© 2014 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd

J Cutan Pathol 2014: 41: 602–605 doi: 10.1111/cup.12316 John Wiley & Sons. Printed in Singapore

Journal of Cutaneous Pathology

Tenosynovitis with rice body formation presenting as a cutaneous abscess A 62-year-old woman with a past medical history of rheumatoid arthritis was referred to the Department of Dermatology because of an enlarging cutaneous lesion on the right thumb which resembled a soft tissue infection. She had received antibiotics without significant improvement. Clinical examination revealed an erythematous nodule involving almost the whole surface of the distal phalanx with spontaneous drainage of countless of small yellowish ovoid granules. Histopathologic study of these structures showed an inner core of amorphous acidophilic material with some interspersed chronic inflammatory cells and a surrounding thin fibrin layer. Special stains and cultures were negative for parasites, bacterium and mycobacterium. Magnetic resonance imaging (MRI) revealed distension of the first and fifth finger flexor sheaths and common finger flexor sheath. These areas were filled by fluid and multiple small nodular lesions. A diagnosis of non-infectious rice body tenosynovitis was rendered and surgical removal was performed. Total recovery was observed with no evidence of recurrence after 6 months of follow-up. To our knowledge, this is the first report of rice body tenosynovitis presenting as a pseudoinflammatory cutaneous lesion with evolution to a cutaneous fistula with drainage of rice grain-like structures. The description of this impressive and peculiar clinical and histopathologic picture is important to further recognize similar cases. Keywords: cutaneous abscess, rice body formation, skin fistula, tendon sheath, tenosynovitis Moreno S, Forcada P, Soria X, Altemir V, Gatius S, Gil M, Matías-Guiu X, Casanova JM, Martí RM. Tenosynovitis with rice body formation presenting as a cutaneous abscess. J Cutan Pathol 2014; 41: 602–605. © 2014 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd

Although initially described in tuberculous arthritis, rice body formation in joints or bursae is a common phenomenon related to rheumatoid arthritis.1,2 It can also been observed in association with systemic lupus erythematosus, seronegative arthritis, infectious arthritis, non-specific arthritis and osteoarthritis. However, multiple rice bodies inside the tendon sheaths are usually seen in tuberculous tenosynovitis

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Sara Moreno1 , Pau Forcada2 , Xavier Soria1 , Victoria Altemir2 , Sonia Gatius3 , Mabel Gil4 , Xavier Matías-Guiu3 , Josep M. Casanova1 and Rosa M. Martí1 1

Department of Dermatology, Hospital Universitari Arnau de Vilanova, Universitat de Lleida, IRB Lleida, Lleida, Spain, 2 Department of Orthopaedic Surgery, Hospital Universitari Arnau de Vilanova, Universitat de Lleida, IRB Lleida, Lleida, Spain, 3 Department of Pathology and Molecular Genetics, Hospital Universitari Arnau de Vilanova, Universitat de Lleida, IRB Lleida, Lleida, Spain, and 4 Department of Diagnostic Radiology and Organ Imaging, Hospital Universitari Arnau de Vilanova, Universitat de Lleida, IRB Lleida, Lleida, Spain

Sara Moreno, MD Department of Dermatology, Hospital Universitari Arnau de Vilanova, Avda. Alcalde Rovira Roure 80, Lleida 25198, Spain Tel: +34 973 705238 Fax: +34 973 702435 e-mail: [email protected] Accepted for publication February 9, 2014

and rarely encountered among non-tuberculosis patients.2,3 Herein, we describe the first reported case of non-infectious rice body tenosynovitis of the first and fifth tendon sheaths that clinically presented mimicking a cutaneous abscess that evolved to skin fistula formation with drainage of multiple rice bodies. Dermatologists and dermatopathologists are

Tenosyovitis with rice bodies A

B

Fig. 1. The clinical appearance is depicted.

not familiar with the clinical and histopathologic features of this disorder, because it is unusual in routine non-rheumatologic practice. Report of a patient In February 2012, a 62-year-old woman with seropositive rheumatoid arthritis sought medical advice for a 2-month history of a non-tender enlarging lesion on the distal side of the thumb of the right hand. There was no associated history of fever, loss of weight or appetite, night’s sweats, malaise or fatigue. There was no history of trauma. The patient had been treated with antibiotics and topical antifungicals without any significant improvement. Physical examination revealed erythema and swelling involving more than half of the second phalanx of the first finger of the right hand. On the external aspect of this area, a central crater filled with a peculiar yellowish material was present. The crater opened by pressure and drained countless shiny A

