Pigmented villonodular synovitis: Monoclonality and metastasis—a case for neoplastic origin?

Choong, Peter F. M.; Willén, Helena; Nilbert, Mef; Mertens, Fredrik; Mandahl, Nils; Carlén, Birgitta; Rydholm, Anders

doi:10.3109/17453679508994643

Cited by 105 publications

(49 citation statements)

References 11 publications

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“…However, most consider that chronic inflammation is the underlying process [3,7,8]. Other authors have suggested that PVNS may be a neoplastic process, as, in rare cases, it may produce metastasis and evidence exists that some lesions are of the monoclonal type [9][10][11][12][13].…”

Section: Discussionmentioning

confidence: 99%

Pigmented villonodular synovitis of the knee in skeletally immature patients

Baroni

Russo

Masquijo

et al. 2010

Journal of Children's Orthopaedics

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Introduction Pigmented villonodular synovitis (PVNS) is a benign idiopathic proliferative disorder that results in villous or nodular formation in the joints, tendons sheaths, and bursae. As PVNS is a rare pathology in children, diagnosis is often delayed. In this study, we analyze the therapeutic methods used and results obtained in the treatment of this pathology. Materials and methods All patients with PVNS of the knee seen between January 1988 and June 2006 were evaluated. We assessed the form of presentation, time to diagnosis, previous diagnosis, type of treatment, relapse, and the need for subsequent treatment. Results Nine patients with age range 2-15 years and a mean follow-up of 8.5 years were evaluated. Four patients had the diffuse form and four had the localized or nodular form; all of them were intra-articular. In only three cases were preoperative radiographic findings observed. The mean delay in diagnosis was 18 months. Open resection was performed in five patients and arthroscopic resection in four. Joint function was satisfactory in 78% of the patients at the last follow-up and there were no postoperative recurrences. Conclusions Magnetic resonance imaging (MRI) is a useful diagnostic tool and the way to detect relapse, and allows accurate determination of the tumor extent. Surgery is the treatment of choice. Worse results are directly related to delay in diagnosis.

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Section: Discussionmentioning

confidence: 99%

Pigmented villonodular synovitis of the knee in skeletally immature patients

Baroni

Russo

Masquijo

et al. 2010

Journal of Children's Orthopaedics

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“…Cases of malignant PVNS have been reported [3,5]. Some authors have described the presence of cytogenetic clonal aberrations in short-term cultures and in uncultured PVNS cells, by using a fluorescence in situ hybridization technique [10,18,26].…”

Section: Discussionmentioning

confidence: 99%

Proliferating cell nuclear antigen labelling index in localised pigmented villo-nodular synovitis and its relationship to the size of nodules

Rosa

Galli

Fadda

et al. 2000

International Orthopaedics

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Proliferating cell nuclear antigen (PCNA) is one of the cell cycle-related proteins directly involved in DNA synthesis. It is a marker of cellular proliferation and has been shown to correlate with ploidy and proliferative activity of cells. Its expression has been used to estimate the growth fraction of human cancer and its prognostic value. Pigmented villo-nodular synovitis (PVNS) is characterised by a nodular lesion in the paratendinous synovial tissue or, less frequently, in a joint. Whether PVNS is a neoplastic or inflammatory lesion remains controversial. We have studied immunohistochemical PCNA expression with pc10 monoclonal antibody in 16 paraffin sections, in 16 cases of localised PVNS, or giant cell tumour of tendon sheath. We have found significant correlation between the size of the lesions and PCNA-LI (labelling index).Résumé L'antigène nucleaire de prolifération cellulaire (PCNA) est une des proteines du cycle cellulaire et il est impliqué directement dans la synthèse du DNA. Il s'agit certainement d'un indicateur de prolifération cellulaire. Son expression a été employé pour évaluer l'index de prolifération de la tumeur humaine et son valeur au point de vue pronostique. La synovite villo-nodulaire pigmentée (PVNS) est caractérisée par une lésion nodulaire plus fréquente dans les localisations tendineuses que dans les articulations. S'il s'agit d'un processus néoplasique ou d'une lésion inflammatoire reste controversée. Nous avons étudiés l'expression immunohistochimique du PCNA avec l'anticorps monoclonal PC10 en 16 lames paraffinés relatives à 16 synovites villo-nodulaires pigmentées localisées ou tumeurs à cellules geantes des gaines tendineuses. Nous avons trouvés une corrélation significative entre les dimensions des lésions nodulaires et le PCNA labelling index (PCNA-LI).

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“…Only 37 malignant tenosynovial GCTs have been reported in the literature since 1979, and these are summarized in ►Tables 1-3. [3][4][5][6][7][8][9][10][11][12][13][14][15][16][17] Of these 37 patients, follow-up outcome was known in 30 patients; one patient had died of other causes (DOC). Of the remaining 29 patients, 11 patients died of disease (DOD), 4 patients were living with disease (LWD) and 14 patients had no evidence of disease (NED) since their last treatment, with a follow-up of 6 months to 49 years.…”

Section: Discussionmentioning

confidence: 99%

Malignant Giant Cell Tumor in the Carpal Tunnel: A Case Report and Review of Literature

et al. 2013

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Giant cell tumors (GCTs) of the soft tissues are, based on their site of origin, divided into nontenosynovial and tenosynovial. Nontenosynovial GCTs are less common, and their biological behavior varies from borderline malignant to full-blown malignant. Tenosynovial GCTs, also known as GCTs of the tendon sheath or pigmented villonodular synovitis are nearly always benign, with a strong capacity for local recurrence because of their pattern of growth. 1,2 We present a case of malignant tenosynovial GCT in the carpal tunnel and a review of the literature. Case ReportA 66-year-old man, with tingling and a loss of sensation in the left hand, was treated in our center for a ganglion cyst in the carpal tunnel of the left hand (►Fig. 1). This ganglion was Keywords ► malignant giant cell ► carpal tunnel syndrome AbstractBackground Malignant tenosynovial giant cell tumors (GCTs) are extremely rare, and their etiology is unknown. However, this type of malignancy is associated with high metastasis and mortality rates. Therefore, the treatment of choice is wide excision. Case Description A 66-year-old man complained of tingling and loss of sensation in the left hand, caused by a tumor that compressed the median nerve. The tumor was excised. Histopathologic examination revealed a ganglion cyst. Two years later, the patient visited our clinic with recurrent and similar complaints of the left hand. This time, however, the lesion turned out to be a malignant tenosynovial GCT and was treated by amputation of the forearm. Literature Review Since 1979, only 37 malignant tenosynovial GCTs have been reported in literature. Follow-up of these patients showed that 11 patients died of the disease, 4 patients were still living with the disease, and 14 patients had no evidence of disease after treatment. The other seven patients were lost to follow-up, and one patient died of other causes. In these 37 patients, a high incidence of lymph node metastasis (41%) and a high mortality rate (30%) were seen. Clinical Relevance Although this malignant tenosynovial GCT is very rare, high mortality rates have been observed because of the high incidence of lymph node metastases. Therefore, more awareness has to be created, to recognize and treat this tumor timely.

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Pigmented villonodular synovitis: Monoclonality and metastasis—a case for neoplastic origin?

Cited by 105 publications

References 11 publications

Pigmented villonodular synovitis of the knee in skeletally immature patients

Pigmented villonodular synovitis of the knee in skeletally immature patients

Proliferating cell nuclear antigen labelling index in localised pigmented villo-nodular synovitis and its relationship to the size of nodules

Malignant Giant Cell Tumor in the Carpal Tunnel: A Case Report and Review of Literature

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