Eosinophilic fasciitis (Shulman disease): new insights into the therapeutic management from a series of 34 patients

Rheumatology (Oxford). 2012 Mar;51(3):557-61. doi: 10.1093/rheumatology/ker366. Epub 2011 Nov 25.

Abstract

Objective: To analyse therapeutic management of eosinophilic fasciitis (EF).

Methods: We reviewed 34 adult patients with biopsy-proven EF. Analyses focused on the therapeutic management, including treatment modalities, responses and associated or predictive factors.

Results: Thirty-four patients were included with a diagnosis age of 53 (15) years. They were featured by cutaneous manifestations (88%) including morphoea (41%), myalgia (86%) and hypereosinophilia (85%). Thirty-two patients (94%) were eligible for treatment evaluation and all received CSs as a first-line therapy. Fifteen patients (47%) received methylprednisolone pulses (MPPs) at treatment initiation and 14 patients (44%) received an immunosuppressive drug (ISD), usually MTX (86%), as a second-line therapy. Complete remission was achieved for 69% of patients, remission with disability 19% and failure 12%. A poor outcome was associated with a diagnosis time delay of >6 months [odds ratio (OR) = 14.7] and the lack of MPPs (OR = 12.9).

Conclusion: Our study reports new insights into the therapeutic management of EF: (i) CS treatment remains the standard therapy for EF, taken alone or in association with an ISD; (ii) MPPs at initiation of treatment are associated with a better outcome and a lower need of ISD use; (iii) an ISD, usually MTX, might be useful as a second-line therapy, mainly in patients with morphoea-like lesions. Naturally, these practical conclusions should be confirmed by a prospective and multicentre study.

MeSH terms

  • Adult
  • Aged
  • Antirheumatic Agents / administration & dosage*
  • Azathioprine / administration & dosage
  • Colchicine / administration & dosage
  • Drug Therapy, Combination
  • Eosinophilia
  • Female
  • Glucocorticoids / administration & dosage*
  • Humans
  • Hydroxychloroquine / administration & dosage
  • Immunosuppressive Agents / administration & dosage*
  • Male
  • Methotrexate / administration & dosage
  • Methylprednisolone / administration & dosage
  • Middle Aged
  • Prednisone / administration & dosage
  • Prognosis
  • Remission Induction
  • Retrospective Studies
  • Scleroderma, Localized / drug therapy
  • Scleroderma, Localized / etiology
  • Synovitis / complications
  • Synovitis / drug therapy*
  • Treatment Outcome

Substances

  • Antirheumatic Agents
  • Glucocorticoids
  • Immunosuppressive Agents
  • Hydroxychloroquine
  • Azathioprine
  • Colchicine
  • Prednisone
  • Methylprednisolone
  • Methotrexate

Supplementary concepts

  • Eosinophilic synovitis