缓解性血清阴性对称性滑膜炎伴凹陷性水肿22例临床回顾
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摘要
目的:总结缓解性血清阴性对称性滑膜炎伴凹陷性水肿(RS3PE)的临床特点及转归,为该病的诊治提供更多的参考依据。方法:回顾性分析我院2004年1月至2016年2月的22例我院收治的RS3PE住院病例的临床数据和随访资料,总结该病的特点。结果:本研究纳入男性13例(59.1%),女性9例(40.9%),平均发病年龄(65.6±10.0)岁(40.0～79.0岁),发病至确诊时间12天至2年,均为急性起病,并伴有不同程度的对称性多关节炎及肢端水肿。发热6例(27.3%)。主要实验室指标:血红蛋白(109.1±16.0)g·L~(-1),血小板计数(277.0±91.8)×109·L~(-1),血清白蛋白(36.1±5.0)g·L~(-1),C反应蛋白(62.5±67.4)mg·L~(-1),血沉(58.1±33.9)mm/h。测血清铁蛋白的6例中有5例(83.3%)升高,测肿瘤标记的10例均正常。18例(81.8%)行关节影像学检查,均未见骨破坏。5例行唇腺粘膜活检、2例行骨髓穿刺活检均无异常。住院期间治疗后症状均明显好转,4例(18.2%)单用糖皮质激素,18例(81.8%)在激素基础上联用慢作用药物。电话随访4.0月～8.0年(中位随访时间5.0年),有5例(22.7%)关节症状和肢端水肿持续缓解,4例(18.2%)症状减轻,1例(4.5%)反复,3例(13.6%)发展为类风湿关节炎,9例(40.9%)失访,确诊淋巴瘤1例(4.5%)。老年组RS3PE患者红细胞计数、血红蛋白、血清总蛋白及白蛋白低于非老年组。结论:RS3PE多为老年患者,急性起病,为对称性多关节炎伴肢端水肿,常伴贫血及低蛋白血症,对糖皮质激素敏感,有时可伴肿瘤,关于RS3PE的随访观察病例数迄今仍不足。
Objective: To explore the clinical characteristics and outcomes of the remitting seronegative symmetrical synovitis with pitting edema(RS3 PE), and help the diagnosis and treatment. Methods: A retrospective review of RS3 PE patients diagnosed from January 1, 2004 to February 29, 2016 was performed. The clinical data and follow-up information were collected. The characteristics of RS3 PE were summarized by description, comparison of the means, ratios or medians. Results: We identified 13 males(59.1 %) and nine females(40.9 %) RS3 PE cases. The mean onset age was 65.6 ±10.0 years(40.0 to 79.0 years). Diagnosis time to onset was 12 days to two years. All patients were acute onset with varying degrees of symmetric polyarthritis and extremity edema. Six cases(27.3 %)presented fever. The means of main laboratory indicators were as following: hemoglobin 109.1 ±16.0 g·L~(-1), platelet count 277.0 ±91.8× 109·L~(-1), serum albumin 36.1 ±5.0 g·L~(-1), c-reactive protein 62.5 ±67.4 mg·L~(-1), erythrocyte sedimentation rate 58.1 ±33.9 mm/h.Serum ferritins tested in six cases were elevated in five(83 %). Tumor markers detected in ten cases were all normal. Eighteen cases(86%) underwent joint iconographic examination, and no bone destruction was found. Both five cases performed labial gland mucosal biopsy and two cases received bone marrow biopsy were normal. Symptoms were significantly improved in all cases during hospitalization. Four cases were prescribed with glucocorticoids alone, and 18(81.8 %) also combined with slow-acting drugs. During the telephone follow-up period of four months to eight years(the median was 5.0 years), the joint symptoms and extremity edema continued to ease in five cases(22.7 %), and relieved in four cases(18.2 %). Only one(4.5 %) was recurrent, and three(13.6 %)developed rheumatoid arthritis. Nine cases(40.9 %) were lost. Malignant tumor happened in one case(4.5 %). The erythrocyte count,hemoglobin, serum total protein and albumin in the elderly RS3 PE patients were lower than those in the non-elderly group. Conclusions:RS3 PE is mostly acute onset in elderly patients, accompanied by symmetrically multiple arthritis with acroedema. In addition to be sensitive to glucocorticoid therapy, RS3 PE often presents as anemia and hypoproteinemia, and sometimes accompanied by tumor. The study of RS3 PE is still insufficient so far.
