Title: Pigmented villonodular synovitis of knee in a patient on oral anticoagulation therapy. A Case report

Transcription

1 Author's response to reviews Title: Pigmented villonodular synovitis of knee in a patient on oral anticoagulation therapy. A Case report Authors: Balasundaram Ramesh (orthoygc@yahoo.com) Sanathkumar Shetty (sanatusha@yahoo.com) Salah S Bastawrous (Salah.Bastawrous@cd-tr.wales.nhs.uk) Version: 3 Date: 29 April 2008 Author's response to reviews: see over

2 Pigmented villonodular synovitis of knee in a patient on oral anticoagulation therapy-a case report Balasundaram Ramesh 1*, Sanathkumar Shetty 1, Salah.S.Bastawrous FRCSEd 1. Address: 1 Department of Trauma & Orthopaedics, Glan Clwyd Hospital, Sarn Lane, Bodelwyddan, Rhyl, North Wales, LL18 5UJ - Sanathkumar Shetty- sanatusha@yahoo.com S.S.Bastawrous- Salah.Bastawrous@cd-tr.wales.nhs.uk * Corresponding author: Balasundaram Ramesh M.B.B.S, FRCS (Glasgow) Department of Trauma &Orthopaedics Glan Clwyd Hospital Sarn Lane, Bodelwyddan Rhyl, North Wales LL18 5UJ - orthoygc@yahoo.com Tel. No Ext-3536 Fax.no

3 Abstract Introduction Pigmented villo nodular synovitis is a disease which affects synovial joints and tendon sheaths. Though the exact aetiological factor is not known, we feel that recurrent haemarthrosis has a role in the aetiology of this condition. Case presentation A case is presented of a 62 year old Caucasian male who presented with progressive worsening of symptoms in his knee. The patient was on anticoagulation therapy and was seen and treated for recurrent spontaneous haemarthrosis of the knee. The International Normalized Ratio done on all these occasions suggested poor control of the anticoagulation therapy. The symptoms in the knee worsened and a diagnosis of Pigmented villo nodular synovitis was made based on the intraoperative findings and this was confirmed by histopathological examination. Conclusion This report is presented to highlight the unusual association of haemarthrosis and pigmented villo nodular synovitis. 2

4 Introduction Pigmented villo nodular synovitis is a disease of unknown aetiology affecting the synovial joints. The etiology of Pigmented villo nodular synovitis remains controversial. A number of theories have been postulated. Haemarthrosis has also been suggested as a possible aetiological factor. There has been just one previous report describing Pigmented villonodular synovitis of the ankle in a patient on anticoagulation therapy. We describe a case of pigmented villonodular synovitis of knee joint in a patient on anticoagulation therapy. Case Report A 62-year-old Caucasian male, who previously had an uneventful total knee replacement on the right side five years previously, was admitted with recurrent effusions and worsening of pain in the left knee. Two years following the total knee replacement on the right side the patient was diagnosed with a dilated cardiomyopathy and was started on warfarin. He was seen and treated in the emergency department with recurrent episodes of spontaneous swelling in his left knee. The INR on these occasions were higher than the therapeutic range. There was no history of trauma. The patient was systemically well and his blood test for full blood count, C-reactive protein was within normal range. The pain in the left knee gradually worsened over time. On examination, knee joint was a diffusely swelling and tender. There was no evidence of instability. His active range of movement was from neutral to 100 degrees of flexion and further flexion was painful. Radiographs of the knee showed advanced arthritic changes and a decision to proceed with total knee replacement was taken to alleviate his symptoms Intra-operatively, it was seen that the synovium was hypertrophic and stained reddish orange. The synovial fluid was reddish-orange in colour (Fig.1). These appearances suggested a diagnosis of PVNS. A Synovectomy was performed followed by total knee replacement. The synovial specimen was sent for histopathological examination. The microscopic feature was consistent with diagnosis of PVNS (Fig.2). The post-operative period was uneventful and the patient is asymptomatic after three-years of follow-up. 3

