Orofacial granulomatosis in a 12-year-old girl successfully treated with intravenous pulse corticosteroid therapy and chloroquine

Pediatr Dermatol. 2017 Nov;34(6):e324-e327. doi: 10.1111/pde.13279. Epub 2017 Sep 21.

Abstract

Orofacial granulomatosis, a rare disease in childhood, is characterized by orofacial swelling in the absence of systemic disease. We report the case of a 12-year-old girl with asymptomatic erythematous infiltration of her upper lip, cheeks, and chin that had persisted for more than 2 years; biopsy confirmed granuloma formation. Because a large area was affected, intralesional corticosteroids were inappropriate and six cycles of 3-day intravenous pulse corticosteroid therapy (dexamethasone 1.5mg/kg), repeated once after 4 weeks, was given. Our patient also received oral chloroquine and topical emollients. At the end of the sixth pulse cycle, the infiltration had completely resolved, leaving slight residual erythema.

Keywords: Melkersson-Rosenthal syndrome; chloroquine; orofacial granulomatosis; pulse corticosteroid therapy.

Publication types

  • Case Reports

MeSH terms

  • Antirheumatic Agents / therapeutic use*
  • Child
  • Chloroquine / therapeutic use*
  • Female
  • Glucocorticoids / administration & dosage*
  • Granulomatosis, Orofacial / drug therapy*
  • Granulomatosis, Orofacial / pathology
  • Humans
  • Pulse Therapy, Drug
  • Skin / pathology

Substances

  • Antirheumatic Agents
  • Glucocorticoids
  • Chloroquine