Aim Multisystem inflammatory syndrome in children (MIS-C), is a severe complication of coronavirus disease 2019 (COVID-19). Although, neurological complications in MIS-C are reported less frequently, rapid diagnosis is crucial to prevent it’s related sequeles.

Material and Method Herein, we presented our evaluation of a patient presented with febrile status and hemiplegia diagnosed as MIS-C.

Results A 13 month-old girl was referred to our hospital with febrile seizure and right-sided weakness following fever, cough and diarrhea for 3 days. She was lethargic and clinical examination showed right hemiparesis with 3/5 muscle tone. Levetiracetam was used for first line treatment but recurrent focal seizures were evulated as status and midazolam infusions started. Laboratory tests revealed elevated CRP, sedimentation rate, ferritin levels and hyponatremia. In further imaging studies brain magnetic resonance imaging (MRI) showed an acute ischemia in frontal area and periventricular region. Anticogulant and ampiric antibiotic treatment also initiated rapidly and further studies revealed positive result for COVID-19 in polymerase chain reaction (PCR) nasal swab. Cardiac and immunological evaluation were normal. She was diagnosed as MIS-C and the current treatment was continued by adding intravenous methylprednisone 2 mg/kg/day and intravenous immunoglobulin 2 gr/kg. The patient was discharged in full recovery after 3 weeks of treatment. In the examinations for coagulopathy, heterozygous PAI mutation and MTHFR mutation were detected.

Conclusions A rapid and holistic approach to suddenly developing neurological symptoms in children is crucial to provide appropriate diagnosis and treatment. In this case, where we presented the neurological manifestations rarely seen in the MIS-C, it has been shown that physicians should be very careful in terms of rare complications.