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Background: PVNS is a rare, benign &amp; aggressive disorder arising from either synovial joints or tendon sheaths; it may erode articular structures and bones. We present a case with unique features of PVNS being extra-synovial and by this report we open a gate for more researches in this field.  Case Presentation: This case report concerns a 35-year-old female with a history of right knee pain for 6-month duration proceeded by gradual swelling over posterior aspect of the knee, she denies any history of trauma, clinical examination was unremarkable but apart from tenderness over the infrapatellar region with full flexion.  MRI shows a heterogenous signal extra-articular and extra-synovial lesion in posterior aspect of the knee suggesting Pigmented Villonodular Synovitis, FNA revealed a hemosiderin-laden macrophages and multinucleated giant cells, Tru-cut biopsy result was suggesting PVNS as synovial cells were seen admixed with hemosiderin-laden macrophages with fibroblastic elements.  Through posterior approach; the lesion was surgically excised and histopathological examination confirmed the diagnosis, the lesion was recurrent after 1-year and MRI revealed the same features, the lesion was excised by arthroscopic intervention.  Conclusion: We concluded that PVNS cannot be excluded when extra-synovial lesion is assessed, and further researches on this topic will expand our understanding of the etiological and pathological aspects of this tumor.

Extra-synovial Pigmented Villonodular Synovitis of the knee joint, Case Repor