A Case of Idiopathic Massive Rice Bodies in the Knee Joint without Rheumatoid Arthritis or Tuberculosis and a Literature Review

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1 Journal of Rheumatic Diseases Vol. 23,. 5, October, Case Report A Case of Idiopathic Massive Rice Bodies in the Knee Joint without Rheumatoid Arthritis or Tuberculosis and a Literature Review Whan Yong Chung 1, Ji-Sun Song 2, Hwa Eun Oh 3, Hee Jin Park 4 Departments of 1 Orthopedics, 2 Pathology, and 4 Rheumatology, Catholic Kwandong University International St. Mary s Hospital, Incheon, 3 Department of Pathology, Korea University Ansan Hospital, Ansan, Korea Rice bodies are materials with an amorphous nucleus and a fibrin layer found floating in the synovial space and bursa. These bodies have often been detected in patients with rheumatoid arthritis, tuberculous arthritis, and bursitis. Although the etiology and pathogenesis of rice bodies are not yet fully understood, it has been hypothesized that they might be caused by chronic inflammation originating from the synovium. However, we report on a case of idiopathic massive rice bodies in the knee joint without evidence of inflammatory articular disease or infection including rheumatoid arthritis, sero spondyloarthritides, tuberculosis, or bacterial or fungal infection. (J Rheum Dis 2016;23: ) Key Words. Rice body, Knee, Rheumatoid arthritis, Tuberculosis INTRODUCTION Floating rice-like particles in the synovial space could be found in the inflammatory joint diseases. These particles are known as rice bodies and were first reported by Reise in 1895 in tuberculous arthritis [1]. Rice bodies have been found in the synovial space in patients with rheumatoid arthritis or sero spondyloarthritis [2] and in the bursa [3] and around tendon sheaths [4] in association with inflammation. However, we experienced a case with massive rice bodies in the knee joint without any inflammatory or infective evidence and report these idiopathic rice bodies with an associated literature review. CASE REPORT A 46-year-old male had developed discomfort in the right knee one year ago, and aggravated swelling and pain two month ago. For these symptoms, he visited to the outpatient clinic of orthopedics department. He did not have any medical history and previous joint problems. He worked in an office and had not experienced any traumatic events involving the right knee joint. He felt discomfort when climbing stairs but no definite tenderness and warmth in the right knee. He did not have any symptoms in the other joints and had not experienced inflammatory back pain, uveitis, psoriasis, or inflammatory bowel disease. At admission, his white blood cell count was 3,870/μL, hemoglobin was 14.9 g/dl, and platelet count was 224,000/μL. Erythrocyte sediment rate was 6 mm/hr, and C-reactive protein was 3.18 mg/l ( range, 0 5 mg/l). Antinuclear antibody, rheumatoid factor, anti-cyclic citrullinated protein antibody, and human histocompatibility leukocyte antigen (HLA) B27 were all. Magnetic resonance imaging (MRI) of the knee showed a large amount of joint effusion with numerous low-signal foci in the suprapatellar bursa against a background of fluid signal intensity on T2-weighted image and intact anterior and posterior cruciate ligaments and collateral ligaments, as shown in Figure 1. Arthroscopic syno- Received:vember 19, 2015, Revised:December 23, 2015, Accepted:December 24, 2015 Corresponding to:hee Jin Park, Department of Rheumatology, Catholic Kwandong University International St. Mary s Hospital, 25 Simgok-ro 100beon-gil, Seo-gu, Incheon 22711, Korea. yolliko@ish.ac.kr pissn: X, eissn: Copyright c 2016 by The Korean College of Rheumatology. All rights reserved. This is a Free Access article, which permits unrestricted non-commerical use, distribution, and reproduction in any medium, provided the original work is properly cited. 316

2 Idiopathic Massive Rice Bodies in the Knee Joint vectomy of the right knee joint was conducted for diagnosis and revealed numerous rice bodies in the synovial space, but no definite synovial proliferation, as shown in Figure 2. Microscopic pathology of these materials revealed multiple nodular fibrocartilaginous tissues consistent with rice bodies and clean synovium without any synovial hyperplasia and inflammatory cells as shown in Figure 3. The results of bacterial, fungal, and acid-fast ba- cilli (AFB) culture studies from the synovium and washing synovial fluid were all, and real-time polymerase chain reaction (PCR) for non-tuberculous mycobacterium (NTM) of synovium was also. His symptoms improved after without any medications and continue to be stable 6 months later. Figure 1. Magnetic resonance images of the right knee. (A) T1-weighted image (T1WI) and (B D) T2-weighted images (T2WI) showed a large amount of joint effusion with numerous low-signal foci against a background of fluid signal intensity on T2WI. Anterior and posterior cruciate ligaments and collateral ligaments were intact. Figure 2. Gross morphology of rice bodies in the washing synovial fluid and arthroscopic findings. (A) Many white amorphous materials were found in the washing synovial fluid collected through arthroscopic irrigation. Arthroscopic findings were consistent with rice bodies in the suprapatellar space (B) and revealed a synovium (C)

