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Mycophenolate mofetil for methotrexate-resistant juvenile localized scleroderma.

Giorgia Martini1, Laura Saggioro1, Roberta Culpo1

  • 1Paediatric Rheumatology Unit, Department of Woman and Child Health, University of Padova, Padova, Italy.

Rheumatology (Oxford, England)
|September 26, 2020
PubMed
Summary

Mycophenolate mofetil (MMF) is effective and safe for severe juvenile localized scleroderma, particularly when methotrexate (MTX) is ineffective. This study supports MMF as a valuable treatment option for refractory cases.

Keywords:
Mychophenolate mofetiljuvenilelocalized sclerodermamethotrexatetreatment

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Area of Science:

  • Pediatric Rheumatology
  • Dermatology
  • Immunosuppressive Therapy

Background:

  • Juvenile localized scleroderma (jLS) can be severe and refractory to standard treatments like methotrexate (MTX).
  • Alternative immunosuppressive agents are needed for patients with severe or MTX-intolerant/resistant jLS.

Purpose of the Study:

  • To evaluate the safety and efficacy of mycophenolate mofetil (MMF) in patients with severe or MTX-refractory juvenile localized scleroderma.
  • To compare outcomes of MMF treatment with MTX responders in a retrospective cohort.

Main Methods:

  • Retrospective longitudinal study of consecutive jLS patients undergoing systemic treatment.
  • Comparison of patients treated with MMF (due to MTX intolerance/refractoriness) versus MTX responders.
  • Assessment of disease activity using the Localized Scleroderma Cutaneous Assessment Tool and thermography; disease course evaluated by relapses and treatment changes; relapse-free survival analyzed using Kaplan-Meier analysis.

Main Results:

  • The MMF group (22 patients) and MTX group (47 patients) showed comparable demographics and follow-up duration.
  • MMF was initiated primarily due to MTX resistance (18 patients).
  • After a mean follow-up of 9.4 years, 90.9% of MMF-treated patients and 100% of MTX-treated patients achieved inactive disease. No significant difference in relapse-free survival was observed, though MMF suggested more persistent remission. MMF was well tolerated.

Conclusions:

  • Mycophenolate mofetil demonstrates significant efficacy and good tolerance in severe and/or MTX-refractory juvenile localized scleroderma.
  • MMF is a valuable therapeutic option for refractory cases of jLS.
  • Further controlled studies are warranted to establish MMF's efficacy as a first-line treatment.