疑难/罕见病例分析

类风湿关节炎合并色素沉着绒毛结节性滑膜炎1例及文献回顾

  • 彭嘉婧 ,
  • 刘田
展开
  • 北京大学人民医院风湿免疫科,北京 100044

收稿日期: 2020-07-25

  网络出版日期: 2020-12-13

Rheumatoid arthritis combined with pigmented villonodular synovitis: A case report and literature review

  • Jia-jing PENG ,
  • Tian LIU
Expand
  • Department of Rheumatology and Immunology, Peking University People’s Hospital, Beijing 100044, China

Received date: 2020-07-25

  Online published: 2020-12-13

摘要

报道北京大学人民医院风湿免疫科诊治的1例类风湿关节炎(rheumatoid arthritis, RA)合并色素沉着绒毛结节性滑膜炎(pigmented villonodular synovitis, PVNS)的临床诊疗过程,并回顾国内外相关文献报道,对该病的诊断、治疗及预后进行综合分析,总结疾病特点,提高对该病的认识,减少漏诊并促进及早诊断。本例患者为45岁女性,RA病史15年,未进行规律治疗,近3个月右膝关节肿痛加重,关节腔穿刺发现大量RA病情难以解释的血性关节液。膝关节磁共振 (magnetic resonance imaging, MRI)提示T1及T2加权像均为低信号的含铁血黄素沉着。患者行膝关节镜检查,镜下表现及组织病理检查符合PVNS的诊断,给予患者切除增生滑膜。术后给予患者积极的功能锻炼及慢作用抗风湿药物规范治疗,随访半年恢复良好,右膝关节未再出现肿胀。RA是以慢性滑膜炎和关节破坏为特征的自身免疫疾病,PVNS是一种以滑膜增殖为特征的侵蚀性关节疾病,二者均为滑膜受累,且临床表现相似,RA合并PVNS的病例罕见,明确诊断具有难度。本研究分别在万方数据知识服务平台和中国知网以“类风湿关节炎”和“色素沉着绒毛结节性滑膜炎”为检索词检索相关文献,未检索到中文文献。在PubMed中以“rheumatoid arthritis”与“pigmented villonodular synovitis”为检索词检索,共检索到2篇英文文献。对于该病,临床医生需充分了解患者的病程发展,通过MRI及组织病理检查做出正确诊断,并进行有针对性地治疗。

本文引用格式

彭嘉婧 , 刘田 . 类风湿关节炎合并色素沉着绒毛结节性滑膜炎1例及文献回顾[J]. 北京大学学报(医学版), 2020 , 52(6) : 1135 -1139 . DOI: 10.19723/j.issn.1671-167X.2020.06.025

Abstract

We reported a case of rheumatoid arthritis (RA) combined with pigmented villonodular synovitis (PVNS) from Peking University People’s Hospital. The clinical data were introduced and the related literature was reviewed. The clinical features, treatment and prognosis of the disease were summarized to improve clinicians’ understanding of this rare disease and avoid misdiagnosis and delayed diagnosis. A 45-year-old female, with 15 years of RA history and unregular treatment, was admitted to the hospital with the complaint of aggravating pain and swelling in the right knee for 3 months. The puncture of the right knee was performed and there was a large amount of bloody synovial fluid that could not be explained by her RA history. Moreover, the magnetic resonance imaging (MRI) of the right knee revealed hemosiderin deposition with low-intensity signals on both T1-weighted and T2-weighted images which suggested PVNS to us. Then, the patient underwent knee arthroscopy and biopsy to assist in diagnosis. The arthroscopic appearance and pathology were consistent with PVNS and the hyperplastic synovium was removed during arthroscopy. After the operation, she did active functional exercises and took disease-modifying antirheumatic drugs to control RA. She recovered well and remained asymptomatic after half a year of follow-up. Also, there was no recurrence of the right knee. As we all know, RA is a systemic autoimmune disease characterized by chronic synovitis and joint damage. And PVNS is characterized by synovial proliferation and infiltrative process. Both of them are synovial involvement and the clinical manifestations are quite similar. PVNS has occasionally been reported in association with RA. So it is difficult to make a clear diagnosis of RA combined with PVNS. Literature was searched with RA+PVNS in the WanFang Medical Network Database and China National Knowledge Infrastructure and there were no related Chinese cases. Then we searched literature from PubMed with RA+PVNS. The cases were still rare and eventually 2 related articles were yielded including 2 similar patients. It is necessary to fully understand the disease development, complicated MRI appearance and various pathological morpho-logy. They can contribute to making a correct diagnosis which is effective to guide the proper treatment.

