Juvenile localized scleroderma: A single-centre experience from India

J Eur Acad Dermatol Venereol. 2023 Mar;37(3):598-604. doi: 10.1111/jdv.18765. Epub 2022 Dec 3.

Abstract

Background: Juvenile localized scleroderma (JLS) or morphoea, a rare chronic autoimmune disease predominantly affects skin, subcutaneous tissue and occasionally the adjacent muscle, fascia and bone. We report the largest single-centre cohort of patients with JLS from India.

Methods: Patients who were diagnosed to have JLS were enrolled from the Paediatric Dermatology Clinic and the Paediatric Rheumatology Clinic of a tertiary care referral hospital in India. Collected data included details of the clinical profile, laboratory investigations and management.

Results: We analysed 84 patients with Juvenile localized scleroderma. Median age of disease onset was 5 years, and median age at diagnosis was 8 years. Commonest subtype was linear scleroderma (57 patients, 67.7%) followed by plaque morphoea and generalized morphoea. Fourteen patients (16.6%) were noted to have extracutaneous manifestations (ECMs). These included arthritis in eight (33.3%), brain parenchymal abnormalities in four (4.7%) and pulmonary involvement in two (8.3%) patients. Antinuclear antibody (ANA) was positive in eight/25 patients (32%; diffuse and speckled pattern in four patients each). One amongst these also had elevated anti-dsDNA titres. Positive ANA was found to have no association with ECMs (p 1.000). Patients were treated using methotrexate (61 patients; 72.6%), dexamethasone oral mini-pulse (OMP; 35 patients; 41.6%), calcipotriol (39 patients; 46.4%), topical corticosteroids (32 patients; 38%) and topical tacrolimus (three patients; 3.7%). Using linear regression analysis, administration of dexamethasone OMP and calcipotriol was found to be a predictor of good treatment response (p 0.034 and 0.019, respectively).

Conclusion: Early use of systemic corticosteroids along with methotrexate may be more beneficial than methotrexate therapy alone.

MeSH terms

  • Child
  • Child, Preschool
  • Dexamethasone / therapeutic use
  • Glucocorticoids / therapeutic use
  • Humans
  • India
  • Methotrexate* / therapeutic use
  • Rare Diseases / complications
  • Rare Diseases / drug therapy
  • Scleroderma, Localized* / complications

Substances

  • Methotrexate
  • Glucocorticoids
  • Dexamethasone