| Summary: | Abstract Background Anticonvulsant hypersensitivity syndrome is a rare adverse drug reaction associated with aromatic anticonvulsant drugs. This syndrome can range from mild cutaneous rash to drug reaction with eosinophilia and systemic symptoms that include fever, rash, lymphadenopathy, pancytopenia, and involvement of multiple internal organs. We aimed to report this case in the literature and make physicians aware of the uncommon symptoms of this syndrome when they prescribe antiepileptic medications in particular. Case presentation A 14-year-old Middle Eastern female patient from Iran with free past medical and allergic history was admitted to hospital because of fever, rash, lymphadenopathy, and pancytopenia after taking anticonvulsants due to new-onset seizure. High fever and cutaneous rash along with lymphadenopathy following administration of anticonvulsant medications that could not be explained by other causes alerted the physician to the possibility of this syndrome. Our investigation revealed no further diagnosis and 1 week after discontinuation of the drugs, her symptoms were resolved. Anticonvulsant hypersensitivity syndrome is a diagnosis of exclusion and immediate discontinuation of the suspicious drugs is necessary. Hence, early recognition can prevent permanent multiorgan damage. Conclusions Chlorpheniramine as a simple treatment was provided for this syndrome.
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