The First Pediatric Case of Acute Generalized Exanthematous Pustulosis Caused by Hydroxychloroquine

Abstract

Introduction: Acute generalized exanthematous pustulosis (AGEP) is a generalized, non-follicular sterile pustular rash, categorized as a severe cutaneous adverse reaction, which usually has a favorable prognosis. In a majority of cases (90%), AGEP is drug induced and different drugs are reported as cause of AGEP. Hydroxychloroquine (HCQ) is an antimalarial drug that is also used in some dermatologic and rheumatic diseases due to its immunosuppressive actions. Some cases of AGEP induced by HCQ are reported in literature but only in adults. Materials, Methods and Results: We describe the first case of AGEP caused by HCQ in a child affected by juvenile Sjögren syndrome. After withdrawal of HCQ and subsequent administration, the patient experienced the same cutaneous reaction. An allergy work-up was performed and patch test showed an ectopic flare of AGEP eruption. Conclusion: Our patient represents the first pediatric case of AGEP to HCQ, posing such a drug as a possible trigger also in children. Therefore, an accurate drug medical history is mandatory in order to rule out potential drug reactions when facing a sudden rash.

Hydroxychloroquine‐induced acute generalized exanthematous pustulosis: a series of seven patients and review of the literature

“…For example, the delay between drug exposure and symptom onset varied from 1 day to 3 weeks in diltiazem‐induced AGEP 10 , and from 1 day to 44 days in terbinafine‐induced AGEP 11 . While, in previously reported HCQ‐induced AGEP cases, the duration of exposure to HCQ prior to symptom onset ranged from 2 12 to 30 days 13,14 . In our patients, except for the lichen planus case who developed AGEP symptoms 122 days after onset of treatment, most cases of our series developed symptoms between 15 and 47 days, which is similar to the literature.…”

Hydroxychloroquine-induced Unusual Generalized Pustular Cutaneous Reaction As a New Clinical Entity: A Case Series

“…Besides the well-known pustular reaction to HCQ in adults and even in children [44], it may be necessary to define a hydroxychloroquine-induced pustular reactionwhich is not completely consistent with AGEP or PP -as a new entity in dermatology. It is a long way to do new research with these hypotheses.…”

Acute generalized exanthematous pustulosis with airway mucosa involvement: A case report

“…The etiology of AGEP is not fully understood. More than 90% of AGEP cases are caused by drugs, such as aminopenicillins, pristinamycin, hydroxychloroquine, quinolones, sulfonamides, terbinafine, ketoconazole, diltiazem, and fluconazole[ 4 - 8 ]. Additional causes of AGEP, such as contact with mercury[ 9 ], have also been described.…”