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MODERN RHEUMATOLOGY Vol.13 No.1

Ochronotic spondylarthropathy: two case reports of progressive destructive changes in the hip
M. Takagi1, H. Ida1, S. Kobayashi1, M. Ishii2, T. Osanai1, Y. T. Konttinen3
(1)Department of Orthopaedic Surgery, Yamagata University School of Medicine, 2-2-2 Iida-Nishi, Yamagata 990-9585, Japan
(2)Department of Orthopaedic Surgery, Saiseikai Yamagata Hospital, Yamagata, Japan
(3)Orton Research Institute, Orthopaedic Hospital of the Invalid Foundation, Helsinki Invartes Medicin, and Department of Internal Medicine, Helsinki University Central Hospital, Finland
 
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Abstract
Abstract We report two cases of ochronotic spondylarthropathy who presented with spinal involvement followed by progressive destructive changes in the hip joint, which led to total hip replacement with a satisfactory outcome. Pathological examination revealed severe deterioration of the affected hip with unique cartilage degeneration associated with active inflammation in the synovium and bone marrow. These features were also evaluated by magnetic resonance imaging in one case which presented with rapidly destructive changes in the hip. Spinal involvement may contribute to progressive destructive hip arthropathy in ochronosis.
 
Key words
Key words Alkaptonuria (AKU) ・ Ochronosis ・ Rapidly destructive hip joint ・ Spondylarthropathy
 
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