First report of a patient meeting criteria for both multisystem inflammatory syndrome in children and adult onset Still’s disease

BMC Rheumatology

Case Report

Maya Alexandri¹  Lynne W. Coule²  Julisa Patel³  Eli Paul⁴ 

[1] Emergency Medicine, Medical College of Georgia, Augusta University Medical Center, 1120 15th Street, 30912, Augusta, GA, USA;Pediatric Emergency Medicine, Medical College of Georgia, Children’s Hospital of Georgia, 1446 Harper Street, 30912, Augusta, GA, USA;Pediatric Rheumatology, Medical College of Georgia, Children’s Hospital of Georgia, 1446 Harper Street, 30912, Augusta, GA, USA;University of Colorado at Boulder, 80309, Boulder, CO, USA;

关键词: Multisystem inflammatory syndrome in children;    MIS-C;    Novel coronavirus disease 2019;    COVID-19;    Systemic juvenile idiopathic arthritis;    SJIA;    Adult onset Still’s disease;    AOSD;    Still’s disease;    Macrophage activation syndrome;    MAS;    Case report;   

DOI  :  10.1186/s41927-022-00320-9

 received in 2022-08-05, accepted in 2022-11-05,  发布年份 2022

来源: Springer

【 摘 要 】

BackgroundCOVID-19 is associated with a postinfectious hyperinflammatory disorder, multisystem inflammatory syndrome in children (MIS-C), that shares characteristics with still’s disease, known as systemic juvenile idiopathic arthritis (SJIA) in children younger than 16, and adult onset Still’s disease (AOSD) in children 16 and older. Both MIS-C and SJIA/AOSD can be complicated by macrophage activation syndrome (MAS), a potentially fatal condition of cytokine storm.Case presentationWe present a 16 year-old male who developed quotidian fever, headache, conjunctival injection, sore throat, nausea and vomiting, diarrhea, rash, and symmetrical polyarticular arthralgia/arthritis 4 weeks after exposure to SARS-CoV-2 and 2 weeks after his first vaccination against COVID-19. Our patient’s laboratory results were significant for elevated inflammatory markers and acute phase reactants. He met criteria for diagnosis with both MIS-C and AOSD. After receiving first-line treatment for both diseases, IVIG and methylprednisolone, our patient improved.ConclusionMAS is a life-threatening rheumatological emergency, and physicians must be able to identify diseases, like MIS-C and AOSD, that may be complicated by MAS. Our patient’s distinguishing feature on presentation was symmetrical polyarticular arthralgia/arthritis, which has not been associated with MIS-C. Simultaneously, AOSD—which is associated with polyarticular arthralgia/arthritis—is only now being recognized as a possible post-infectious entity in the aftermath of COVID-19 infection. In patients like our own, who meet criteria for both MIS-C and AOSD, administering first line treatment for both diseases may be best practice.

【 授权许可】

CC BY   
© This is a U.S. Government work and not under copyright protection in the US; foreign copyright protection may apply 2022