Bronchiolitis obliterans as a long-term sequela of Stevens-Johnson syndrome and toxic epidermal necrolysis in children

Clin Exp Dermatol. 2019 Dec;44(8):897-902. doi: 10.1111/ced.13969. Epub 2019 Jul 17.

Abstract

Toxic epidermal necrolysis (TEN) and Stevens-Johnson syndrome (SJS) are characterized by widespread skin and mucosal blistering and necrosis. The triggers and long-term sequelae in children may differ from those reported for adults. Bronchiolitis obliterans (BO) is an uncommon complication, with only 15 previously reported cases, but can lead to significant long-term morbidity, requiring lung transplantation in some cases. We report three children with nondrug-related SJS (n = 1) and TEN (n = 2) who developed BO. Two were treated with intravenous immunoglobulin therapy (2-2.4 g/kg) and all three survived. We highlight salient learning points from our cases and potential pitfalls in diagnosis of BO, including delayed onset, and we also review the literature.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Adolescent
  • Bronchiolitis Obliterans / diagnosis
  • Bronchiolitis Obliterans / diagnostic imaging
  • Bronchiolitis Obliterans / etiology*
  • Bronchiolitis Obliterans / therapy
  • Child
  • Child, Preschool
  • Female
  • Humans
  • Immunoglobulins, Intravenous / therapeutic use
  • Lung / diagnostic imaging
  • Male
  • Respiratory Function Tests
  • Stevens-Johnson Syndrome / complications*
  • Tomography, X-Ray Computed

Substances

  • Immunoglobulins, Intravenous