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DRESS syndrome with cerebral vasculitis provoked by piperacillin/tazobactam: a case report

  • Jildau W. Wijtsma*
  • , Jonathan M. Coutinho
  • , Ernesto A. C. Beenakker
  • *Corresponding author for this work
  • Frisius MC
  • Amsterdam UMC - University of Amsterdam

Research output: Contribution to journalArticleAcademicpeer-review

Abstract

Background: Drug Reaction with Eosinophilia and Systemic Symptoms (DRESS) syndrome is a rare, potentially life-threatening hypersensitivity reaction to medications. While commonly associated with hepatic, renal, and pulmonary involvement, central nervous system complications such as cerebral vasculitis are exceedingly rare and not well characterized. Case presentation: We present the case of a 69-year-old woman with a history of hypertension who developed DRESS syndrome following a 2.5-week course of piperacillin/tazobactam for an infected malleolar wound. She presented with fever, generalized malaise, nausea, anorexia, and a diffuse maculopapular rash. Laboratory findings revealed marked eosinophilia, leukocytosis, and elevated liver enzymes. A skin biopsy was consistent with toxicodermia. Based on clinical and laboratory criteria, a diagnosis of probable DRESS syndrome was made (RegiSCAR score: 8). High-dose intravenous corticosteroids were initiated. During hospitalization, the patient developed new-onset neurological deficits, including hemiplegia and bilateral paresis. Brain MRI revealed multifocal ischemic lesions with diffusion restriction in both cerebral and cerebellar hemispheres. Extensive diagnostic work-up, including echocardiography, vascular imaging, lumbar puncture, PET-CT, and autoimmune panels, excluded infectious, embolic, and systemic vasculitis etiologies. In the context of DRESS, cerebral vasculitis was suspected. Corticosteroid therapy was escalated to intravenous methylprednisolone, followed by a slow taper. Subsequent MRI showed regression of the lesions, and the patient made significant neurological recovery. Conclusion: This case illustrates a rare but severe neurological complication of DRESS syndrome—cerebral vasculitis—triggered by piperacillin/tazobactam. It highlights the importance of early recognition of atypical organ involvement in DRESS and the need for comprehensive neurological evaluation when new deficits emerge. Timely escalation of immunosuppressive therapy may improve outcomes in such cases.
Original languageEnglish
JournalActa neurologica Belgica
Early online date2025
DOIs
Publication statusE-pub ahead of print - 2025

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