Objective: To report a rare case of DHS that was initially misdiagnosed as generalized seizures.

Background: Parkinson’s disease (PD) is a progressive neurodegenerative disorder with bradykinesia, rigidity, gait disturbance, and resting tremor. Although hyperpyrexia is rare in PD, it may occur as serotonin syndrome, neuroleptic malignant syndrome, parkinsonism hyperpyrexia syndrome (PHS), or dyskinesia–hyperpyrexia syndrome (DHS). Among these, DHS is characterized by severe generalized dyskinesia with high fever and altered consciousness and it is extremely rare in south Korea.

Method: A 74-year-old man with a 9-year history of PD (Hoehn and Yahr stage 3) was treated with levodopa (187.5 mg × 5/day), pramipexole (0.5 mg × 3/day), and rasagiline (1 mg/day). He had mild peak-dose dyskinesia for 3 months without wearing-off. He presented with stupor and 39.5 °C fever. Vital signs showed tachycardia (117/min) and tachypnea (25–30/min) with severe generalized myoclonic dyskinesia and neck extension. Generalized seizure was suspected, and intravenous lorazepam (4 mg) was given, partially improving jerks. Brain MRI and EEG were normal. Laboratory findings showed elevated CK (1265 IU/L) but normal blood, hepatic, and renal results. He admitted to self-increasing his levodopa dose up to a double dose before admission.

DHS was diagnosed according to the criteria proposed by Wang et al., based on generalized dyskinesia, hyperpyrexia, elevated CK level, altered consciousness, and recent levodopa overdose. Supportive treatment with intravenous hydration and acetaminophen led to normalization of consciousness and temperature (37.5 °C) within 6 hours, and dyskinesia subsided completely after 8 hours. He was discharged with education on proper medication use.

Results: DHS is a rare but serious complication of advanced PD, often related to high-dose levodopa and dopaminergic fluctuation. Proposed diagnostic features include generalized dyskinesia, hyperpyrexia, elevated CK, altered mental status. It is presumed to result from impaired striatal dopamine buffering and abrupt dopaminergic changes in the nigrostriatal system. Management involves supportive care, hydration, fever control, and adjustment of dopaminergic drugs.

Conclusion: DHS can mimic generalized seizure in PD patients presenting with fever and abnormal movements. Early recognition, supportive therapy, and dopaminergic stabilization ensure rapid recovery and favorable outcome.

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MDS Abstracts - https://www.mdsabstracts.org/abstract/dyskinesia-hyperpyrexia-syndrome-mimicking-a-generalized-seizure-in-parkinsons-disease/