Objective: To explore the clinical characteristics and outcomes of the remitting seronegative symmetrical synovitis with pitting edema(RS3 PE), and help the diagnosis and treatment. Methods: A retrospective review of RS3 PE patients diagnosed from January 1, 2004 to February 29, 2016 was performed. The clinical data and follow-up information were collected. The characteristics of RS3 PE were summarized by description, comparison of the means, ratios or medians. Results: We identified 13 males(59.1 %) and nine females(40.9 %) RS3 PE cases. The mean onset age was 65.6 ±10.0 years(40.0 to 79.0 years). Diagnosis time to onset was 12 days to two years. All patients were acute onset with varying degrees of symmetric polyarthritis and extremity edema. Six cases(27.3 %)presented fever. The means of main laboratory indicators were as following: hemoglobin 109.1 ±16.0 g·L~(-1), platelet count 277.0 ±91.8× 109·L~(-1), serum albumin 36.1 ±5.0 g·L~(-1), c-reactive protein 62.5 ±67.4 mg·L~(-1), erythrocyte sedimentation rate 58.1 ±33.9 mm/h.Serum ferritins tested in six cases were elevated in five(83 %). Tumor markers detected in ten cases were all normal. Eighteen cases(86%) underwent joint iconographic examination, and no bone destruction was found. Both five cases performed labial gland mucosal biopsy and two cases received bone marrow biopsy were normal. Symptoms were significantly improved in all cases during hospitalization. Four cases were prescribed with glucocorticoids alone, and 18(81.8 %) also combined with slow-acting drugs. During the telephone follow-up period of four months to eight years(the median was 5.0 years), the joint symptoms and extremity edema continued to ease in five cases(22.7 %), and relieved in four cases(18.2 %). Only one(4.5 %) was recurrent, and three(13.6 %)developed rheumatoid arthritis. Nine cases(40.9 %) were lost. Malignant tumor happened in one case(4.5 %). The erythrocyte count,hemoglobin, serum total protein and albumin in the elderly RS3 PE patients were lower than those in the non-elderly group. Conclusions:RS3 PE is mostly acute onset in elderly patients, accompanied by symmetrically multiple arthritis with acroedema. In addition to be sensitive to glucocorticoid therapy, RS3 PE often presents as anemia and hypoproteinemia, and sometimes accompanied by tumor. The study of RS3 PE is still insufficient so far.
引文
[1]Mc Carty DJ,O'Duffy JD,Pearson L,et al.Remitting seronegative symmetrical synovitis with pitting edema,RS3PE syndrome[J].JAMA,1985,254(19):2763-2767
[2]Bucaloiu ID,Olenginski TP,Harrington TM.Remitting seronegative symmetrical synovitis with pitting edema syndrome in a rural tertiary care practice:a retrospective analysis[J].Mayo Clinic Proceedings,2007,82(12):1510-1515
[3]Russell EB,Hunter JB,Pearson L,et al.Remitting seronegative symmetrical synovitis with pitting edema-13 additional cases[J].The Journal of Rheumatology,1990,17(5):633-639
[4]郭惠芳,于庆海,刘凤珍,等.缓解性血清阴性的对称性滑膜炎伴可凹陷性水肿综合征21例分析[J].中华老年医学杂志,2005,24(5):345-347
[5]李桂叶,安媛,栗占国.缓解性血清阴性对称性滑膜炎伴凹陷性水肿综合征例临床分析并文献复习[J].中华风湿病学杂志,2012,16(7):468-472