yellow-whitish ovoid granular structures, 2–4 mm in size (Fig. 1), which were subjected to pathologic evaluation. The patient was then referred to the Trauma and Orthopedics Department. Initially the patient did not note difficulty with movement of the thumb. A radiograph of the first finger did not show significant changes. Drainage of some ovoid yellow-whitish nodules continued during the next 15 days, and the patient noted difficulty on motility. A magnetic resonance of the hand and wrist revealed distension of the first and fifth finger flexor sheaths up to both distal phalanges and common flexor finger sheath in her metacarpal path. The distended structures contained a fluid collection and multiple tiny areas of low-signal intensity on T2-weighted and T1 isointensity with-in the fluid (Fig. 2). On the basis of these findings, a diagnosis of rice body tenosynovitis was rendered. Surgical intervention was performed and revealed multiple ovoid shiny bodies in the thickened common flexor tendon sheath extending distally through the B

Fig. 2. A and B) Magnetic resonance imaging (MRI) shows rice bodies as hypointense areas (arrows).

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Moreno et al. Special stains for parasites, fungi or acid fast bacilli, detection of Mycobacterium tuberculosis by polymerase chain reaction (PCR) analysis and cultures for aerobic or anaerobic microorganisms or mycobacterium were negative. Stains for amyloid were also negative. Pathological examination of the tissue obtained from the surgical intervention revealed synovial tissue with chronic synovitis and fibrinoid necrosis. There was focal synovial hyperplasia. The synovial surface was focally denuded, and covered by fibrin. Chronic inflammatory cells were present. Some tendon sheets contained amorphous, eosinophilic material, as well as focal fibrinoid necrosis, which were interpreted as initial changes of the process. Some tendon sheets were also surrounded by fibrin. There were no epithelioid granulomata or giant cells. Again, special stains and cultures were negative.

sheath to the tip of the first and fifth finger. Sheaths of the three central fingers were preserved. The excised tissue was submitted for pathologic evaluation. Total recovery was observed following surgery, and there has been no evidence of recurrence with 6 months of follow-up. Histopathologic findings Microscopic examination of the material initially drained showed multiple rounded to ovoid corpuscles with smooth margins (Fig. 3A). Most of the bodies were composed of an inner core of amorphous acidophilic material, but some others had a central area with vascularized collagenous tissue. The amorphous material that was present in central part of the corpuscles was brightly eosinophilic. Some interspersed chronic inflammatory cells were also present. A thin fibrin layer was frequently seen surrounding the ovoid corpuscles (Fig. 3B and C). Some fragments were exclusively composed of condensed fibrin, which had the typical reticular eosinophilic appearance, but also contained numerous vacuoles of varying sizes. In some fragments there was a close spatial relationship between organized fibrin and immature collagenous tissue, with transition areas between fibrin and early fibrous tissue. Occasional deposits of hemosiderin were also seen. The rounded to ovoid corpuscles did not contain cartilage or bone. Epithelioid granulomas were not present. A

Discussion Rice bodies are soft tissue collections of variable size which macroscopically resemble shiny white rice grains and microscopically consist of an inner amorphous core of acidophilic material, with or without interspersed chronic inflammatory cells, surrounded by fibrin and collagen.2,3 Rice bodies occurring in joints affected by tuberculosis were first described in 1895.4 They are a common finding in chronic diseases involving joints or bursae including many B

C

Fig. 3. A–C) Rice bodies are shown histopathologically at low and high magnification.

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Tenosyovitis with rice bodies rheumatic diseases such as rheumatoid arthritis, systemic lupus erythematosus and seronegative arthritis, as well as infectious diseases such as non-specific tuberculous arthritis5,6 and atypical mycobacterium infections. Also, they may be found in osteoarthritic joints.7 However, multiple rice bodies within tendon sheaths are rarely encountered among non-tuberculosis patients.8 – 10 In rheumatoid arthritis, rice bodies are often observed in the subacromial bursa but are uncommon in the tendon sheaths, as was seen in this patient. In this case, the personal medical history, histopathologic findings, PCR studies and cultures all excluded the diagnosis of tuberculosis.11 An interesting feature in our case was not only the formation of rice bodies in tendon sheaths, but also the clinical presentation as a cutaneous abscess draining multiple rice grain-like structures through the skin. To our knowledge, no case of tenosynovitis with rice body formation and cutaneous fistula formation has been described in the literature before. The pathogenesis of rice body formation is unclear.12 Some investigators have suggested that they are the result of microinfarctions after intraarticular synovial inflammation and ischemia with subsequent synovial shedding and encasement by fibrin derived from synovial fluid. Other authors proposed that rice bodies are formed de novo in synovial fluid independently of synovial elements and progressively enlarge with aggregation of fibrin.2,3 MRI has been proposed as the best imaging procedure to detect rice bodies. Using MRI, rice