参考文献

[1] Croia C, Bursi R, Sutera D, et al. One year in review 2019: pathogenesis of rheumatoid arthritis[J]. Clin Exp Rheumatol, 2019,37(3):347-357.
[2] Ottaviani S, Ayral X, Dougados M, et al. Pigmented villonodular synovitis: a retrospective single-center study of 122 cases and review of the literature[J]. Semin Arthritis Rheum, 2011,40(6):539-546.
[3] Hirose I, Mori Y, Fujimoto A. Case report: rheumatoid arthritis occurring during observation after surgery for localized pigmented villonodular synovitis[J]. Mod Rheumatol, 2003,13(3):281-284.
[4] Zhao X, Ji W, Qian X, et al. Pigmented villonodular synovitis developing in a patient with rheumatoid arthritis[J]. J Clin Rheumatol, 2014,20(5):283-286.
[5] Dougados M. Synovial fluid cell analysis[J]. Baillieres Clin Rheumatol, 1996,10(3):519-534.
[6] Myers BW, Masi AT. Pigmented villonodular synovitis and tenosynovitis: A clinical epidemiologic study of 166 cases and literature review[J]. Medicine (Baltimore), 1980,59(3):223-238.
[7] Mastboom M, Verspoor F, Verschoor A, et al. Higher incidence rates than previously known in tenosynovial giant cell tumors[J]. Acta orthopaedica, 2017,88(6):688-694.
[8] Fiocco U, Sfriso P, Lunardi F, et al. Molecular pathways involved in synovial cell inflammation and tumoral proliferation in diffuse pigmented villonodular synovitis[J]. Autoimmun Rev, 2010,9(11):780-784.
[9] Gouin F, Noailles T. Localized and diffuse forms of tenosynovial giant cell tumor (formerly giant cell tumor of the tendon sheath and pigmented villonodular synovitis)[J]. Orthop Traumatol Surg Res, 2017,103(1S):S91-S97.
[10] Staals E, Ferrari S, Donati D, et al. Diffuse-type tenosynovial giant cell tumour: Current treatment concepts and future perspectives[J]. Eur J Cancer, 2016,63:34-40.
[11] Cupp J, Miller M, Montgomery K, et al. Translocation and expression of CSF1 in pigmented villonodular synovitis, tenosynovial giant cell tumor, rheumatoid arthritis and other reactive synovitides[J]. Am J Surg Pathol, 2007,31(6):970-976.
[12] Oliviero F, Galozzi P, Ramonda R, et al. Unusual findings in synovial fluid analysis: A review[J]. Ann Clin Lab Sci, 2017,47(3):253-259.
[13] 列锐锋. 膝关节色素沉着绒毛结节性滑膜炎MRI表现[J]. 罕少疾病杂志, 2020,27(2):67-69.
[14] Houdek MT, Scorianz M, Wyles CC, et al. Long-term outcome of knee arthroplasty in the setting of pigmented villonodular synovitis[J]. Knee, 2017,24(4):851-855.
[15] Barile A, Sabatini M, Iannessi F, et al. Pigmented villonodular synovitis (PVNS) of the knee joint: magnetic resonance imaging (MRI) using standard and dynamic paramagnetic contrast media. Report of 52 cases surgically and histologically controlled[J]. Radiol Med, 2004,107(4):356-366.
[16] 巫娟, 徐钢, 傅静. 膝关节色素沉着绒毛结节性滑膜炎的临床病理分析[J]. 四川医学, 2017,38(9):1010-1013.
[17] Bernthal N, Ishmael C, Burke Z. Management of pigmented villonodular synovitis (PVNS): an orthopedic surgeon’s perspective[J]. Curr Oncol Rep, 2020,22(6):63.
[18] Palmerini E, Staals E, Maki R, et al. Tenosynovial giant cell tumour/pigmented villonodular synovitis: outcome of 294 patients before the era of kinase inhibitors[J]. Eur J Cancer, 2015,51(2):210-217.
[19] Tap W, Gelderblom H, Palmerini E, et al. Pexidartinib versus placebo for advanced tenosynovial giant cell tumour (ENLIVEN): a randomised phase 3 trial[J]. Lancet, 2019,394(10197):478-487.
[20] Monestime S, Lazaridis D. Pexidartinib (TURALIO?): The first FDA-indicated systemic treatment for tenosynovial giant cell tumor[J]. Drugs R D, 2020,20(3):189-195.
[21] Gelderblom H, Cropet C, Chevreau C, et al. Nilotinib in locally advanced pigmented villonodular synovitis: a multicentre, open-label, single-arm, phase 2 trial[J]. Lancet Oncol, 2018,19(5):639-648.
文章导航

/