Abstract
BackgroundAcute generalized exanthematous pustulosis (AGEP) and toxic epidermal necrolysis (TEN) are severe cutaneous adverse reactions. These rare conditions differ in clinical presentation, pathological features, treatment and prognosis, but overlap has been described implying a challenging clinical management.Case presentationWe describe a case of overlap between TEN and AGEP probably secondary to beta-lactams in a 77-year-old patient treated for a complicated cholangitis. We review the diagnosis and the management of these two conditions. The diagnosis of TEN was suggested by the initial clinical presentation with severe hemodynamic instability, skin detachment, positive Nikolsky sign and mucosal involvement. However, the skin biopsy as well as the rapid improvement of the skin lesions were discriminative for AGEP. This indicated an overlap presentation. Unfortunately, the patient refused allergy investigations in order to find the culprit drug. Medical photographs, proper physical examination and histopathological results are integrated.ConclusionDespite clinical features indicating a diagnosis of TEN, histopathology was conclusive for AGEP thus indicating a possible clinical-pathological overlap between the two conditions, a scarcely described situation in the medical literature. To our knowledge, this is one of the few cases that portrays a TEN–AGEP overlap probably secondary to Piperacillin Tazobactam. Understanding the immunological implications of these conditions can help us better distinguish and manage these severe reactions.
Highlights
Acute generalized exanthematous pustulosis (AGEP) and toxic epidermal necrolysis (TEN) are severe cutaneous adverse reactions
Despite clinical features indicating a diagnosis of TEN, histopathology was conclusive for acute generalized exanthematous pustulosis (AGEP) indicating a possible clinical-pathological overlap between the two conditions, a scarcely described situation in the medical literature
We describe the case of a 77-yearold female who developed an unusual AGEP–TEN overlap probably secondary to Piperacillin Tazobactam
Summary
The initial diagnosis of these skin conditions is based on the clinical presentation. Further pathological investigations and the overall clinical evolution oriented us towards an AGEP diagnosis. We must ask ourselves if these conditions should be described as an overlap presentation or as the manifestation of a severe, aggressive AGEP because of the convincing biopsy?. Further case descriptions and systematic research are need to help us elucidate if these atypical cases are an overlap of the two conditions, a two-phase clinical disease entity of a manifestation of severe AGEP mimicking TEN. We encourage clinicians to describe these cases and we believe that an international register of these conditions as well as developing new investigational tools such as blister fluid analysis can help us evaluate this co-presentation of two different immunological processes
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