3 Whan Yong Chung et al. Figure 3. Pathology of rice body and synovium. (A) Microscopic examination (H&E, x40) revealed multiple nodular fibrocartilaginous tissues consistent with rice bodies. (B) The synovium of the knee joint (H&E, x100) showed a histologically clean synovial surface. Table 1. Clinical characteristics and prognosis after treatment of idiopathic rice bodies Age (yr)/ gender 38/F Site of rice body Right ankle Duration of symptom Laboratory data Culture Pathology PPD Tenosynovitis skin test ( ) 51/M Left wrist /HLA Tenosynovitis B27 31/M Left knee 3 mo Bacterial Synovitis culture ( ) AFB ( ) 4/F Both shoulder Unknown ESR (18 mm/hr)/ CRP and knee 11/M Left knee 4 mo Chronic synovitis RF 54/F Both shoulder 14 mo Inflammatory bursitis Treatment Follow-up duration Teno 5 yr Radical Synovectomy 1 yr Surgery without Subtotal Synovectomy Prognosis Reference yr yr mo F: female, M: male, ANA: anti-nuclear antibodies, AFB: acid-fast bacilli, CRP: C-reactive protein, ESR: erythrocyte sedimentation rate, HLA: human histocompatibility leukocyte antigen, PPD: purified protein derivative, RF: rheumatoid factor, : ence. DISCUSSION Rice bodies were most commonly detected in rheumatoid arthritis [2], tuberculous arthritis, and bursitis [5,6]. Rice bodies had been found in 72% of joints affected by rheumatoid arthritis after aspiration and lavage of syno318 vial fluid [2]. These materials could occur adjunctive to inflamed synovium, in the bursa [3], and around tendon sheaths [4], as well as in the pleural fluid of patients with rheumatoid arthritis [7]. They have also been reported in patients with juvenile arthritis [8], sero spondyloarthritis [2], and osteoarthritis [2]. In the tuberculous J Rheum Dis Vol. 23,. 5, October, 2016

