Pigmented villonodular synovitis of the knee in a patient on oral anticoagulation therapy: a case report
© Ramesh et al; licensee BioMed Central Ltd. 2009
Received: 07 January 2008
Accepted: 13 November 2009
Published: 13 November 2009
Pigmented villonodular synovitis is a disease which affects the synovial joints and tendon sheaths. Although the exact aetiological factors are not known, we believe that recurrent haemarthrosis has a role in the aetiology of this condition.
A 62-year-old Caucasian man presented with gradually worsening pain and stiffness in his right knee. The patient was on anticoagulation therapy and had been treated for recurrent episodes of spontaneous haemarthrosis of the knee. The International Normalized Ratio on each occasion suggested poor control of the anticoagulation therapy. A diagnosis of pigmented villonodular synovitis was made based on intra-operative findings and was further confirmed by a histopathological examination.
This report is presented to highlight the unusual association of haemarthrosis and pigmented villonodular synovitis.
Pigmented villonodular synovitis (PVNS) is a disease of unknown aetiology affecting the synovial joints. The aetiology of PVNS remains controversial and a number of theories have been postulated. Haemarthrosis has been suggested as a possible aetiological factor. Only one description of PVNS of the ankle in a patient on anticoagulation therapy has been previously reported ; we describe the second known case of PVNS of the knee joint in a patient on anticoagulation therapy.
A 62-year-old Caucasian man had an uneventful total left knee replacement five years prior to presentation. Two years following the total knee replacement, the patient was diagnosed with dilated cardiomyopathy and was started on warfarin. Following this, he had recurrent episodes of sudden pain and swelling on his right knee. During each of these episodes, there was no history of trauma and the patient was systemically well. Although his International Normalized Ratio (INR) was high, his blood tests for full blood count and C-reactive protein were within the normal range. All of these episodes were in the initial phase of his warfarin therapy and they ceased once his INR was stabilized.
PVNS typically occurs in adults in their third or fourth decade of life, with a male-to-female ratio of 1.9 to 1.3. Involvement is usually monoarticular . The knee joint is the most frequently affected site, followed by the fingers, feet, ankles, hips, wrists and shoulders in a decreasing order of frequency [2, 3].
Since the first description of this condition by Jaffe et al. in 1941 , the aetiology of this benign tumour involving the synovial membrane has remained unclear. Jaffe proposed that a hypervascular cellular phase occurs after trauma produc ing hyalinization and fibrosis . Various aetiologies including trauma , inflammation , haemorrhage , neoplasia  and genetic factors  have been suggested. Chronic recurrent microtrauma and haemarthrosis have also been postulated .
It has also been postulated that PVNS in children arises through a different mechanism to that in adults, and it is also possible that not all lesions interpreted as PVNS share the same mechanism .
There are very few cases of PVNS reported in patients on anticoagulation therapy  and with a bleeding disorder . 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.
Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
International Normalized Ratio
pigmented villonodular synovitis.
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