Oide T, Ohara S, Oguchi K, Maruyama M, Yazawa M, Inoue K, Sekijima Y, Tokuda T, Ikeda S
Third Department of Medicine, Shinshu University School of Medicine, Department of Neurology, National Chushin-Matsumoto Hospital, Matsumoto, Japan.
Clin Exp Rheumatol. 2004 Jan-Feb;22(1):91-8.
Remitting seronegative symmetrical synovitis with pitting edema (RS3PE) has so far been reported almost exclusively from the USA and Europe. We carried out this study to define the clinical characteristics of this syndrome in Japanese patients.
Prospectively, we identified 13 Japanese patients with RS3PE (5 men and 8 women, age 72.7 +/- 11.8 years (mean +/- SD)) without underlying neoplasm. Their clinical features were summarized, pertinent laboratory data including serum/synovial interleukin-6 (IL-6) concentrations were obtained, and extensive radiologic studies using magnetic resonance imaging and 67gallium-citrate (67Ga) whole body scintigram were performed.
All patients suffered from proximal arthralgia/myalgia in addition to typical distal symptoms of RS3PE, and all experienced systemic symptoms such as fever, malaise, and weight loss. In laboratory examinations, anemia and elevated inflammatory markers were often remarkable. Magnetic resonance imaging showed severe tenosynovitis of the hands. 67Ga-scintigram revealed radioisotope accumulation in both proximal and distal joints of the extremities. IL-6 activity was markedly elevated both in the serum (mean 82.4 +/- 62.1 (SD) pg/ml, normal range 0.157-2.94) and in the synovial fluid (mean 3350 +/- 633 (SD) pg/ml).
Compared with cases reported previously from the USA/Europe, Japanese patients with RS3PE are characterized by more prominent systemic symptoms/signs associated with marked inflammatory responses including elevated IL-6 activity. All patients had proximal as well as distal synovitis which could be demonstrated by 67Ga-scintigram. These clinical features were very similar to those of polymyalgia rheumatica, suggesting that RS3PE and polymyalgia rheumatica are closely related disorders which may have a common pathogenesis.
迄今为止，几乎仅有来自美国和欧洲的关于伴有指凹性水肿的血清阴性对称性滑膜炎（RS3PE）的报道。我们开展这项研究以明确日本患者中该综合征的临床特征。
我们前瞻性地确定了13例无潜在肿瘤的日本RS3PE患者（5例男性和8例女性，年龄72.7±11.8岁（均值±标准差））。总结了他们的临床特征，获取了包括血清/滑膜白细胞介素-6（IL-6）浓度在内的相关实验室数据，并进行了使用磁共振成像和枸橼酸镓（67Ga）全身闪烁扫描的广泛影像学研究。
所有患者除了有RS3PE典型的远端症状外，还患有近端关节痛/肌痛，并且都经历过发热、不适和体重减轻等全身症状。在实验室检查中，贫血和炎症标志物升高常常很明显。磁共振成像显示手部严重腱鞘炎。67Ga闪烁扫描显示放射性同位素在四肢近端和远端关节均有积聚。血清中IL-6活性（均值82.4±62.1（标准差）pg/ml，正常范围0.157 - 2.94）和滑液中IL-6活性（均值3350±633（标准差）pg/ml）均显著升高。
与先前美国/欧洲报道的病例相比，日本RS3PE患者的特点是伴有包括IL-6活性升高在内的明显炎症反应的更突出的全身症状/体征。所有患者近端和远端均有滑膜炎，这可通过67Ga闪烁扫描显示。这些临床特征与风湿性多肌痛非常相似，提示RS3PE和风湿性多肌痛是密切相关的疾病，可能有共同的发病机制。
