Abstract
Introduction:Generalized pustular psoriasis(GPP)is a rare life-threatening subtype of psoriasis and continues to be a therapeutic challenge.The first line therapies include acitretin,cyclosporin A,and methotrexate which have long-term side effects for children.Adalimumab has a favorable safety profile with low rates of adverse events,and has been described for successful treatment of adults with GPP in a few reports.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.During 14-month follow-up,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.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.
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