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Tocilizumab for uncontrollable systemic inflammatory response syndrome complicating adult-onset Still disease: Case report and review of literature.

Abstract Adult-onset Still disease (AOSD) is a rare systemic inflammatory disease of unknown etiology characterized by evanescent salmon-pink rash, fever spikes, arthralgia, and lymphadenopathy. AOSD usually has a good prognosis, but it can sometimes be fatal, especially when it is complicated by systemic inflammatory response syndrome (SIRS) and multiple organ failure.
PMID
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Authors

Mayor MeshTerms
Keywords
Journal Title medicine
Publication Year Start




PMID- 28723802
OWN - NLM
STAT- MEDLINE
DA  - 20170720
DCOM- 20170808
LR  - 20170808
IS  - 1536-5964 (Electronic)
IS  - 0025-7974 (Linking)
VI  - 96
IP  - 29
DP  - 2017 Jul
TI  - Tocilizumab for uncontrollable systemic inflammatory response syndrome
      complicating adult-onset Still disease: Case report and review of literature.
PG  - e7596
LID - 10.1097/MD.0000000000007596 [doi]
AB  - RATIONALE: Adult-onset Still disease (AOSD) is a rare systemic inflammatory
      disease of unknown etiology characterized by evanescent salmon-pink rash, fever
      spikes, arthralgia, and lymphadenopathy. AOSD usually has a good prognosis, but
      it can sometimes be fatal, especially when it is complicated by systemic
      inflammatory response syndrome (SIRS) and multiple organ failure. PATIENT
      CONCERNS: A previously healthy 26-year-old woman was referred to our hospital for
      persistent high fever and mild systemic edema. Five days later, the patient
      presented with dyspnea, hypotension, and anuria. Anasarca developed with massive 
      pleural effusion, ascites, and systemic edema, resulting in an increase of 47 kg 
      in body weight. DIAGNOSES: The patient was diagnosed as AOSD after infection,
      malignancy, hematologic disorders, and other autoimmune diseases were excluded.
      INTERVENTIONS: We administered tocilizumab, an IL-6 receptor inhibitor,
      intravenously in addition to cyclosporine, prednisolone, plasma exchange, and
      continuous hemodiafiltration. OUTCOMES: The patient's systemic condition
      improved. After stabilization by all medications, the patient was managed and
      responded to tocilizumab alone. To the best of our knowledge, this was the first 
      case of severe SIRS complicating AOSD that was successfully treated with an anti-
      IL-6 receptor antibody. LESSONS: SIRS should not be overlooked in a patient with 
      steroid-resistant AOSD and edema. Inhibitors of the IL-6 receptor can be used
      safely and effectively to control AOSD complicated with severe SIRS.
FAU - Masui-Ito, Asami
AU  - Masui-Ito A
AD  - aEmergency and Critical Care Center, Mie University Hospital bDepartment of
      Cardiology and Nephrology, Mie University Graduate School of Medicine, Tsu,
      Japan.
FAU - Okamoto, Ryuji
AU  - Okamoto R
FAU - Ikejiri, Kaoru
AU  - Ikejiri K
FAU - Fujimoto, Mika
AU  - Fujimoto M
FAU - Tanimura, Muneyoshi
AU  - Tanimura M
FAU - Nakamori, Shiro
AU  - Nakamori S
FAU - Murata, Tomohiro
AU  - Murata T
FAU - Ishikawa, Eiji
AU  - Ishikawa E
FAU - Yamada, Norikazu
AU  - Yamada N
FAU - Imai, Hiroshi
AU  - Imai H
FAU - Ito, Masaaki
AU  - Ito M
LA  - eng
PT  - Case Reports
PT  - Journal Article
PT  - Review
PL  - United States
TA  - Medicine (Baltimore)
JT  - Medicine
JID - 2985248R
RN  - 0 (Antibodies, Monoclonal, Humanized)
RN  - 0 (Immunologic Factors)
RN  - I031V2H011 (tocilizumab)
SB  - AIM
SB  - IM
MH  - Adult
MH  - Antibodies, Monoclonal, Humanized/*administration & dosage
MH  - Diagnosis, Differential
MH  - Female
MH  - Humans
MH  - Immunologic Factors/*administration & dosage
MH  - Still's Disease, Adult-Onset/*complications/diagnostic imaging/pathology
MH  - Systemic Inflammatory Response Syndrome/*complications/diagnosis/*drug
      therapy/pathology
PMC - PMC5521942
EDAT- 2017/07/21 06:00
MHDA- 2017/08/09 06:00
CRDT- 2017/07/21 06:00
AID - 10.1097/MD.0000000000007596 [doi]
AID - 00005792-201707210-00067 [pii]
PST - ppublish
SO  - Medicine (Baltimore). 2017 Jul;96(29):e7596. doi: 10.1097/MD.0000000000007596.