Severe Generalized Bullous Fixed Drug Eruption Treated with Cyclosporine: A Case Report and Literature Review

Abstract

Generalized bullous fixed drug eruptions (GBFDEs) are rare in the paediatric population. We present the case of a 7-year-old girl with GBFDE believed to be secondary to oral ibuprofen, who experienced rapid resolution of lesions and cessation of blistering with a 3-week course of oral cyclosporine. To the best of our knowledge, this is the first report of a paediatric case of GBFDE treated with cyclosporine. In our report, we review published cases of GBFDE in children, and all adult cases managed with cyclosporine.

Bullous Erythema Multiforme Secondary to Trimethoprim-Sulfamethoxazole Use, Treated With Cyclosporine in a 91-Year-Old Male

“…Cyclosporine has been reported to be successful in treating a case of recurrent EM [ 5 ], a rare form of EM that, as its name suggests, is chronic and associated with HSV [ 1 ]. It has also been used in a case of severe bullous EM [ 14 ], and generalized bullous FDE [ 8 , 15 ]. Its use in generalized bullous FDE has been supported by studies that show a rapid cessation of blistering and healing of skin [ 15 ], while providing a steroid-sparing effect.…”

“…It has also been used in a case of severe bullous EM [ 14 ], and generalized bullous FDE [ 8 , 15 ]. Its use in generalized bullous FDE has been supported by studies that show a rapid cessation of blistering and healing of skin [ 15 ], while providing a steroid-sparing effect. There are no studies comparing cyclosporine to steroids for EM.…”

A Review of Fixed Drug Eruption with a Special Focus on Generalized Bullous Fixed Drug Eruption

“…Due to increasing reports of severe cases of GBFDE, including the aforementioned study describing a high mortality rate in this condition [43], there has been recent interest in the use of cyclosporine for treatment. Thus far, there have been six cases of GBFDE treated with cyclosporine described in the literature [63][64][65][66][67]. Five of the cases were in adults, and these patients were treated with five to 14 days of cyclosporine, typically at doses of three or five mg/kg daily, with resolution of erythema and cessation of further blistering [63][64][65][66].…”

“…Five of the cases were in adults, and these patients were treated with five to 14 days of cyclosporine, typically at doses of three or five mg/kg daily, with resolution of erythema and cessation of further blistering [63][64][65][66]. One pediatric case of GBFDE was treated with five mg/kg cyclosporine divided into two daily doses for one week followed by 2.5 mg/kg/day for another two weeks [67]. In this patient, improvement of erythema and cessation of further blistering were noted within 24 h of cyclosporine therapy [67].…”

“…One pediatric case of GBFDE was treated with five mg/kg cyclosporine divided into two daily doses for one week followed by 2.5 mg/kg/day for another two weeks [ 67 ]. In this patient, improvement of erythema and cessation of further blistering were noted within 24 h of cyclosporine therapy [ 67 ]. There are no clinical trials comparing the efficacy of supportive care alone versus treatments such as topical steroids, systemic steroids, or cyclosporine for GBFDE.…”