Summertime Dyskinesia-Hyperpyrexia Syndrome: The “Dual Heat” Hypothesis

Abstract

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.

Dyskinesia‐Hyperpyrexia Syndrome in Parkinson's Disease: A Heat Shock–Related Emergency?

“…Most of the subjects were treated with high doses of dopaminergic drugs (half of them with LCIG). A distinctive feature of DHS is the relationship with high ambient temperature, described in a patient with recurrent DHS over three summers and in our 3 cases. All our patients, suffering from DHS during summer, showed the same sequence of clinical‐pathological events: sustained hyperthermia and impaired thermoregulation, dehydration, dyskinesia, rhabdomyolysis, and alteration of mental state.…”

“…DHS is a poorly described and under‐reported acute complication in PD patients with an opposite clinical spectrum (dyskinesia), but under similar precipitating systemic factors (infectious diseases, trauma, and dehydration). In all reported cases, including ours (Table , n = 8 patients), DHS occurred in patients with long duration PD and was associated with a trigger event. Most of the subjects were treated with high doses of dopaminergic drugs (half of them with LCIG).…”

“…Nonphysiological dopaminergic stimulation can cause Dyskinesia‐Hyperpyrexia syndrome (DHS), a rare medical emergency associated with Parkinson's Disease (PD) . DHS is characterized by severe continuous dyskinesia, rhabdomyolysis, and hyperthermia that may progress to mental state alteration, renal and cardiac failure, and death.…”

Sindrom diskinezija-hiperpireksija – novo hitno stanje kod Parkinsonove bolesti: prikaz slučaja

“…Uz prethodno opisane slučajeve 4,6-8 možemo pridruži i naš, s obzirom na to da im je zajednička značajka dugogodišnje bolovanje od Parkinsonove boles s fl uktuacijom motoričkih simptoma i kon nuirano primanje dopaminergičkih lijekova. U jednom slučaju, prije nastanka simptoma, dodan je lijek ropinirol 7 , dok je kod troje pacijenata prethodio temperaturni klimatski maksimum 4,6,8 . Iako osnovni mehanizmi sindroma diskinezija-hiperpireksija još uvijek nisu dobro razjašnjeni, vjerojatan zajednički predisponirajući čimbenik bila je dugotrajna Parkinsonova bolest s fl uktuacijama motoričkih simptoma povezanih s levodopom, što upućuje na smanjenu pufersku sposobnost za dopaminergijsku nadomjesnu terapiju 9 .…”

“…Iako osnovni mehanizmi sindroma diskinezija-hiperpireksija još uvijek nisu dobro razjašnjeni, vjerojatan zajednički predisponirajući čimbenik bila je dugotrajna Parkinsonova bolest s fl uktuacijama motoričkih simptoma povezanih s levodopom, što upućuje na smanjenu pufersku sposobnost za dopaminergijsku nadomjesnu terapiju 9 . Također, u predisponirajuće čimbenike svrstaju se primjena D-3 agonista 7 i termostatska disregulacija 6 . U jednom slučaju pacijen ca je prije napada doživjela traumu.…”