Remitting seronegative symmetrical synovitis with pitting oedema syndrome, associated with prostate adenocarcinoma: a case report

Remitting seronegative symmetrical synovitis with pitting oedema (RS3PE) of the dorsum of the hands and/or feet can be observed in different inflammatory rheumatic diseases as well as in haematological and solid malignancies. McCarty et al, described this syndrome for the first time more than twenty years ago. Underlying malignancy should always be excluded in patients with RS3PE syndrome.     

High serum matrix metalloproteinase 3 is characteristic of patients with paraneoplastic remitting seronegative symmetrical synovitis with pitting edema syndrome

Recently, it was reported that remitting seronegative symmetrical synovitis with pitting edema (RS3PE) syndrome could be complicated with solid tumors. In a retrospective, multicenter study between October, 2003 and September, 2010, we investigated the characteristics of patients with paraneoplastic RS3PE syndrome who fulfilled following criteria: (1) bilateral pitting edema of hands or feet or both, (2) sudden onset of polyarthritis, and (3) age >50?years, (4) seronegativity for rheumatoid factor (RF). A total of 33 cases fulfilled the above criteria. Eight patients (seven men and one woman) developed cancer within 2?years of RS3PE syndrome onset. There was no significant difference between the neoplastic and nonneoplastic groups in the proportions of patients with fever, symmetrical polyarthritis, pitting edema, and good response to corticosteroids. Serum matrix metalloproteinase 3 (MMP-3) level (median 437.3?ng/ml) in the paraneoplastic RS3PE patients was significantly higher than that in patients without neoplasia (median 114.7?ng/ml) (p?相似文献   

High serum matrix metalloproteinase 3 is characteristic of patients with paraneoplastic remitting seronegative symmetrical synovitis with pitting edema syndrome

Abstract

Recently, it was reported that remitting seronegative symmetrical synovitis with pitting edema (RS3PE) syndrome could be complicated with solid tumors. In a retrospective, multicenter study between October, 2003 and September, 2010, we investigated the characteristics of patients with paraneoplastic RS3PE syndrome who fulfilled following criteria: (1) bilateral pitting edema of hands or feet or both, (2) sudden onset of polyarthritis, and (3) age >50 years, (4) seronegativity for rheumatoid factor (RF). A total of 33 cases fulfilled the above criteria. Eight patients (seven men and one woman) developed cancer within 2 years of RS3PE syndrome onset. There was no significant difference between the neoplastic and nonneoplastic groups in the proportions of patients with fever, symmetrical polyarthritis, pitting edema, and good response to corticosteroids. Serum matrix metalloproteinase 3 (MMP-3) level (median 437.3 ng/ml) in the paraneoplastic RS3PE patients was significantly higher than that in patients without neoplasia (median 114.7 ng/ml) (p < 0.05). We found that high serum MMP-3 is characteristic of patients with paraneoplastic RS3PE syndrome.     

Oedematous polyarthritis and myopericarditis as presenting features of periarteritis nodosa

Remitting symmetrical seronegative synovitis with pitting edema (RS3PE) syndrome is a rare type of seronegative polyarthritis occurring in the elderly. It can be associated to various diseases. We report a case of RS3PE syndrome associated with myopericarditis, leading to the diagnosis of polyarteritis nodosa in a 71-year-old patient admitted to the hospital for a febrile acute polyarthritis with pitting edema of the hands associated with a marked inflammatory syndrome. On second day of hospitalization, a sustained chest pain led to the diagnosis of myopericarditis. Muscular biopsy showed necrotizing vasculitis, characteristic of polyarteritis nodosa. The coexistence of RS3PE and myopericarditis has never been described in the literature. Its association with polyarteritis nodosa is also very rare and only one case has been previously reported.     

The rate of polymyalgia rheumatica (PMR) and remitting seronegative symmetrical synovitis with pitting edema (RS3PE) syndrome in a clinic where primary care physicians are working in Japan

We analyzed the rate of polymyalgia rheumatica (PMR) and remitting seronegative symmetrical synovitis with pitting edema (RS3PE) syndrome, both characterized as seronegative inflammatory arthritis in elderly, in an outpatient unit where primary care physicians are working in Japan to better understand the epidemiological characteristics of the diseases in Japan. Consecutive outpatients who newly visited at Department of General Medicine, Asahikawa Medical University Hospital, Japan, between April 2004 and March 2010 were analyzed. Each parameter such as age, sex, diagnosis, and biochemical examination was investigated. During the 6?years, 10 or 3 patients were diagnosed as PMR or RS3PE syndrome, respectively. The patients with PMR were 7 women and 3 men, and the average age at diagnosis was 69. Out of all patients aged over 50 (n?=?3,347), the rate of PMR was 0.22% in men or 0.36% in women, respectively. On the other hand, RS3PE syndrome was diagnosed in 3 men (76, 76, and 81?years old). The rate of patients with RS3PE syndrome was 0.09% among outpatients aged over 50 indicating that the rate of PMR in an outpatient clinic in Japan is not far from previous findings reported from western countries. When compared with PMR, the rate of RS3PE syndrome was approximately one-third, providing for the first time the rate of RS3PE syndrome when compared with PMR. These epidemilogical data might help us pick up the diseases in primary care setting in Japan.     

