Colchicine-resistant Sacroiliitis in a Japanese Patient with Familial Mediterranean Fever.

The articular involvement in patients with familial Mediterranean fever (FMF) represents a clinical characteristic of acute monoarthritis with pain and hydrarthrosis, which always resolve spontaneously. Colchicine prevents painful arthritis attacks in most FMF cases. Spondyloarthritis (SpA) is rarely associated with Japanese patients with FMF. Here, we report a Japanese male patient with FMF-related axial joint involvement. A 43-year-old male Japanese patient resented with recurrent febrile episodes with hip joint and back pain was referred to our hospital. He carried heterozygous variants in exon 2 (L110P/E148Q) of the MEFV gene. FMF was suspected and oral administration of colchicine (1 mg/day) was initiated. Colchicine treatment improved his febrile attack with hip joint pain. He was diagnosed as having FMF based on the Tel-Hashomer's diagnostic criteria for FMF, since he fulfilled 1 major criteria (repeated febrile attack accompanied by hip joint pain) and 1 minor criteria (improvement with colchicine treatment). Although HLA-B27 allele was not detected, sacroiliitis-related symptoms progressed despite the ongoing colchicine treatment. Salazosulfapyridine and methotrexate were administrated in addition to colchicine; however, these treatments were not effective. Canakinumab treatment successfully resolved this unique aspect of sacroiliitis, and he was finally diagnosed with FMF-associated axial joint involvement.