• Internal medicine · Jun 2021

    Case Reports

    A Recurrent Case of Adult-onset Still's Disease with Concurrent Acalculous Cholecystitis and Macrophage Activation Syndrome/Hemophagocytic Lymphohistiocytosis Successfully Treated with Combination Immunosuppressive Therapy.

    • Yuri Arai, Yuichi Ishikawa, Kazuya Abe, Yuri Kato, Daijiro Abe, Michio Fujiwara, and Yasuhiko Kita.
    • Department of Rheumatology, Yokohama Rosai Hospital, Japan.
    • Intern. Med. 2021 Jun 15; 60 (12): 195519611955-1961.

    AbstractWe herein report the case of 21-year-old female diagnosed with adult-onset Still's disease (AOSD) three years earlier who presented with fever and right upper abdominal pain. She was diagnosed with acute acalculous cholecystitis (AAC) based on hepatic dysfunction, elevated C-reactive protein, and gallbladder wall thickening on abdominal ultrasound. Based on the presence of pancytopenia, hyperferritinemia, and hemophagocytosis by a bone marrow examination, she was diagnosed with macrophage activation syndrome (MAS)/hemophagocytic lymphohistiocytosis (HLH) which was refractory to glucocorticoid pulse therapy. The combination of intravenous cyclosporine A with glucocorticoids was able to successfully control the disease activity of AOSD-related AAC and MAS/HLH.

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