Department of Rheumatology, Yokohama Rosai Hospital, Japan.
Department of Gastroenterology, Yokohama Rosai Hospital, Japan.
Intern Med. 2021 Jun 15;60(12):1955-1961. doi: 10.2169/internalmedicine.5781-20. Epub 2021 Feb 1.
We 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.
我们在此报告一例 21 岁女性,其三年前被诊断为成人Still 病(AOSD),此次因发热和右上腹痛就诊。根据肝功能异常、C 反应蛋白升高和腹部超声提示胆囊壁增厚,诊断为急性非结石性胆囊炎(AAC)。骨髓检查发现全血细胞减少、铁蛋白升高和噬血细胞现象,诊断为巨噬细胞活化综合征(MAS)/噬血细胞性淋巴组织细胞增生症(HLH),对糖皮质激素脉冲治疗无效。环孢素 A 静脉输注联合糖皮质激素成功控制了与 AOSD 相关的 AAC 和 MAS/HLH 的疾病活动。