and 1419-1422 1419 Journal of Neurology, Neurosurgery, Psychiatry 1994;57: J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.57.11.1419 on 1 November 1994. Downloaded from

SHORT REPORT

Epileptic seizures in patients with acute catatonic syndrome

Alberto Primavera, Alessandra Fonti, Paolo Novello, Giuseppe Roccatagliata, Leonardo Cocito

Abstract Kahlbauml first described catatonia as a syn- Acute catatonic syndrome is a condition drome consisting of catalepsy and waxy flexi- that can be caused by a variety of meta- bility, mutism, muscle rigidity, psychotic bolic, neurological, psychiatric, and toxic negativism, and autonomic dysfunctions. conditions, including neuroleptic malig- Although he considered catatonia as strongly nant syndrome. Although ictal catatonia associated with affective illness, he also as a manifestation of non-convulsive sta- emphasised the possible association with tus epilepticus has been described, refer- other diseases, such as alcoholism, epilepsy, ence to the occurrence of seizures in malaria, syphilis, and tuberculosis. After the patients with acute catatonic syndrome is work of Kraepelin and Bleuler, however, anecdotal. Twenty nine patients with there has been a tendency to consider catato- acute catatonic syndrome were reviewed nia as a purely psychiatric disorder, com- to identify patients with seizures after the monly associated with schizophrenia.23 onset of acute catatonic syndrome. Recently, many reports have emphasised the Patients were divided into four diagnos- association between catatonia and a variety tic groups: affective (15), schizophrenic of medical, neurological, and psychiatric con- (eight), toxic (two), and organic (four). ditions4 and catatonia has again been Seizures occurred in four patients regarded as a non-specific entity that has a Clinica Neurologica (13-8%): two patients with dystonic wide range of organic and psychiatric causes.2 dell'Universita di seizures had viral encephalitis and schiz- Genova, via A De Toni 5, 16132 Genova, ophrenic disorder respectively; one Italy patient with complex partial seizures had Table 1 Causes ofacute catatonia A Primavera viral encephalitis and one patient with No ofpatients A Fonti absence status had neuroleptic malignant G Roccatagliata Affective disorders 15 (51-7%) L Cocito syndrome. The results indicate the value Major depression 10 with features 2 of EEG in detection of epileptic activity Major depression psychotic http://jnnp.bmj.com/ Divisione Bipolar disorders 3 Neurologica, Ospedali in patients with acute catatonic syn- Schizophrenic disorders 8 (27 6%) Gailiera, 16128 drome, both at onset and in the course of Schizophrenia 4 Genova, Italy Schizophreniform disorders 3 P Novello such disturbance, particularly to provide Brief reactive psychosis 1 a differential diagnosis between pseudo- Toxic disorders 2 (6-9%) Correspondence to: Neuroleptic malignant syndrome 2 Dr Leonardo Cocito. seizures and neuroleptic-induced acute Organic disorders 4 (13-8%) Received 23 December 1993 dystonia. Encephalitis 3 and in revised form Nutritional encephalopathy 1 29 June 1994.

Total 29 on September 26, 2021 by guest. Protected copyright. Accepted 6 July 1994 (J Neurol Neurosurg Psychiatry 1994;57: 1419-1422)

Table 2 Clinical details ofpatients with acute catatonic syndrome and seizures Outcome of acute Sex! Main clinical Previous psychiatric Previous Type of catatonic No age Diagnosis findings disorders epilepsy seizure Frequency AED treatment syndrome 1 F/17 Schizophrenic Fever 38'C Obsessive-compulsive No Dystonic Recurrent Phenobarbitone Full disorder Tachycardia disorder in childhood seizures recovery 2 F/23 Viral encephalitis Fever >40'C No No Dystonic Recurrent Phenobarbitone Improvement Respiratory failure seizures Serum CK = 259 U/I 3 F/22 Neuroleptic Fever 39-5°C Recent acute psychosis No Absence Status Diazepam Improvement malignant Tachycardia (150) status syndrome Serum CK = 2000 U/I epilepticus Myoglobinuria Respiratory failure Adynamic ileus 4 F/67 Viral encephalitis Fever 38'C Affective illness No Complex Recurrent Carbamazepine Improvement Respiratory failure (bipolar) partial seizures CK = creatine kinase; AED = antiepileptic drug. Primavera, Fonti, Novello, Roccatagliata, Cocito 1420 J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.57.11.1419 on 1 November 1994. Downloaded from B C 1-5 A

