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

Abstract

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 cryopyrinassociated 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.

Monoclonal Antibodies as Neurological Therapeutics

“…The most frequently affected organ was the thyroid, with up to 29% of patients developing thyroiditis [ 254 ], followed by idiopathic thrombopenic purpura (ITP) [ 255 ], and Goodpasture Syndrome with autoantibodies against the glomerular basement membrane [ 256 ]. Many other autoimmune conditions have been reported with alemtuzumab including but not limited to immune-mediated neutropenia and autoimmune hemolytic anemia [ 257 ], diabetes mellitus type 1 [ 258 ], Still’s disease [ 259 ], myositis [ 260 ] and alopecia areata universalis [ 261 ]. Although most alemtuzumab-associated autoimmune conditions are autoantibody-mediated some others such as alemtuzumab-related vitiligo are T cell mediated [ 262 ].…”