The dyskinesia-hyperpyrexia syndrome is a rare complication of Parkinson disease. Here, we present an exceptional case affected by this syndrome. Its singularity is 2-fold. On one hand, the patient presented periodic episodes of dyskinesia-hyperpyrexia during 3 consecutive summers. On the other, after unsuccessfully trying several therapies, she had an excellent response to an infusion of enteral L-dopa. Both features allow us to hypothesize that this complication is central in origin and propose a new potential mean of treating future refractory cases.