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

doi:10.19723/j.issn.1671-167X.2020.06.025

摘要/Abstract

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。

关键词: 色素沉着绒毛结节性滑膜炎, 类风湿关节炎, 滑液, 关节积血

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.

Key words: Pigmented villonodular synovitis, Rheumatoid arthritis, Synovial fluid, Hemarthrosis

中图分类号:

R593.22

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.

Jia-jing PENG,Tian LIU. Rheumatoid arthritis combined with pigmented villonodular synovitis: A case report and literature review[J]. Journal of Peking University (Health Sciences), 2020, 52(6): 1135-1139.

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参考文献 21

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Study	Gender	Age/years	Duration/years	Affected joints of PVNS	Affected joints of RA	WBC/ (×10⁹/L)	HB/ (g/L)	PLT/ (×10⁹/L)
This study	Female	45	15	Right knee	Wrist, MCP, PIP, knee	4	129	188
Hirose et al^[3]	Female	38	5.3	Right knee	Wrist, MCP, ankle, knee	3.5	121	300
Zhao et al^[4]	Male	56	3	Right knee	Wrist, MCP, PIP, knee	-	-	-
Study	ESR/(mm/h)	CRP/(mg/L)	RF/(IU/mL)	Anti-CCP/(U/mL)	MRI	Synovial fluid	Arthroscopy	Radiation therapy
This study	11	0.39	<20	137.9	Soft-tissue mass with low intensity signals on T1-weighted and T2-weighted images(all these three patients had such MRI manifestations)	Bloody	Once	No
Hirose et al^[3]	82	2.7	151	-		Bloody	Twice	Yes
Zhao et al^[4]	34	44.6	238	>200		Bloody	Once	No