Sambrook P N, de Jager J P, Champion G D, Doust B D, McGirr E E, Kozlowski K S, Sillence D O
Department of Rheumatology, St. Vincent's Hospital, Sydney, Australia.
Arthritis Rheum. 1988 Feb;31(2):282-7. doi: 10.1002/art.1780310219.
We describe 2 patients with a late-onset type of spondylepiphyseal dysplasia who developed multiple synovial complications. The synovial manifestations in the first patient included recurrent episodes of acute arthritis due to calcium pyrophosphate dihydrate crystal deposition and, later, chronic synovitis with radiologically evident chondrocalcinosis. In the second patient, cholesterol crystals in synovial fluid, multiple osteochondromata, and a histologic appearance resembling pigmented villonodular synovitis on synovial biopsy were seen. Osteochondromata were identified in 4 other members of this patient's family. Synovial manifestations in the osteochondrodysplasias may be more common than previously recognized.
我们描述了2例迟发型脊椎骨骺发育不良患者,他们出现了多种滑膜并发症。首例患者的滑膜表现包括因二水焦磷酸钙晶体沉积导致的急性关节炎反复发作,以及后来出现的慢性滑膜炎,影像学检查显示有软骨钙质沉着症。在第二例患者中,发现其滑液中有胆固醇晶体、多发骨软骨瘤,滑膜活检的组织学表现类似于色素沉着绒毛结节性滑膜炎。在该患者家族的其他4名成员中也发现了骨软骨瘤。骨软骨发育不良中的滑膜表现可能比之前认为的更为常见。