Case report
Parkinsonism–hyperpyrexia syndrome: The role of electroconvulsive therapy

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Abstract

Herein, we present a case of a parkinsonism–hyperpyrexia syndrome (PHS) in a 58-year-old man with a 10-year history of Parkinson’s disease. The patient presented with a 2-week history of fever and increasing confusion, in the context of a number of changes to his medication regimen. On presentation, he was noted to be febrile with autonomic instability, diaphoresis and marked rigidity. He was disoriented and responding to visual hallucinations. Investigations revealed an elevated creatine kinase and a provisional diagnosis of PHS was made. After the patient failed to respond during a 2-week period to supportive measures, electroconvulsive therapy (ECT) treatment was commenced. A good response to eight bilateral ECT treatments was achieved, with resolution of his confusional state and associated psychotic phenomena. We discuss the nosological and management issues associated with this case and discuss the role of ECT as a treatment modality in this condition.

Introduction

Neuroleptic malignant syndrome (NMS) is serious and potentially fatal complication of neuroleptic therapy. Neuroleptic malignant syndrome is characterized by extrapyramidal symptoms, altered consciousness, hyperthermia, autonomic dysfunction and an elevated creatine kinase (CK) level occurring in the context of a neuroleptic medication.1

Patients with clinically advanced Parkinson’s disease (PD) have been shown to be at high risk of NMS, especially after the withdrawal of anti-parkinsonian medication.2 A neuroleptic malignant-like syndrome (NMLS) has also been shown to occur in the absence of any change to the anti-parkinsonian medication regimen.3 The increased risk in parkinsonian disorders is likely to be secondary to central dopaminergic suppression.4 In recognition of the fact that NMS can be triggered by factors other than exposure to neuroleptic medication, there has been a nosological shift in the research literature towards the term parkinsonism–hyperpyrexia syndrome (PHS);[5], [6] however, it has also been referred to as NMLS or malignant syndrome (MS) in PD.7

Electroconvulsive therapy (ECT) is recognised to be a safe and effective treatment for a range of affective and catatonic disorders in patients with neurological disorders.8 Case reports have also indicated that ECT has therapeutic effects in patients with severe PD but without affective or psychotic disorders.9 Multiple case reports have demonstrated the effectiveness of ECT in the treatment of NMS.10 However, an extensive search of Medline, EMBASE, PubMed and CINAHL databases showed no case reports of ECT in the treatment of patients with PHS.

Section snippets

Case report

A 58-year-old man presented with a 2-week history of increasing rigidity and agitation, associated with a confusional state with psychotic features. He developed persecutory and nihilistic delusions during this 2-week period and was noted by his family to be responding to visual hallucinations.

There was a 10-year history of PD, which had been stable over the past 4 years with no recent medication adjustments. The patient also had a background history of mixed anxiety and depression that had

Discussion

This is the first published case report highlighting the effectiveness of ECT in the treatment of PHS. Thus far, the literature has focused on medical therapy for this potentially fatal condition, although the effectiveness of ECT in the treatment of NMS in patients without PD has been acknowledged in numerous case reports.10

The nosology surrounding the condition of PHS is confusing. The term ‘PHS’ has been used in the literature to refer to a clinical syndrome that is essentially identical to

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