Simultaneous involvement of pigmented villonodular synovitis in the left shoulder glenohumeral joint and ankle joint: a rare case report

Abstract

Background: Pigmented Villonodular Synovitis, although uncommon, can lead to significant joint destruction if not diagnosed and treated early. Concurrent polyarticular presentation in adults is exceedingly rare, appearing in less than 1% of cases.

Case presentation: This case report presents a unique and rare occurrence of Pigmented Villonodular Synovitis involving both the ankle and shoulder joints simultaneously in an adult patient. The patient initially developed a diffuse form Pigmented Villonodular Synovitis in the left ankle, which was managed with total synovectomy at another tertiary hospital. Two months later, a separate localized form Pigmented Villonodular Synovitis was diagnosed in the left glenohumeral joint, necessitating arthroscopic marginal resection at our hospital. At the one-year follow-up, the magnetic resonance imaging of the left shoulder showed no signs of Pigmented Villonodular Synovitis recurrence, with full range of motion and no pain. However, the magnetic resonance imaging of the left ankle revealed a recurrence of the Pigmented Villonodular Synovitis, despite the range of motion remaining within normal limits.

Conclusions: This case demonstrates the importance of considering multiarticular Pigmented Villonodular Synovitis when symptoms manifest in multiple joints. Early diagnosis and appropriate intervention are critical to prevent joint destruction and optimize patient outcomes.

Keywords: Case report; Marginal resection; Multiarticular pigmented villonodular synovitis; Pigmented villonodular synovitis; Shoulder; Synovectomy.

Fig. 1

The Preoperative Plain Radiographs and MRI of the Ankle. A, B, C The images (Anterior Posterior, Mortise, and Lateral views) reveal tibiotalar osteoarthritis, with subchondral bone cystic changes in the talus and posterior tibia, as well as anterior osteophytes. D, E, F The images are shown on the axial, coronal, sagittal views, respectively, which illustrate a diffuse intraarticular dark signal intensity soft tissue proliferation within the tibiotalar joint in T2-weighted fat-saturated images accompanied by tibiotalar osteoarthritis

Fig. 2

Preoperative Plain Radiographs, MRI, and 3-Month Follow-up MRI of the Shoulder. A, B The images (Anterior Posterior and Axillary views) show no abnormalities. C, D The images display a nodular mass measuring approximately 3 cm within the axillary recess. In T2-weighted fat-saturated oblique coronal image, this mass shows heterogeneous high signal intensity with focal areas of hypointense signal, while in proton density fat-saturated axial image, it demonstrates intermediate high signal intensity. The image also reveals the presence of glenohumeral joint effusion accompanied by synovitis. Importantly, there are no communication detected between the subacromial space and the glenohumeral joint. E, F The 3-month postoperative follow-up MRI of the shoulder, shown in T2-weighted fat-saturated oblique coronal and proton density fat-saturated axial images, does not reveal any findings indicative of PVNS recurrence

Fig. 3

Arthroscopic and Gross Specimen Images. A A 2.7 × 1.5 × 2.0 cm sized yellowish-brown nodular mass attaches to the joint capsule of the axillary recess, causing impingement within the glenohumeral joint. B An arthroscopic view of the marginal resection of a mass, which is adherent to the joint capsule and synovium, using a grasper C An arthroscopic view of the inferior glenohumeral joint capsule after marginal resection and partial synovectomy. D Gross specimen image shows a well-defined yellowish-brown nodular mass measuring 2.7 × 1.5 × 2.0 cm

Fig. 4

A Microscopic Image (hematoxylin-eosin, x100). The image shows proliferating synovial-like cells with multinucleated giant cells, histiocytes, and hemosiderin-laden cells

Fig. 5

1-Year Follow-up MRI of the Shoulder and Ankle. A, B Follow-up MRI of the shoulder, shown in T2-weighted fat-saturated oblique coronal and proton density fat-saturated axial images, shows no signs of PVNS recurrence. C, D Follow-up MRI of the ankle, shown in T2-weighted fat-saturated sagittal and axial images, reveals the presence of a PVNS lesion, suggesting the possibility of recurrence