Successful Treatment of Generalized Pustular Psoriasis with Adalimumab in a Pediatric Patient: a case report

Abstract

Introduction: Generalized pustular psoriasis (GPP) is a rare life-threatening subtype of psoriasis and continues to be a therapeutic challenge. We herein describe a 3-year-old girl with a body weight of 14.2 kg in whom adalimumab was used to successfully treat GPP. Case presentation: A 3-year-old girl with a 10-day history of systemic symptoms of fever, malaise, and the onset of studded painful pustules overlying an erythematous base on the scalp, face, neck, trunk, and bilateral extremities, affecting a body surface area of 90%. Laboratory examination results were notable for leukocytosis and elevated inflammatory markers. Biopsy confirmed the diagnosis of GPP. After failure of conventional therapies including cyclosporine and methotrexate, the patient began off-label treatment with adalimumab and achieved complete lesion clearance. At the time of this writing, she remains in remission on maintenance adalimumab therapy. Discussion: No consensus on the standard treatment regimen for GPP has been reached. Unfortunately, all drugs that have been reported to be effective for GPP are off-label. These treatments include systemic immunomodulators such as cyclosporine and methotrexate, systemic retinoids such as acitretin and isotretinoin, and various biologics. The FDA has approved adalimumab, an anti-tumor necrosis factor-alpha (anti-TNF-alpha) monoclonal antibody, for the treatment of plaque psoriasis. Our findings lend weight to the scant evidence describing the effective use of adalimumab in the treatment of pediatric GPP. Conclusion: Off-label use of adalimumab may be an effective and safe therapeutic option for GPP.