[6]武加标,任敏,赵东宝.非典型缓解性血清阴性对称性滑膜炎伴凹陷性水肿综合征的临床特点[J].新医学,2017,48(1):40-43
[7]谢长好,李志军,陈琳洁,等.缓解性血清阴性对称性滑膜炎伴凹陷水肿及文献复习[J].中华临床免疫和变态反应杂志,2011,5(4):307-312
[8]Wang J,Pu C,Wang Z,et al.Remitting Seronegative Symmetrical Synovitis With Pitting Edema:Appearance on FDG PET/CT[J].Clinical Nuclear Medicine,2017,42(6):492-495
[9]Eguia HA,Parodi Garcia JF,Ramas DC,et al.Remitting seronegative symmetrical synovitis with pitting oedema(RS3PE)case presentation and comparison with other polyarthritides affecting older people[J].Age&Ageing,2017,46(2):333-334
[10]Origuchi T,Arima K,Umeda M,et al.Clinical outcomes in the first year of remitting seronegative symmetrical synovitis with pitting edema(RS3PE)syndrome[J].Modern Rheumatology,2016,27(1):150-154
[11]Schaeverbeke T,Fatout E,Marce S,et al.Remitting seronegative symmetrical synovitis with pitting oedema:disease or syndrome?[J].Annals of the Rheumatic Diseases,1995,54(8):681-684
[12]Guma M,Casado E,Tena X,et al.RS3PE:six years later[J].Annals of the Rheumatic Diseases,1999,58(11):722-723
[13]Fietta P,Manganelli P.Remitting seronegative symmetrical synovitis with pitting edema and primary Sj觟gren's syndrome[J].Clinical and Experimental Rheumatology,2000,18(2):269
[14]Gisserot O,Cremades S,Landais C,et al.RS3PE revealing recurrent non-Hodgkin's lymphoma[J].Joint,Bone,Spine:Revue Du Rhumatisme,2004,71(5):424-426
[15]Tunc SE,Arslan C,Ayvacioglu NB,et al.Paraneoplastic remitting seronegative symmetrical synovitis with pitting edema(RS3PEsyndrome):a report of two cases and review of the literature[J].Rheumatology International,2004,24(4):234-237
[16]Paira S,Graf C,Roverano S,et al.Remitting seronegative symmetrical synovitis with pitting edema:a study of 12 cases[J].Clinical Rheumatology,2002,21(2):146-149
[17]Vinci M,Malaguarnera L,Pistone G.RS3PE and ovarian cancer[J].Annals of the Rheumatic Diseases,2001,60(4):429-431
[18]Arima K,Origuchi T,Tamai M,et al.RS3PE syndrome presenting as vascular endothelial growth factor associated disorder[J].Annals of the Rheumatic Diseases,2005,64(11):1653-1655
[19]Oide T,Ohara S,Oguchi K,et al.Remitting seronegative symmetrical synovitis with pitting edema(RS3PE)syndrome in Nagano,Japan:clinical,radiological,and cytokine studies of 13patients[J].Clinical and Experimental Rheumatology,2004,22(1):91-98
[20]Origuchi T,Arima K,Kawashiri SY,et al.High serum matrix metalloproteinase 3 is characteristic of patients with paraneoplastic remitting seronegative symmetrical synovitis with pitting edema syndrome[J].Modern Rheumatology,2012,22(4):584-588
[21]Sibilia J,Friess S,Schaeverbeke T,et al.Remitting seronegative symmetrical synovitis with pitting edema(RS3PE):a form of paraneoplastic polyarthritis?[J].The Journal of Rheumatology,1999,26(1):115-120
[22]Li H,Altman RD,Yao Q.RS3PE:Clinical and Research Development[J].Current Rheumatology Reports,2015,17(8):49
[23]郑文洁,王昱,蒋颖,等.8例RS3PE临床回顾性分析[J].基础医学与临床,2008,28(8):882-885
[24]Kimura M,Tokuda Y,Oshiawa H,et al.Clinical characteristics of patients with remitting seronegative symmetrical synovitis with pitting edema compared to patients with pure polymyalgia rheumatica[J].The Journal of Rheumatology,2012,39(1):148-153
