉荣, 等. 以关节屈曲挛缩为首发表现的淀粉样关节病5例并文献复习[J]. 解放军医学杂志, 2023, 48(12): 1451-1456. DOI： 10.11855/j.issn.0577-7402.2252.2023.0818.

Wan Yue-Hua,Yang Jin-Shui,Zhao Yu-Rong,et al.Amyloid arthropathy with joint flexion contracture as the initial manifestation: a report of 5 cases and literature review[J].Medical Journal of Chinese People′s Liberation Army,2023,48(12):1451-1456. DOI： 10.11855/j.issn.0577-7402.2252.2023.0818.

摘要

目的,2,报道5例以关节屈曲挛缩为首发表现的淀粉样关节病病例，并进行文献复习，以提高对该病的认识。,方法,2,收集2012年1月－2022年2月就诊于解放军总医院第一医学中心风湿免疫科的、以关节屈曲挛缩为首发表现的、免疫球蛋白轻链(AL)型淀粉样变性相关的淀粉样关节病患者的临床资料进行回顾性分析，检索中国知网、万方数据知识服务平台、PubMed数据库，结合文献报道总结AL型淀粉样变性相关的淀粉样关节病的临床表现、实验室及影像学检查结果。,结果,2,通过病例回顾，本中心共有5例以关节屈曲挛缩为首发表现的淀粉样关节病患者，其中男4例，女1例。检索中国知网、万方数据知识服务平台、PubMed数据库(截至2022年2月)，筛选出2例符合条件的患者，均为男性。共纳入7例，男女比例6∶1，中位年龄30岁(19~73岁)。脊柱及四肢大小关节均受累，尤其是手关节(7例，100%)。共有5例(71.4%)出现软组织受累：腕管综合征4例，巨舌3例，肩垫征2例，其他部位肌肉肿胀2例。5例被误诊为类风湿关节炎(RA)，1例被误诊为脊柱关节炎(SpA)，延迟诊断的中位时间为24(2~204)个月。实验室检查结果发现，3例(42.9%)免疫电泳阳性，均为λ轻链。仅有1例人类白细胞抗原B27(HLA-B27)阳性，其余患者的自身抗体及类风湿因子均为阴性。仅1例的C反应蛋白(CRP)和红细胞沉降率(ESR)升高。5例(71.4%)病变部位的超声检查可见关节周围低回声区，韧带增厚，或关节周围腱鞘炎症。5例(71.4%)完善了X线检查，1例可见软组织肿胀伴糜烂及软骨下囊肿，其余4例未发现骨质破坏。5例(71.4%)完善了磁共振成像检查，其中1例关节周围肌肉内可见T,1,加权像低信号、T,2,加权像中等信号，2例可见周围腱鞘水肿信号。1例(14.3%)行全身骨核素扫描(ECT)未见异常摄取。,结论,2,关节屈曲挛缩可为淀粉样关节病的首发表现。当合并软组织受累，血清学阴性，影像未见关节破坏但出现周围软组织异常时，需警惕淀粉样关节病。

Abstract

Objective,2,To report 5 cases of amyloid arthropathy with joint flexion contracture as the first manifestation, and perform a literature review to improve the understanding of the disease.,Methods,2,Retrospective analysis of the clinical manifestations, laboratory and imaging results clinical data of patients with immunoglobulin light chain (AL) amyloidosis related amyloid arthropathy with joints flexion contracture as the first manifestation at the Department of Rheumatology and Immunology, the First Medical Center of People's Liberation Army General Hospital from January 2012 to February 2022. Meanwhile searching the database (CNKI, Wanfang Data, PubMed), excluding myeloma-associated amyloid arthropathy, and analyzing the literature results comprehensively.,Results,2,There were a total of 5 patients with amyloid arthropathy with joint flexion contracture as the initial manifestation in this center, including 4 males and 1 female. By February 2022 (searching CNKI, Wanfang Data and PubMed), two eligible patients were selected, all males. A total of 7 patients were included with male-to-female ratio of 6:1. The median age was,30-year-old (range from 19 to 73). Spine and limb joints were involved, especially finger joints (7 cases, 100%). Soft tissue was involved in 5 patients (71.4%), carpal tunnel syndrome in 4 patients (57.1%), giant tongue in 3 patients (42.9%), shoulder pad sign in 2 patients (28.6%), muscle swelling in 2 patients (28.6%). Five patients were misdiagnosed as rheumatoid arthritis (RA) and 1 patient was misdiagnosed as spondyloarthritis (SpA). The median time of delayed diagnosis was 24 months (2-204 months). The laboratory results showed that 3 patients (42.9%) were positive for immunoelectrophoresis, all of which were λ light chain. Only 1 patient was positive for HLA-B27, while the other patients were negative for autoantibodies and rheumatoid factors. Only 1 patient had elevated C reactive protein (CRP) and erythrocyte sedimentation rate (ESR). Ultrasound examination of the lesion site of 5 patients (71.4%) showed hypoechoic area around the joint, thickening of ligaments or periarticular tenosynovitis. Five patients (71.4%) underwent,X-ray examination, of which 4 patients had no bone destruction, 1 patient had soft tissue swelling with bone erosion and subchondral cyst. MRI was performed in 5 patients (71.4%), in which low signal on T,1, weighted image, medium signal on T,2, weighted image and edema signal on peripheral tendon sheath were seen in 1 patient. One patient (14.3%) underwent emission computed tomography (ECT) without abnormal uptake.,Conclusion,2,Joint flexion contracture may be the first sign of amyloid arthropathy. Vigilance for amyloid arthropathy is warranted when there is associated soft tissue involvement, negative autoantibodies, and imaging showing no joint destruction but surrounding soft tissue abnormalities.

关键词

关节挛缩淀粉样关节病AL型淀粉样变性

Keywords

joint contractureamyloid arthropathyAL amyloidosis

references

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