Case Report: Adult Still's Disease in an Alemtuzumab-Treated Multiple Sclerosis Patient

被引:1
作者
Kraemer, Julia [1 ]
Kroemer-Olbrisch, Tanja [2 ]
Lakomek, Heinz-Juergen [3 ]
Schellinger, Peter D. [4 ]
Foell, Dirk [5 ]
Meuth, Sven G. [1 ]
Straeten, Vera [4 ]
机构
[1] Univ Hosp Munster, Dept Neurol, Inst Translat Neurol, Munster, Germany
[2] Ruhr Univ Bochum, Dept Dermatol Venerol Allergol & Phlebol, Johannes Wesling Hosp Minden, Univ Hosp, Minden, Germany
[3] Ruhr Univ Bochum, Dept Rheumatol & Phys Med, Johannes Wesling Hosp Minden, Univ Hosp, Minden, Germany
[4] Ruhr Univ Bochum, Dept Neurol & Neurogeriatr, Johannes Wesling Hosp Minden, Univ Hosp, Minden, Germany
[5] Univ Hosp Munster, Dept Pediat Rheumatol & Immunol, Munster, Germany
关键词
multiple sclerosis; Adult onset still disease; secondary autoimmunity; alemtuzumab; rituximab; anakinra; case report; CLASSIFICATION; CRITERIA;
D O I
10.3389/fimmu.2020.02099
中图分类号
R392 [医学免疫学]; Q939.91 [免疫学];
学科分类号
100102 ;
摘要
Background Autoimmune adverse events are the most relevant risks of alemtuzumab therapy. We present a patient with relapsing-remitting multiple sclerosis, who developed adult-onset Still's disease (AOSD) following alemtuzumab treatment. Case Presentation The patient suffered from sore throat, swallowing difficulties, high spiking quotidian fever, generalized skin rash, arthritis, and myalgia 2 months after the second course of alemtuzumab. Laboratory tests revealed elevated acute-phase reactants, anemia, neutrophilic leukocytosis, and thrombocytosis. Serum calprotectin, interleukin-2, and interleukin-6 levels were strongly increased. Autoimmune, rheumatic, neoplastic, infectious, and granulomatous disorders were excluded. The NLRP1 and NLRP3 gene test, which was performed under the presumption of a cryopyrin-associated autoinflammatory syndrome, was negative. Based on the Yamaguchi and Fautrel criteria, and supported by the histological findings from a skin biopsy of the rash, the diagnosis of AOSD was established. Therapy with the anti-IL-1 agent (anakinra) led to a significant improvement of symptoms and blood parameters. However, anakinra had to be converted to rituximab due to generalized drug eruption. Following therapy with rituximab, the patient has fully recovered. Conclusion The current case highlights AOSD as another rare and potentially life-threatening secondary autoinflammatory/autoimmune event following alemtuzumab treatment.
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