4 Idiopathic Massive Rice Bodies in the Knee Joint arthritis and bursitis, it had been also often combined with rice bodies [5,6]. Rice bodies have been found in various joints including shoulder, knee, wrist, and elbow [2]. MRI is a helpful diagnostic tool for rice bodies in the synovial space and bursa ahead to operation. The MRI features of rice bodies are described as iso- or hypo-intense on T1-weighted and T2-weighted images. They are difficult to distinguish from bursal and synovial fluid on T1-weighted images, and a honeycomb-like pattern could be seen from the border of each body that is distinguished from the surrounding fluid on T2-weighted images, as shown in our report [9]. These appearances are not specific findings of rice bodies, and they have to be differentiated from various benign synovial proliferative disorders, including chronic synovial proliferation, pigmented villonodular synovitis, and synovial osteochondromatosis [9,10]. The etiology of rice bodies is not fully understood, but they might originate from microinfarction of the synovium due to chronic inflammation [10]. Microscopic findings of rice bodies had shown an amorphous core surrounded by thin fibrin, and the core compositions were similar to those of inflamed synovium including types I, II, and V collagen and microvasculature [10]. Thus, floating materials might originate from proliferative synovium due to chronic inflammation. However, rice bodies had been rarely reported in the joint with nonspecific synovitis as shown in Table 1. Several cases with rice bodies of the wrist joints and flexor tendon sheath in the absence of rheumatic disease or tuberculosis have been reported, and these might be caused by overuse because they usually occurred on the patient s dominant side [11]. And two cases of rice bodies in the knee joint with nonspecific synovitis have been reported, and both patients had no signs or laboratory findings of rheumatoid arthritis or infection [12,13]. And pediatrics also had rice bodies in knee and shoulder joints without definite inflammatory articular diseases [13,14]. All cases with idiopathic rice bodies had been accompanied with synovitis or tenosynovitis and any symptoms had not been ed after only teno. Rice bodies without any inflammatory signs in the joint have not been previously reported. In our report, erythrocyte sedimentation rate (ESR) and C-reactive protein (CRP) were within range, and no definite clinical features reflecting inflammation such as tenderness or joint warmth were noted. Results of culture and PCR for tuberculosis and NTM were, and all ligaments and menisci were intact. Furthermore, arthroscopic finding and pathology of the synovium showed clean synovium without definite inflammatory cells infiltrates. Based on our case and literature reviews, we have to check immunologic markers including rheumatoid factor (RF), anti-citrullinated protein antibody (ACPA), anti-nuclear antibodies (ANA) and HLA B27 and culture studies when rice bodies in the joint space are suspected in ultrasonography and MRI. Idiopathic rice bodies with nonspecific tenosynovitis could be clearly cured by teno. And idiopathic rice bodies without any inflammatory reaction also cloud be cured by surgical removal for relieving symptoms without additional medical treatments. SUMMARY Rice bodies were commonly detected in the synovial space in patients with rheumatoid arthritis or tuberculosis and could often be found in the bursa and around tendon sheaths. The etiology of rice bodies had not been clearly understood, but they might be formed from chronic inflammation. However, we reported that massive rice bodies in the suprapatellar space were founded without any evidence of synovitis in the knee joint. Therefore, the pathogenesis of rice bodies must be studied in addition to inflammation in the future. CONFLICT OF INTEREST potential conflict of interest relevant to this article was reported. REFERENCES 1. Reise H. Die Reiskörperchen in tuberculös erkrankten Synovialsäcken. Deutsch Z Chir 1895;42: Popert AJ, Scott DL, Wainwright AC, Walton KW, Williamson N, Chapman JH. Frequency of occurrence, mode of development, and significance or rice bodies in rheumatoid joints. Ann Rheum Dis 1982;41: Thevenon A, Cocheteux P, Duquesnoy B, Mestdagh H, Lecomte-Houcke M, Delcambre B. Subacromial bursitis with rice bodies as a presenting feature of sero rheumatoid arthritis. Arthritis Rheum 1987;30: Bulut M, Yilmaz E, Karakurt L, Özercan MR. Rice body formation characterized by the chronic non-specific tenosynovitis in the tibialis anterior tendon. Acta Orthop Traumatol Turc 2013;47: Kim RS, Lee JY, Jung SR, Lee KY. Tuberculous subdeltoid bursitis with rice bodies. Yonsei Med J 2002;43:

5 Whan Yong Chung et al. 6. Chau CL, Griffith JF, Chan PT, Lui TH, Yu KS, Ngai WK. Rice-body formation in atypical mycobacterial tenosynovitis and bursitis: findings on sonography and MR imaging. AJR Am J Roentgenol 2003;180: Kassimos D, George E, Kirwan JR. Rice bodies in the pleural aspirate of a patient with rheumatoid arthritis. Ann Rheum Dis 1994;53: Chung C, Coley BD, Martin LC. Rice bodies in juvenile rheumatoid arthritis. AJR Am J Roentgenol 1998;170: Griffith JF, Peh WC, Evans NS, Smallman LA, Wong RW, Thomas AM. Multiple rice body formation in chronic subacromial/subdeltoid bursitis: MR appearances. Clin Radiol 1996;51: Cheung HS, Ryan LM, Kozin F, McCarty DJ. Synovial origins of Rice bodies in joint fluid. Arthritis Rheum 1980;23: Forse CL, Mucha BL, Santos ML, Ongcapin EH. Rice body formation without rheumatic disease or tuberculosis infection: a case report and literature review. Clin Rheumatol 2012;31: Kang DJ, Ahn JM, Rhee SJ. A case of multiple rice bodies by the nonspecific synovitis in the knee joint. J Korean Knee Soc 2010;22: Mutlu H, Silit E, Pekkafali Z, Karaman B, Omeroglu A, Basekim CC, et al. Multiple rice body formation in the subacromial-subdeltoid bursa and knee joint. Skeletal Radiol 2004;33: Aşik M, Eralp L, Cetik O, Altinel L. Rice bodies of synovial origin in the knee joint. Arthroscopy 2001;17:E J Rheum Dis Vol. 23,. 5, October, 2016