An abrupt onset of seropositive polyarthritis with prominent distal tenosynovitis concomitant with bronochiolitis obliterans organizing pneumonia (BOOP): consideration of the relationship with RS3PE syndrome

A 64-year-old Japanese woman with a two-week history of polyarthralgia and persistent cough was diagnosed as seropositive polyarthritis and fulfilled the criteria of early rheumatoid arthritis (RA). In addition, inflammatory pitting edema of the distal extremities was apparent, suggestive of the remitting seronegative symmetrical synovitis with pitting edema (RS3PE) syndrome. A number of investigations including hand MRI, bone scintigraphy and HLA typing supported a diagnosis of RS3PE syndrome rather than RA. Chest computed tomography revealed concomitant evidence of bronchiolitis obliterans organizing pneumonia (BOOP). Treatment with 30 mg of prednisolone daily immediately ameliorated the polyarthritis and the BOOP. Seropositive polyarthritis with distal pitting edema may be categorized as both RA and the RS3PE syndrome.     

Remitting seronegative symmetrical synovitis with pitting oedema: disease or syndrome?

OBJECTIVE--To evaluate the outcome of patients with remitting seronegative symmetrical synovitis with pitting oedema (RS3PE). METHODS--In a retrospective chart review study, we identified all the patients presenting with polyarthritis and pitting oedema in the past 20 years. We tried to recall the 24 patients with characteristics of RS3PE according to McCarty et al. Two patients had died and four could not be traced. Five could not be seen after the initial period of follow up; relevant data were obtained from their practitioner. For the remaining 13 patients, clinical, radiological, and biological evaluation was performed in our department, with the last assessment in 1993. RESULTS--The follow up period was from one to 18 years (mean 4.6 (SD 4.5) years). Eleven patients developed one or several recurrences of articular manifestations consisting of mild oligoarthritis (n = 8), definite spondyloarthropathy (n = 2), and rheumatoid arthritis (n = 1). The delay of the first recurrence was 18 months to 12 years after the first attack. Thirteen patients had no recurrence, but three of them developed remarkable features: rheumatoid factor, antinuclear antibodies (1/2000), Sjögren's syndrome. HLA B typing was performed in nine patients and revealed B7 (n = 2), B27 (n = 2) and B22 (n = 2). Isolated HLA B27 typing was performed in two other patients and was positive in one. CONCLUSION--The long term outcome of RS3PE can lead to different rheumatic diseases. RS3PE appears to be a syndrome related to the elderly onset of the rheumatic diseases, including spondyloarthropathy and rheumatoid arthritis, rather than a specific entity.     

Remitting seronegative symmetrical synovitis with pitting edema associated with acute myeloid leukemia

Remitting seronegative symmetrical synovitis with pitting edema (RS3PE) almost exclusively affects elderly men and can occur independently or may be associated with a vast array of clinical conditions including underlying malignancy. Patients present with a polyarthritis similar to rheumatoid arthritis. We describe a case of an elderly man presenting with RS3PE who developed acute myeloid leukemia. It is important for clinicians to be aware of this possibility and initiate appropriate investigations, particularly if systemic symptoms are prominent, to detect an occult malignancy at a potentially earlier stage.     

RS3PE syndrome: a clinical and immunogenetical study

This study analyses the clinical, radiological, evolutive, and immunogenetical characteristics of a series of patients diagnosed with remitting seronegative symmetrical synovitis with pitting edema (RS3PE) syndrome. Reviewed were the clinical charts and human leukocyte antigen (HLA) profiles of all patients treated at a single teaching hospital fulfilling the features of this syndrome according to the definition of McCarty. Twelve cases were detected in ten men and two women aged from 62 to 85 years. Rheumatoid factor was negative in all cases, and antinuclear antibodies (ANA) were positive in two. All patients achieved complete resolution of their condition within 1 year with glucocorticoid (GC) use. Two relapsed after remission but responded again to low doses of GC. Four patients showed clinical and electrodiagnostic studies consistent with carpal tunnel syndrome. No specific HLA association could be found in this report. To date, none of these patients has developed definite rheumatic diseases, infections, or malignant diseases. Although the real nature of the syndrome is still a matter of debate, at least in our context, RS3PE remains a definite condition with an excellent prognosis.     