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ECG 50R L.. 50os L.. is 1s who had seizures associated with cortical electrodecremental events (CEEs). (A) (t = 0): onset Ictal EEG in patient No I (a 17year old woman) dystonic = 30 s): waves and no clinical change; (B) (t = 20 s): onset ofCEE, rapidly followed by tonic convulsion; (C) (t of the antack with generalised sharp of motor activity; continuation of both CEE and motor seizure activity; (D) (t = 40s) and (E) (t = 50 s): persistence of CEE despite cessation (F) (t = 60 s): gradual restoration of basal EEG activity. Epileptic seizures in patients with acute catatonic syndrome 1421 J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.57.11.1419 on 1 November 1994. Downloaded from

The association between catatonia and included seizures occurring outside the cata- epilepsy was emphasised by Kahlbaum in his tonic periods. As these rates are definitely original monograph,3 but it has received scant higher than those of an age matched general attention in the modem literature.2 Despite a population, a merely coincidental association few reports of adequately documented catato- of seizures and catatonia is unlikely. Seizures nia with concurrent epileptic discharges on were more frequent in patients with organic the EEG5-7 and of non-convulsive status catatonia, but they occasionally occurred in epilepticus presenting as ictal catatonia,' the acute catatonic syndrome due to psychiatric reference to the occurrence of seizures in disorders as well. Epileptic seizures in the catatonic patients is usually anecdotal. The course of acute catatonic syndrome can be aim of this study was to investigate the occur- triggered by miscellaneous causes such as rence of seizures in patients with acute cata- hyperpyrexia, liver and renal failure, throm- tonic syndrome. boembolism, neuroleptic drugs, and CNS infections. Critically ill patients with acute metabolic events or drug toxicity may have Patients and methods new onset seizures.'0 One of our patients had Twenty nine patients (11 men, 18 women; a non-convulsive status epilepticus, as age range 15-78; mean age 43-2 (SD 200) reported by Lim et al.8 Absence status epilep- with acute catatonic syndrome have been ret- ticus occurring de novo in adults often results rospectively identified from clinical charts of from a conjunction of several epileptogenic patients admitted to the Department of factors, such as an excessive amount or with- Neurology of the University of Genova in the drawal of psychotropic drugs or toxic or years 1984-92. According to Barnes et al,'2 the metabolic factors"I 12; all of these conditions minimum definition of acute catatonic syn- are likely to occur in acute catatonic syn- drome required that patients rapidly devel- drome and can thus account for epileptic oped, over a period of days or weeks, at least seizures in some patients. Such a non-specific one motor sign (catalepsy, posturing, or waxy mechanism is unlikely, however, to represent flexibility) in combination with at least one of the only explanation. One half of patients the following clinical signs: (a) negativism, with epileptic seizures during acute catatonic mutism, or stupor; (b) excitement; (c) bizarre syndrome had dystonic seizures with cortical repetition behaviour. All patients had a electrodecremental events, which are quite detailed assessment that included physical, unusual in adults.9 These seizures are sup- neurological, and psychiatric examination, posed to arise from a subcortical focus, possi- neuroimaging, EEG, CSF examination, and bly located in the brainstem.9 1' 14 Subcortical appropriate screening tests for the known discharges in the EEG have occasionally been metabolic and systemic causes of the syn- reported in patients with catatonic stupor.'5 drome.4 Table 1 indicates the likely causes of The relation between the brainstem (particu- acute catatonic syndrome in the 29 patients. larly the midbrain) and catatonic states has The clinical charts of patients with acute been a recurring theme, which has received catatonic syndrome were reviewed to identify some support from experimental and patho- the occurrence of epileptic seizures during the logical evidence3 and, more recently, by neu- catatonic periods. The diagnosis of seizures roimaging.'' It can be speculated that both was based on the clinical findings and EEG seizures and acute catatonic syndrome might recording. sometimes result from an involvement of the http://jnnp.bmj.com/ same neural structures, possibly located in the diencephalon, midbrain, and pons. Then Results epileptic seizures in the course of acute cata- Seizures during the catatonic periods tonic syndrome are likely to arise from multi- occurred in four out of the 29 patients with ple mechanisms, some of which are possibly acute catatonic syndrome (13-8%). Table 2 related to the pathophysiology of catatonia.

