Migrating BMES in a patient with Behcet's disease

초록

Bone marrow edema syndrome (BMES) is a rare, unknown clinical condition that accompanies severe bone and joint pain. We present the first case of migrating BMES occurring in a patient with Behcet’s disease (BD). A 38-year-old woman with BD visited us with acute left ankle pain. She had two previous occurrences of osteomyelitis of the ankle. Review of her previous occurrences and magnetic resonance imaging (MRI) findings suggested migrating BMES. She had a low protein S acitivity—63 % when she was first diagnosed with BD— and this remained low at 62 % (normal, 71-103 %). Although the association between BD and BMES remains unclear and we cannot discount the possibility of coincidence, we estimate that the uncontrolled disease activity, blood flow obstruction induced by vasculitis, and protein S deficiency may have all played roles in the development of recurrent BMES in our patient. We think that recurrent BMES may progress to AVN in patients with BD because of ischemic changes and thrombus formation caused by vasculitis and protein S deficiency. Therefore, BMES should be closely monitored in a patient with BD, especially when accompanied by protein S deficiency. Early treatment with anti-coagulant or anti-platelet therapies should be considered in BMES patients with risk factors such as BD or other forms of vasculitis.