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Jean-Jacques Dubost, Julie Ledoux-Eberst, Julie Vaure-Chiffre, Denis Pezet, Martin Soubrier, Imatinib-induced bone oedema mimicking Type 1 complex regional pain syndrome, Rheumatology, Volume 59, Issue 6, June 2020, Pages 1444–1446, https://doi.org/10.1093/rheumatology/kez503
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DearSir, Imatinib (Glivec) is a protein kinase inhibitor primarily indicated for managing chronic myeloid leukaemia and stromal tumours of the gastrointestinal tract. We report herein a case involving juxta-articular bone oedema, suggesting the possibility of imatinib-induced algodystrophy.
A 63-year-old woman was referred to us for algodystrophy of her left ankle and knee. Approximately 15 months earlier, the patient had undergone surgical resection of the duodenojejunal angle for a gastrointestinal stromal tumor. Following this operation, the patient was given adjuvant therapy consisting of 400 mg/day imatinib. Three months later, following a snowshoeing expedition, the patient suddenly experienced left ankle pain and swelling, which she attributed to a sprained ankle. After temporary improvement, the pain and swelling recurred. X-rays were normal. MRI revealed large bone marrow oedema of the talus, the inferior end of the tibia, and fibula, as well as a tibiotalar and subtalar effusion, along with thickening of periarticular soft tissues (Fig. 1). Three months before the consultation, the patient had suddenly experienced pain and swelling of the left knee, without any known trigger. Her bone scintigraphy revealed intense radiotracer uptake involving the left patella, talus, the distal third of left tibia, and the middle third of right tibia (Fig. 1). While computed tomography of the knees proved normal, MRI revealed a bone marrow oedema of the patella and adjoining soft tissues, without joint effusion. The clinical examination revealed erythrocyanosis of the left leg and foot in dangling position, increased volume of the ankle and left leg, and enhanced local temperature. Joint mobilization was non-painful, with normal range of motion. The patella presented slight sensitivity to percussion, while the right tibia was without symptoms. X-rays revealed the presence of a mild periosteal reaction at the distal outer border of the left tibia. No demineralization was observed. Further laboratory work-up revealed no inflammatory syndrome, normal levels of calcium, phosphorus, alkaline phosphatase, vitamin D, and parathormone. As imatinib was suspected to be the culprit, the medication was discontinued, and the pain disappeared within 72 h. Following a 15-day medication-free interval, imatinib was re-initiated and the painful symptoms recurred, quite rapidly. It was therefore decided to definitively withdraw treatment. As a result, the pain disappeared once more, and at the time of writing, 1 year later, the patient was without any recurrences and had resumed walking, along with other activities, without any limitations.
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