summarises the clinical features of these The reported efficacy of anticonvulsive treat- on September 26, 2021 by guest. Protected copyright. patients. ment in catatonial7 and, conversely, the rela- One patient had complex partial seizures, tion between periodic electrographic one patient had absence status epilepticus, complexes and dopamine insufficiency in and two patients had dystonic seizures associ- neuroleptic malignant syndrome,'8 indirectly ated with cortical electrodecremental events suggest that some common mechanisms may as described by Fariello et al.9 The figure underly both catatonia and seizures. shows the EEG findings in one of these The possible occurrence of epileptic patients. seizures during acute catatonic syndrome emphasises the importance of EEG monitor- ing in these patients, both at the onset and in Discussion the course of the syndrome. In the absence of Our series has confirmed that catatonia is a an ictal EEG, the occurrence of fits with non-specific entity that has a wide range of uncommon features may lead to a wrong organic and psychiatric causes,2 provided that diagnosis of non-epileptic attacks, as pseudo- schizophrenic disorders accounted for only seizures and neuroleptic-induced acute dys- 27-6% of the cases. The frequency of seizures tonia are common in patients with acute in patients with acute catatonic syndrome behaviour disturbances. 9 A correct identifica- (13-8%) was close to the 16% reported by tion of epileptic seizures is essential, however, Barnes et al,2 although this figure also given the different therapeutic implications. 1422 Primavera, Cocito Fonti, Novello, Roccatagliata, J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.57.11.1419 on 1 November 1994. Downloaded from

This study was partially supported by CNR (Center for electrodecremental event: clinical and neurophysio- Cerebral Neurophysiology, Geneva) and by MURST. logical observations in patients with dystonic seizures. Arch Neurol 1979;36:285-91. 10 Wijdicks EFM, Sharbrough FW. New-onset seizures in critically ill patients. Neurology 1993;43:1042-4. 1 Kahlbaum KL. Die Katatonie, oder das 11 Van Sweden B, Mellerio F. Toxic ictal confusion. Spannungsirresein. Berlin, Klinische Abhandlungen iuber JEpilepsy 1988;1:157-63. psychische Krankheiten, 1874. 12 Thomas P, Lebrun C, Chatel M. De novo absence status 2 Barnes MP, Saunders M, Walls TJ, Saunders I, Kirk CA. epilepticus as a benzodiazepine withdrawal syndrome. The syndrome of Karl Ludwig Kahlbaum. Neurol Epilepsia 1993;34:355-8. Neurosurg Psychiatry 1986;49:991-6. 13 Mutani R, Bergamini L, Fariello R. A case of status 3 Johnson J. Catatonia: the tension insanity. Br Psychiatry epilepticus with tonic expression (so-called "reticular 1993;162:733-8. epilepsy"). Epilepsia 1970;11:321-6. 4 Gelenberg AJ. The catatonic syndrome. Lancet 1976:i: 14 Nathanson M, Krumholz A, Biddle D. Seizures of axial 1339-41. structures. Arch Neurol 1978;35:448-52. 5 Thompson SW, Greenhouse AH. Petit mal status in 15 Johnson J. Stupor: a review of 25 cases. Acta Psychiatr adults. Ann Intern Med 1968;68:1271-9. Scand 1984;70:370-7. 6 Goldensohn ES, Gold AP. Prolonged behavioral distur- 16 Joseph AB, Anderson WH, O'Leary DH. Brain stem and bances as ictal phenomena. Neurology (Minneap) 1960; vermis atrophy in catatonia. Am Psychiatry 1985: 10:1-9. 142:352-4. 7 Gomez EA, Comstock BS, Rosario A. Organic versus 17 Rankel HW, Rankel LE. Carbamazepine in the treatment functional etiology in catatonia: case report. Y Clin of catatonia. Am Psychiaty 1988;145:361-2. Psychiatry 1982;43:200-1. 18 Nelson L, Kramer LD, Locke GE. Periodic EEG com- 8 Lim J, Yagnik P, Schraeder P, Wheeler S. Ictal catatonia plexes, myoclonus, and neuroleptic malignant syndrome as a manifestation of nonconvulsive status epilepticus. [abstract]. Epilepsia 1987;28:607. Neurol Neurosurg Psychiatry 1986;49:833-6. 19 Lloyd GG. Acute behaviour disturbances. Neurol 9 Fariello RG, Doro JM, Forster FM. Generalized cortical Neurosurg Psychiatry 1993;56: 1149-56. http://jnnp.bmj.com/ on September 26, 2021 by guest. Protected copyright.