bodies have been described as iso or hypointense areas with effusion on T1 and T2-weighted images. The rice bodies found our patient showed similar signal intensity.1,13 Because multiple rice body formation involving the tendon sheath is rare, limited information exists regarding therapy. However, tenosynovectomy is performed to avoid complications such as tendon rupture and persisted motility restriction.3,11,14,15 In conclusion, we have presented a case of 62year-old woman with rice body formation because of tenosynovitis of the flexor tendon sheath of the fingers, associated with rheumatoid arthritis, presenting as a dermal abscess, and subjected to dermatopathologic analysis. Although it is a rare entity for dermatologists or even for orthopedic surgeons and rheumatologists, we should keep in mind its clinical features that will allow an early diagnosis and correct management of the disease avoiding unnecessary treatments such as antibiotics. From the histopathologic point of view, the macroscopic and microscopic appearance is quite unique and diagnosis is easy. However, the microscopic features are not well described in dermatopathology textbooks, because the process is not usually seen in dermatopathology practice. Acknowledgments Supported by grants from ISCIII (PI1200260 to RMM) and from Generalitat de Catalunya (2009SGR794 to XMG). SM holds a predoctoral fellowship from IRBLleida/Diputaci´o de Lleida.

References 1. Amrami KK, Ruggleri AP, Sundaram M. Radiologic case study. Rheumatoid arthritis with rice bodies. Orthopaedics 2004; 27: 426. 2. Popert AJ, Scott DL, Wainwright AC, Walton KW, Williamson N, Chapman JH. Frequnecy of occurrence, mode of development, and significance or rice bodies in rheumatoid joints. Ann Rheum Dis 1982; 41: 109. 3. Ergun T, Lakadamyali H, Aydin O. Multiple rice body formation accompanying the chronic non-specific tenosynovitis of the flexor tendons of the wrist. Radiat Med 2008; 26: 545. 4. Reise H. Die Reiskorpschen in tuberculs erranken synovalsacken. Dtsch Z hir 1895; 42: 1. 5. Pimm LH, Waugh W. Tuberculous tenosynovitis. J Bone Joint Surg 1957; 39B: 91. 6. Cheung HS, Ryan LM, Kozin F, McCarty DJ. Synovial origins of rice bodies in joint fluid. Arthritis Rheum 1980; 23: 72.

7. Bucki B, Lansaman J, Janson X, BillionGalland MA, Marty C, Ruel M. Osteoarthritis with rice bodies rich in calcium microcrystals. 4 cases with ultrastructural study. Rev Rhum Ed Fr 1994; 61: 415. 8. 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. Am J Roentgenol 2003; 180: 1455. 9. Sugano H, Nagao T, Tajima Y, Ishida Y, Nagao K, Ohno T. Variation among giant rice bodies: report of four cases and their clinicopathological features. Skeletal Radiol 2000; 29: 525. 10. Lee EY, Rubin DA, Brown DM. Recurrent mycobacterium marinum tenosynovitis of the wrist mimicking extrarticular synovial chondromatosis on MR images. Skeletal Radiol 2004; 33: 405.

11. Iyengar K, Manickavasagar T, Nadkarni J, Mansour P, Loh W. Bilateral recurrent whist flexor tenosynovitis and rice body formation in a patient with sero-negative rheumatoid arthritis: A case report and review of literature. Int J SUrg Case Rep 2011; 2: 208. 12. Tyllianakis M, Kasimatis G, Athanaselis S, Melachrinou M. Rice-body formation and tenosynovitis of the wrist: a case report. J Orthop Surg 2006; 14: 208. 13. Nagasawa H, Okada K, Senma S, Chida S, Shimada Y. Tenosynovitis with rice body formation in a non-tuberculous patient: a case report. Ups J Med Sci 2009; 114: 184. 14. Cuomo A, Pirpiris M, Outsuka NY. Case report: biceps tenosynovial rice bodies. J Pediatr Orthop B 2006; 15: 423. 15. Suso S, Peidro L, Ramon R. Tubercolous synovitis with ‘‘rice bodies’’ presenting as carpal tunnel syndrome. J Hand Surg 1988; 13: 574.

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Tenosynovitis with rice body formation presenting as a cutaneous abscess.

A 62-year-old woman with a past medical history of rheumatoid arthritis was referred to the Department of Dermatology because of an enlarging cutaneou...
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