Clinical Review

Pigmented Villonodular Synovitis of the Hip: A Systematic Review

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Pigmented villonodular synovitis is a rare proliferative condition of the synovium that affects large joints. The primary treatment options are synovectomy and a combination of synovectomy and arthroplasty.

We performed a systematic review of the literature, excluding all nonclinical and review articles with follow-up of less than 2 years. Primary outcomes reported were disease recurrence, symptom progression, and revision surgery. Student t tests were used to compare outcomes after synovectomy with outcomes after synovectomy combined with arthroplasty. Twenty-one studies (82 patients) were included. All represented level IV or V evidence. Fifty-one patients (59.3%) were female. Mean (SD) age was 33.2 (12.6) years. Synovectomy alone was performed in 45 patients (54.9%), and synovectomy with arthroplasty was performed in 37 patients (45.1%). Mean (SD) follow-up was 8.4 (5.9) years. The groups’ revision rates were not significantly different (26.2% vs 24.3%; P = .17). Mean (SD) time to revision was significantly (P = .02) longer in the synovectomy-with-arthroplasty group, 11.8 (4.5) years, than in the synovectomy-only group, 6.5 (3.9) years. Study results showed revisions are common after surgery for hip pigmented villonodular synovitis, affecting 1 in 4 patients regardless of which surgery they have—either synovectomy alone or synovectomy combined with arthroplasty. Revision is required sooner in synovectomy-only patients than in patients who also undergo arthroplasty.


 

References

Pigmented villonodular synovitis (PVNS) is a rare monoarticular disorder that affects the joints, bursae, or tendon sheaths of 1.8 per million patients.1,2 PVNS is defined by exuberant proliferation of synovial villi and nodules. Although its etiology is unknown, PVNS behaves much as a neoplastic process does, with occasional chromosomal abnormalities, local tissue invasion, and the potential for malignant transformation.3,4 Radiographs show cystic erosions or joint space narrowing, and magnetic resonance imaging shows characteristic low-signal intensity (on T1- and T2-weighted sequences) because of high hemosiderin content. Biopsy remains the gold standard for diagnosis and reveals hemosiderin-laden macrophages, vascularized villi, mononuclear cell infiltration, and sporadic mitotic figures.5 Diffuse PVNS appears as a thickened synovium with matted villi and synovial folds; localized PVNS presents as a pedunculated, firm yellow nodule.6

PVNS has a predilection for large joints, most commonly the knee (up to 80% of cases) and the hip.1,2,7 Treatment strategies for knee PVNS have been well studied and, as an aggregate, show no superiority of arthroscopic or open techniques.8 The literature on hip PVNS is less abundant and more case-based, making it difficult to reach a consensus on effective treatment. Open synovectomy and arthroplasty have been the mainstays of treatment over the past 60 years, but the advent of hip arthroscopy has introduced a new treatment modality.1,9 As arthroscopic management becomes more readily available, it is important to understand and compare the effectiveness of synovectomy and arthroplasty.

We systematically reviewed the treatment modalities for PVNS of the hip to determine how synovectomy and arthroplasty compare with respect to efficacy and revision rates.

Methods

Search Strategy

We systematically reviewed the literature according to PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) guidelines using the PRISMA checklist.10 Searches were completed in July 2014 using the PubMed Medline database and the Cochrane Central Register of Clinical Trials. Keyword selection was designed to capture all level I to V evidence English-language studies that reported clinical and/or radiographic outcomes. This was accomplished with a keyword search of all available titles and manuscript abstracts: (pigmented [Title/Abstract] AND villonodular [Title/Abstract] AND synovitis [Title/Abstract]) AND (hip [Title/Abstract]) AND (English [lang])). Abstracts from the 75 resulting studies were reviewed for exclusion criteria, which consisted of any cadaveric, biomechanical, histologic, and/or kinematic results, as well as a lack of any clinical and/or radiographic data (eg, review or technique articles). Studies were also excluded if they did not have clinical follow-up of at least 2 years. Studies not dedicated to hip PVNS specifically were not immediately excluded but were reviewed for outcomes data specific to the hip PVNS subpopulation. If a specific hip PVNS population could be distinguished from other patients, that study was included for review. If a study could not be deconstructed as such or was entirely devoted to one of our exclusion criteria, that study was excluded from our review. This initial search strategy yielded 16 studies.1,6,7,11-28

Bibliographical review of these 16 studies yielded several more for review. To ensure that no patients were counted twice, each study’s authors, data collection period, and ethnic population were reviewed and compared with those of the other studies. If there was any overlap in authorship, period, and place, only the study with the most relevant or comprehensive data was included. After accounting for all inclusion and exclusion criteria, we selected a total of 21 studies with 82 patients (86 hips) for inclusion (Figure 1).

Data Extraction

Details of study design, sample size, and patient demographics, including age, sex, and duration of symptoms, were recorded. Use of diagnostic biopsy, joint space narrowing on radiographs, treatment method, and use of radiation therapy were also abstracted. Some studies described multiple treatment methods. If those methods could not be differentiated into distinct outcomes groups, the study would have been excluded for lack of specific clinical data. Studies with sufficient data were deconstructed such that the patients from each treatment group were isolated.

Fewer than 5 studies reported physical examination findings, validated survey scores, and/or radiographic results. Therefore, the primary outcomes reported and compared between treatment groups were disease recurrence, clinical worsening defined as progressive pain or loss of function, and revision surgery. Revision surgery was subdivided into repeat synovectomy and eventual arthroplasty, arthrodesis, or revision arthroplasty. Time to revision surgery was also documented. Each study’s methodologic quality and bias were evaluated with the Modified Coleman Methodology Score (MCMS), described by Cowan and colleagues.29 MCMS is a 15-item instrument that has been used to assess randomized and nonrandomized patient trials.30,31 It has a scaled potential score ranging from 0 to 100, with scores from 85 through 100 indicating excellent, 70 through 84 good, 55 through 69 fair, and under 55 poor.

Pages

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