Adult-onset Still's disease: analysis of a monocentric cohort of patients

Scand J Rheumatol. 2024 Jul;53(4):263-268. doi: 10.1080/03009742.2024.2328439. Epub 2024 May 2.

Abstract

Objective: Adult-onset Still's disease (AOSD) is a multigenic autoinflammatory disease with a severe systemic involvement. Because of the rarity of the disease, most published cohorts are multicentric. The aim of this report is to describe a monocentric cohort of AOSD patients, reporting clinical features and response to therapy in a long follow-up.

Method: Thirty-eight patients, attending the Clinical Immunology Unit and fulfilling Yamaguchi, Fautrel, or Daghor-Abbaci classification criteria for AOSD, were recruited for this study. In all patients, clinical and serological data were collected at diagnosis and every 6 months thereafter. The Pouchot score was calculated at every visit.

Results: Fever, arthromyalgia, and skin rash were the most frequent manifestations, followed by lymphadenopathy, sore throat, arthritis, splenomegaly, hepatic involvement, pleuropericarditis, and weight loss. As far as the disease course is concerned, 25% presented a monocyclic and 35% a polycyclic pattern, and 40% developed chronic articular involvement. Severe complications were observed at disease onset in 21% of the patients. All of the patients were treated with steroids; 74% also received conventional synthetic disease-modifying anti-rheumatic drugs (methotrexate in most cases) and 71% biological disease-modifying anti-rheumatic drugs (interleukin-1 inhibitors in most cases). Therapeutic switching for lack/loss of efficacy or adverse drug reactions was necessary in 66%.

Conclusion: The analysis of this cohort confirms that AOSD is a complex, severe, and heterogeneous disease. However, despite long-term treatment and comorbidities, therapies are effective and well tolerated. The therapeutic armamentarium now available allows long-lasting remission with low immunosuppression to be achieved in most patients.

MeSH terms

  • Adult
  • Aged
  • Antirheumatic Agents* / therapeutic use
  • Arthritis / drug therapy
  • Cohort Studies
  • Exanthema / etiology
  • Female
  • Fever / etiology
  • Follow-Up Studies
  • Humans
  • Male
  • Methotrexate* / therapeutic use
  • Middle Aged
  • Still's Disease, Adult-Onset* / diagnosis
  • Still's Disease, Adult-Onset* / drug therapy
  • Young Adult

Substances

  • Methotrexate
  • Antirheumatic Agents