Atypical presentations of drug-induced hypersensitivity syndrome caused by carbamazepine with concurrent use of corticosteroid: Report of a case with a brief literature review

ABSTRACT

Department of Rehabilitation Medicine, Seoul National University Hospital, Seoul, KoreaCarbamazepine (CBZ) is the common antiepileptic drug prescribed for the management of neuropathic pain in patients with neurologic injury. CBZ can rarely be the causative agent to result in drug-induced hypersensitivity syndrome (DIHS), which is a life-threatening drug reaction. Delayed diagnosis of DIHS may occur in complicated clinical settings. We have reported a case of a 12-year-old girl whose initial manifestations of CBZ-induced DIHS were atypical due to concurrent administration of corticosteroids. After open biopsy of brainstem glioma, she was administered corticosteroids and CBZ to mitigate postoperative complications, including mild weakness of ankle dorsiflexion and otalgia. Not only fever but also elevation of liver enzymes and inflammatory markers were presented 1 month after CBZ initiation, without any skin rash or leukocytosis. A maculopapular rash on the whole body and leukocytosis developed afterward; consequently, the diagnostic criteria for DIHS were fulfilled. After discontinuation of CBZ, her fever and increased liver enzymes subsided and the rash slowly improved. This case indicates that typical signs, such as skin rash or leukocytosis, can be initially masked or delayed in CBZ-induced DIHS if it overlaps with corticosteroid administration. The atypical manifestations of DIHS should be considered if CBZ is concurrently administered with corticosteroids.