Remitting, seronegative (A) symmetrical synovitis with pitting edema--two cases of RS3PE syndrome

The sparing effect of neurological damage on the development and progression of several arthritic conditions has been documented. We describe the first 2 cases of unilateral remitting seronegative symmetrical synovitis with pitting edema (RS3PE) syndrome in individuals with neurologic disorders. Case 1 suffered from birth trauma resulting in paresis of the right upper extremity and developed RS3PE syndrome in the nonaffected extremities. Case 2 developed RS3PE syndrome on the nonparetic side 7 years after a cerebrovascular accident resulting in hemiparesis.     

Remitting seronegative symmetrical synovitis with pitting edema (RS3PE) syndrome accompanied by Parkinson's disease

We encountered two cases of RS3PE (remitting seronegative symmetrical synovitis with pitting edema) syndrome accompanied by Parkinson's disease (PD). Although the etiology of RS3PE syndrome is still unknown, several possible associations, such as malignancies and viral infections, have been reported; RS3PE syndrome is thought to be an autoimmune-mediated disorder. The present patients did not have any factors which are reported to be associated with RS3PE. Whether or not the complication of PD and RS3PE syndrome is incidental needs to be further examined, and we discuss here the possible cause of association between PD and RS3PE syndrome, including dopamine agonists one of the anti-PD medications.     

Paraneoplastic remitting seronegative symmetrical synovitis with pitting edema

Remitting seronegative symmetrical synovitis with pitting edema (RS3PE) is a syndrome that may be associated with many conditions, including malignancy. Three further cases of paraneoplastic RS3PE are described and the literature is reviewed. Paraneoplastic RS3PE is more frequently associated with solid tumors, in particular adenocarcinoma. The two clinical characteristics suggestive of paraneoplastic RS3PE are systemic sign/symptoms and the poor response to corticosteroid therapy.     

Magnetic resonance imaging findings in a case of remitting seronegative symmetrical synovitis with pitting edema

We describe a case of remitting seronegative symmetrical synovitis with pitting edema (RS3PE syndrome) in a 66-year-old man. This report discusses magnetic resonance imaging findings of RS3PE syndrome and the changes after steroid therapy.     

Remitting seronegative symmetrical synovitis with pitting edema syndrome: followup for neoplasia

OBJECTIVE: To investigate whether the remitting seronegative symmetrical synovitis with pitting edema (RS3PE) syndrome may represent a paraneoplastic disorder in a significant percentage of cases. METHODS: Patients diagnosed with RS3PE syndrome at the Medical College of Wisconsin before 1995 were telephoned and asked about their rheumatologic course since initial diagnosis of RS3PE and whether they had been diagnosed with any cancer. If so, permission was obtained to review tissue pathology. Criteria used for diagnosis of RS3PE syndrome included sudden onset of painful diffuse swelling of both hands associated with pitting edema of the dorsa of the hands without other synovitis or evidence of disease, negative rheumatoid factor, absence of radiologic abnormalities, and resolution within 6-12 months without sequelae. Data from the national SEER databank on population incidence of cancers in the appropriate sex, age, and ethnic groups for the years under study were used to assess relative risk for cancer. RESULTS: There were 10 patients for whom followup data were available. Four had a cancer diagnosed following recognition of the RS3PE syndrome; 1 patient developed non-Hodgkin's lymphoma initially diagnosed as hairy cell leukemia after 4 years; 1 developed acute lymphocytic leukemia with hyperdiploidy after 14 years; 1 was diagnosed with male breast cancer after 2.5 years; and 1 developed squamous cell lung cancer 12 months after RS3PE diagnosis. SEER data project an estimated rate of 2-3 cancers in a similar group of 10 patients of the same sex, age, and time period for this geographic area. CONCLUSION: The small sample size in this longterm followup precludes extrapolation to larger populations but suggests that there may be a slightly higher than expected rate of neoplasia in patients diagnosed with RS3PE syndrome. The interval between onset of RS3PE syndrome and diagnosis of cancer was fairly long, indicating that patients should be monitored for neoplasia with prudent age and sex specific surveillance for an extended period after diagnosis with RS3PE.     

Gastric carcinoma in association with remitting seronegative symmetrical synovitis with pitting edema.

We describe an elderly woman who developed remitting seronegative symmetrical synovitis with pitting edema (RS3PE). In addition to typical findings, she had increased alkaline phosphatase; and investigations revealed metastatic gastric carcinoma. The literature relating to RS3PE in association with neoplasia is reviewed.     