[25]Karmacharya P,Donato AA,Aryal MR,et al.RS3PE revisited:a systematic review and meta-analysis of 331 cases[J].Clinical&Experimental Rheumatology,2016,34(3):404-415
[26]Filetti M,Anselmi E,Macrini S,et al.Resolution of remitting seronegative symmetrical synovitis with pitting edema(RS3PE)during Nivolumab therapy for non-small cell lung cancer:a case report[J].Seminars in Arthritis and Rheumatism,2018,pii:S0049-0172(17)30781-3
[27]郝振叶,崔静,郭莹莹,等.RS3PE综合征发病前特发性膜性肾病1例[J].中国现代医生,2018,56(3):135-136+139
[28]Yamauchi K,Sato Y,Yamashita K,et al.RS3PE in association with dipeptidyl peptidase-4 inhibitor:report of two cases[J].Diabetes Care,2012,35(2):e7
[29]Yoshioka Y,Mitsuhashi M,Kato A,et al.The association of the early onset of remitting seronegative symmetrical synovitis with pitting oedema(RS3PE)syndrome with dipeptidyl peptidase-4(DPP4)inhibitor[J].Annals of the Rheumatic Diseases,2018,77(suppl 2):1686
[30]Oyama K,Taniguchi J,et al.Remitting seronegative symmetrical synovitis with pitting edema syndrome in individuals with type 2diabetes mellitus or impaired glucose tolerance[J].Diabetes research and clinical practice,2015,110(1):e5-e8
[2]Bucaloiu ID,Olenginski TP,Harrington TM.Remitting seronegative symmetrical synovitis with pitting edema syndrome in a rural tertiary care practice:a retrospective analysis[J].Mayo Clinic Proceedings,2007,82(12):1510-1515
[3]Russell EB,Hunter JB,Pearson L,et al.Remitting seronegative symmetrical synovitis with pitting edema-13 additional cases[J].The Journal of Rheumatology,1990,17(5):633-639
[4]郭惠芳,于庆海,刘凤珍,等.缓解性血清阴性的对称性滑膜炎伴可凹陷性水肿综合征21例分析[J].中华老年医学杂志,2005,24(5):345-347
[5]李桂叶,安媛,栗占国.缓解性血清阴性对称性滑膜炎伴凹陷性水肿综合征例临床分析并文献复习[J].中华风湿病学杂志,2012,16(7):468-472
[6]武加标,任敏,赵东宝.非典型缓解性血清阴性对称性滑膜炎伴凹陷性水肿综合征的临床特点[J].新医学,2017,48(1):40-43
[7]谢长好,李志军,陈琳洁,等.缓解性血清阴性对称性滑膜炎伴凹陷水肿及文献复习[J].中华临床免疫和变态反应杂志,2011,5(4):307-312
[8]Wang J,Pu C,Wang Z,et al.Remitting Seronegative Symmetrical Synovitis With Pitting Edema:Appearance on FDG PET/CT[J].Clinical Nuclear Medicine,2017,42(6):492-495
[9]Eguia HA,Parodi Garcia JF,Ramas DC,et al.Remitting seronegative symmetrical synovitis with pitting oedema(RS3PE)case presentation and comparison with other polyarthritides affecting older people[J].Age&Ageing,2017,46(2):333-334
[10]Origuchi T,Arima K,Umeda M,et al.Clinical outcomes in the first year of remitting seronegative symmetrical synovitis with pitting edema(RS3PE)syndrome[J].Modern Rheumatology,2016,27(1):150-154
[11]Schaeverbeke T,Fatout E,Marce S,et al.Remitting seronegative symmetrical synovitis with pitting oedema:disease or syndrome?[J].Annals of the Rheumatic Diseases,1995,54(8):681-684
[12]Guma M,Casado E,Tena X,et al.RS3PE:six years later[J].Annals of the Rheumatic Diseases,1999,58(11):722-723
[13]Fietta P,Manganelli P.Remitting seronegative symmetrical synovitis with pitting edema and primary Sj觟gren's syndrome[J].Clinical and Experimental Rheumatology,2000,18(2):269
