Abstract |
Macrophage activation syndrome (MAS) is a rare and serious complication of adult-onset Still's disease. We describe a case in a 49-year-old woman with Still's disease refractory to glucocorticoids, methotrexate, and infliximab. Anakinra provided satisfactory disease control for 1 year, after which escape phenomenon occurred. After four tocilizumab injections, cutaneous melanoma developed. The persistent systemic manifestations prompted treatment with two canakinumab injections. Ten days later, she had a spiking fever, dyspnea, low back pain, abdominal pain, odynophagia, and hepatomegaly. Laboratory tests showed liver cytolysis (180 IU/L; N: 10-35), acute renal failure ( creatinine, 407 μmol/L; N:50-100), thrombocytopenia (60 G/L; N: 150-400), leukocytosis (12,200/mm(3); N: 4000-10,000), hypertriglyceridemia (5070 mmol/L; N: 0.4-1.6), lactate dehydrogenase elevation (4824 IU/L; N: 135-250), and hyperferritinemia (97 761 μg/L; N:15-150). Examination of a bone marrow biopsy showed phagocytosis. Tests were negative for viruses and other infectious agents. Glucocorticoid therapy (1.5 mg/Kg/d) and intravenous polyvalent immunoglobulins (0.5 g/Kg/d) were given. Her condition improved despite the many factors of adverse prognostic significance ( thrombocytopenia, absence of lymphadenopathy, and glucocorticoid therapy at diagnosis). This is the first reported case of MAS after canakinumab therapy in a patient with adult-onset Still's disease.
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Authors | Christopher Banse, Olivier Vittecoq, Ygal Benhamou, Maud Gauthier-Prieur, Thierry Lequerré, Hervé Lévesque |
Journal | Joint bone spine
(Joint Bone Spine)
Vol. 80
Issue 6
Pg. 653-5
(Dec 2013)
ISSN: 1778-7254 [Electronic] France |
PMID | 23751410
(Publication Type: Case Reports, Journal Article)
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Copyright | Copyright © 2013 Société française de rhumatologie. Published by Elsevier SAS. All rights reserved. |
Chemical References |
- Antibodies, Monoclonal
- Antibodies, Monoclonal, Humanized
- Antirheumatic Agents
- Interleukin-1beta
- canakinumab
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Topics |
- Antibodies, Monoclonal
(adverse effects)
- Antibodies, Monoclonal, Humanized
- Antirheumatic Agents
(adverse effects)
- Female
- Humans
- Interleukin-1beta
(antagonists & inhibitors)
- Macrophage Activation Syndrome
(etiology)
- Middle Aged
- Still's Disease, Adult-Onset
(complications, drug therapy)
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