Baseline characteristics and long-term outcomes of eosinophilic fasciitis in 89 patients seen at a single center over 20 years

Int J Rheum Dis. 2020 Feb;23(2):233-239. doi: 10.1111/1756-185X.13770. Epub 2019 Dec 6.

Abstract

Aim: Eosinophilic fasciitis (EF) is a rare, fibrosing disorder of skin and subcutaneous tissue. This study was undertaken to describe its clinical and laboratory features and identify prognostic factors associated with outcome.

Methods: We conducted a retrospective review of all EF patients evaluated at our institution from 1 January1997 to 30 December 2016. Kaplan-Meier methods were used to determine treatment response rates over time. Potential associations between baseline characteristics and complete response were examined using Cox models adjusted for age and sex. Time-dependent covariates were used to examine treatment effects.

Results: We identified 89 EF patients, with a female-to-male ratio of 1:1. Clinical features included groove sign in 26 (29%), peau d'orange/dimpling in 22 (25%), inflammatory arthritis in 9 (10%) and muscle weakness in 9 (10%). Aldolase was elevated in 11/36 (31%). Complete response rate was 60% (95% confidence interval [CI]: 35-75) at 3 years. Diagnostic delay was inversely associated with treatment response (hazards ratio: 0.84 per 1 month increase; 95% CI: 0.73-0.98). No baseline characteristics correlated with treatment response, but a trend toward positive association of elevated aldolase, hypergammaglobulinemia and presence of hematologic disorders was noted. Methotrexate was the most commonly used immunosuppressant in 79%, hydroxychloroquine in 45%, mycophenolate mofetil in 18% and azathioprine in 8%. No single immunosuppressant agent was associated with a superior response during treatment.

Conclusions: EF is characterized by relatively high response rates. Consensus diagnostic criteria, standardized management algorithms, and large prospective multi-center cohorts are needed to develop an evidence-directed approach to this challenging condition.

Keywords: eosinophilia; eosinophilic fasciitis; sclerosing disorder; thickened skin.

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Biopsy
  • Child
  • Delayed Diagnosis*
  • Diagnosis, Differential
  • Eosinophilia / diagnosis*
  • Eosinophilia / drug therapy
  • Fasciitis / diagnosis*
  • Fasciitis / drug therapy
  • Female
  • Follow-Up Studies
  • Forecasting*
  • Humans
  • Immunosuppressive Agents / therapeutic use
  • Male
  • Methotrexate / therapeutic use*
  • Middle Aged
  • Retrospective Studies
  • Treatment Outcome
  • Young Adult

Substances

  • Immunosuppressive Agents
  • Methotrexate

Supplementary concepts

  • Eosinophilic Fasciitis