5 Discussion Since the first description of this condition by Jaffe et. al. in 1941 [1], the etiology of this benign tumour involving the synovial membrane remains unclear. Jaffe proposed a hypervascular cellular phase subsequent to trauma produced hyalinization and fibrosis [1]. The etiology of the condition is unknown, although varyious etiologies including trauma [2] inflammation [3] hemorrhage [4] neoplasia [5] and genetic factors [6] have been suggested. It has also been postulated that pigmented villonodular synovitis in children arises through different mechanism than adults and it is also possible that not all lesions interpreted as pigmented villonodular synovitis share the same mechanism [7]. Pigmented villonodular synovitis typically occurs in adults in their third and fourth decades of life, with male/female ratio of 1.9/1.3. Involvement is usually monoarticular [2]. The knee joint is the most usual site and the fingers, feet, ankles, hips, wrists and shoulders are affected in decreasing order of frequency [2,3]. Chronic recurrent microtrauma and haemarthrosis have been postulated [2]. There have been many isolated cases of PVNS reported in patients with on anticoagulation therapy [8] and bleeding disorder [9]. In our patient the symptoms in the knee worsened following these repeated episodes of haemarthrosis and the INR on these occasions showed a poor control of his anticoagulation therapy. Conclusion This case augments the argument put forth by earlier reports [8, 9] that, repeated haemarthrosis may have a role in the etiology of Pigmented villo nodular synovitis and encourages the reporting of 4

6 similar cases in the hope that it might lead to a better understanding of the etiology of this condition. Abbreviation- PVNS- pigmented villonodular synovitis. INR- International Normalized ratio Authors Contributions SS made substantial contributions to acquisition of data, reviewing literature and preparing the manuscript. BR performed the knee replacement operation and also contributed in reviewing literature and drafting the manuscript. SSB gave final approval to the draft to be published. Competing interests The authors declare that they have no competing interests. Consent The patient gave written informed consent for publishing his data as case report. 5

7 References 1. Jaffe HL, Lichtenstein L, Sutro CJ: Pigmentedvillonodular synovitis, bursitis and tenosynovitis.arch Pathol 1941; 31: Myers BW, Masi AT: Pigmented villonodular synovitis and tenosynovitis: A clinical epidemiologic study of 166 cases and literature review. Medicine 1980; 19: Granowitz SP, O'Antorio J, Mankin H: The pathogenesis and long term end results of pigmented villonodular synovitis. Clin Orthop Rel Res 1976; 114: Leszczynski JR, Huckell JS, Perry JC, et al: Pigmented villonodular synovitis in multiple joints, Occurrence in a child with cavernous hemangioma of lip and pulmonary stenosis. Ann Rheum Dis 1975; 34: Rao AS, Vigorite VJ: Pigmented villonodular synovitis (giant cell tumor of tendon sheath and synovial membrane). Review of 81 cases. J Bone Joint Surg Am 1984; 66: Wendt RG, Wolfe F, MC Queen D, et al: Pigmented villonodular synovitis in children: evidence for genetic contribution. J Rheumatol 1986; 13(5):

8 7. Schumacher HR, Lotke P, Athreya B, et al: Pigmented villonodular synovitis: Light and electron microscopic studies. Semin Arth Rheumatol 1982; 129(1): Pearse OE, Klass B, Bendall SP. Pigmented Villonodular synovitis of the ankle occuring in a patient on anticoagulation therapy. J Surg Orthop Adv. 2004; 13 (4): Matsui H, Takahashi Y, Matsunaga T, Tanaka-Horie T, Minowa H, Sugimoto M, Tsukino R, Mii Y, Giddings J, Yoshioka A. Successful Arthroscopic Treatment of Pigmented Villonodular Synovitis of the Knee in a Patient with Congenital Deficiency of Plasminogen Activator Inhibitor-1 and Recurrent haemarthrosis. Haemostasis 2001; 31:

9 Legends Fig.1- Intraoperative photographs showing the reddish-orange stained synovium (black arrow). Fig.2- photomicrograph showing the presence of haemosiderin deposits, foamy histiocytes and giant cells confirming Pigmented Villonodular Synovitis (H&E). 8