[14]Gisserot O,Cremades S,Landais C,et al.RS3PE revealing recurrent non-Hodgkin's lymphoma[J].Joint,Bone,Spine:Revue Du Rhumatisme,2004,71(5):424-426
[15]Tunc SE,Arslan C,Ayvacioglu NB,et al.Paraneoplastic remitting seronegative symmetrical synovitis with pitting edema(RS3PEsyndrome):a report of two cases and review of the literature[J].Rheumatology International,2004,24(4):234-237
[16]Paira S,Graf C,Roverano S,et al.Remitting seronegative symmetrical synovitis with pitting edema:a study of 12 cases[J].Clinical Rheumatology,2002,21(2):146-149
[17]Vinci M,Malaguarnera L,Pistone G.RS3PE and ovarian cancer[J].Annals of the Rheumatic Diseases,2001,60(4):429-431
[18]Arima K,Origuchi T,Tamai M,et al.RS3PE syndrome presenting as vascular endothelial growth factor associated disorder[J].Annals of the Rheumatic Diseases,2005,64(11):1653-1655
[19]Oide T,Ohara S,Oguchi K,et al.Remitting seronegative symmetrical synovitis with pitting edema(RS3PE)syndrome in Nagano,Japan:clinical,radiological,and cytokine studies of 13patients[J].Clinical and Experimental Rheumatology,2004,22(1):91-98
[20]Origuchi T,Arima K,Kawashiri SY,et al.High serum matrix metalloproteinase 3 is characteristic of patients with paraneoplastic remitting seronegative symmetrical synovitis with pitting edema syndrome[J].Modern Rheumatology,2012,22(4):584-588
[21]Sibilia J,Friess S,Schaeverbeke T,et al.Remitting seronegative symmetrical synovitis with pitting edema(RS3PE):a form of paraneoplastic polyarthritis?[J].The Journal of Rheumatology,1999,26(1):115-120
[22]Li H,Altman RD,Yao Q.RS3PE:Clinical and Research Development[J].Current Rheumatology Reports,2015,17(8):49
[23]郑文洁,王昱,蒋颖,等.8例RS3PE临床回顾性分析[J].基础医学与临床,2008,28(8):882-885
[24]Kimura M,Tokuda Y,Oshiawa H,et al.Clinical characteristics of patients with remitting seronegative symmetrical synovitis with pitting edema compared to patients with pure polymyalgia rheumatica[J].The Journal of Rheumatology,2012,39(1):148-153
[25]Karmacharya P,Donato AA,Aryal MR,et al.RS3PE revisited:a systematic review and meta-analysis of 331 cases[J].Clinical&Experimental Rheumatology,2016,34(3):404-415
[26]Filetti M,Anselmi E,Macrini S,et al.Resolution of remitting seronegative symmetrical synovitis with pitting edema(RS3PE)during Nivolumab therapy for non-small cell lung cancer:a case report[J].Seminars in Arthritis and Rheumatism,2018,pii:S0049-0172(17)30781-3
[27]郝振叶,崔静,郭莹莹,等.RS3PE综合征发病前特发性膜性肾病1例[J].中国现代医生,2018,56(3):135-136+139
[28]Yamauchi K,Sato Y,Yamashita K,et al.RS3PE in association with dipeptidyl peptidase-4 inhibitor:report of two cases[J].Diabetes Care,2012,35(2):e7
[29]Yoshioka Y,Mitsuhashi M,Kato A,et al.The association of the early onset of remitting seronegative symmetrical synovitis with pitting oedema(RS3PE)syndrome with dipeptidyl peptidase-4(DPP4)inhibitor[J].Annals of the Rheumatic Diseases,2018,77(suppl 2):1686
[30]Oyama K,Taniguchi J,et al.Remitting seronegative symmetrical synovitis with pitting edema syndrome in individuals with type 2diabetes mellitus or impaired glucose tolerance[J].Diabetes research and clinical practice,2015,110(1):e5-e8