Remitting seronegative symmetrical synovitis with pitting edema following intravesical bacillus Calmette-Guérin instillation

Remitting seronegative symmetrical synovitis with pitting edema (RS3PE) is a rare syndrome of undetermined etiology occurring in the elderly. We describe the first case of RS3PE in a HLA-B27 positive 65-year-old man following intravesical bacillus Calmette-Guérin (BCG) instillation for bladder carcinoma. He developed symmetrical arthritis and synovitis involving wrists, knees, ankles, and metatarsophalangeal joints, with marked pitting edema of the dorsa of both hands and feet, fever, and elevated acute phase reactants. Right knee effusion revealed nonspecific sterile inflammatory fluid. He responded dramatically to nonsteroidal antiinflammatory drugs. BCG instillation may have triggered active symmetrical synovitis via local T cell activation and a T-helper-1 (Th-1)/Th-2 inflammatory profile.     

Remitting seronegative symmetrical synovitis pitting oedema after BCG instillation

Remitting seronegative symmetrical synovitis pitting oedema (RS3PE) is a distinct form of seronegative rheumatoid arthritis like polyarthritis. It is characterized by late onset symmetrical joint involvement and pitting oedema of hands and feet (JAMA 254(19):2763–2767, [1]). Polyarthritis secondary to intravesical Bacillus Calmette Guerin (BCG) therapy has been reported (Clin Rheumatol 21:536–537, [2]). To our knowledge, about 0.5% of patients receiving BCG instillation presented polyarthritis, but only one case of RS3PE has been reported (J Rheumatol 28:1699–1701, [3]). We described the second case of RS3PE following intravesical BCG instillation of bladder carcinoma.     

缓解性血清阴性对称性滑膜炎伴凹陷水肿及文献复习

目的对缓解性血清阴性对称性滑膜炎伴凹陷水肿（RS3PE）的临床和实验室特点以及治疗和预后进行分析,提高对该病的认识。方法收集蚌埠医学院第一附属医院2004年1月至2011年5月就诊的RS3PE患者共5例,其中住院患者4例、门诊患者1例。分析其临床表现、实验室特点、治疗反应及转归,并对文献进行复习。结果 5例患者均急性起病,有对称性多关节炎及肢端水肿。5例患者起病时红细胞沉降率（59.6±29.0）mm/h,C反应蛋白（78.7±28.8）mg/L,血白细胞（10.25±1.92）×109/L,血红蛋白（87.4±6.26）g/L,血小板（487.2±148.4）×109/L,血清类风湿因子、抗环瓜氨酸多肽抗体、抗核抗体均为阴性。双手X线均未见骨侵蚀性改变。5例患者经泼尼松和/或免疫抑制剂治疗后完全缓解,其中3例完全停药,1例在1年后复发诊断为急性粒-单核细胞白血病。结论 RS3PE是一种异质性临床症候群,可能是单纯的独立疾病,也可发展为自身免疫性疾病或肿瘤,临床上应保持警惕。     

Parvovirus B19: another agent associated with remitting seronegative symmetrical synovitis with pitting edema

A number of conditions have been associated with remitting seronegative symmetrical synovitis with pitting edema (RS3PE) and the controversy of whether this should be considered a syndrome rather than a disease continues. There are few reports on the role of infectious agents in the etiology of RS3PE, and human parvovirus B19 has not previously been linked to this syndrome. We describe a patient with RS3PE syndrome in association with positive serology and viremia for parvovirus B19. A 9-year followup failed to uncover another cause for RS3PE.     

Remitting seronegative symmetrical synovitis with pitting edema of the hands: ultrasound, color doppler ultrasound, and magnetic resonance imaging findings

OBJECTIVE: To assess the value of gray-scale ultrasound (US), color Doppler ultrasound (CDUS), contrast-enhanced CDUS, and magnetic resonance imaging (MRI) in the diagnostic evaluation of the hands in patients with remitting seronegative symmetrical synovitis with pitting edema (RS3PE). METHODS: Eight patients (5 men, 3 women; mean +/- SD age 69.3 +/- 7.2 years) with clinical diagnosis of RS3PE syndrome underwent US, CDUS, contrast-enhanced CDUS, and MRI. US was performed with a linear array transducer operating at 12 MHz. The US contrast agent (SHU 508; Levovist, Schering, Germany) was intravenously infused in a concentration of 300 mg/ml at a rate of 1 ml/minute. RESULTS: All patients showed symmetric subcutaneous edema and synovitis of tendons and finger joints on both US and MRI. Vascularity was detected subcutaneously in tendon sheaths and in the joint synovia on CDUS and MRI. Detection of increased vascularity was improved after contrast administration compared with unenhanced CDUS (P < 0.01). CONCLUSION: Ultrasound, CDUS, contrast-enhanced CDUS, and MRI are valuable tools in the diagnostic evaluation of involved anatomic structures in patients with RS3PE. Contrast-enhanced CDUS is superior to CDUS in assessment of inflammatory edema, effusion, and synovitis.