Untitled

Acta neurol. belg., 2007, 107, 47-50
Catatonia and neuroleptic malignant syndrome : two sides of a coin ?
Dept. of Neurology, University Hospital Gent, Gent, Belgium in mood disorders (especially mania) (Taylorand Fink, 2001 ; Peralta et al., 1997). A major point Catatonia was first described by Kahlbaum in 1874. Ever since, the concept of catatonia has been the focus in the discussion is the nosological status of catato- of debate, a major point of discussion being its nosolog- nia ; is it to be considered an aspecific syndrome ical status. The question rises whether it is to be consid- with a panoply of possible clinical signs and symp- ered a syndrome with a wide variety of causes and clin- toms (Table 1) or is it a separate entity for which ical signs or a distinct clinical entity. Since catatonia proper diagnostic criteria should be developed, as shares a number of symptoms with the neuroleptic suggested by Taylor and Fink (2003) (Table 2).
malignant syndrome (NMS) and similar treatments can Since the introduction of neuroleptics, the inci- be used in both conditions, it has also been suggested dence of catatonia has decreased significantly that NMS and catatonia are two variants of the same (Northoff, 1997). However, it became clear that disorder. In this article we describe five cases of catato- administration of neuroleptics could eventually nia and NMS in order to approach this nosological ques- lead to catatonia-like symptoms (Bush et al., tion. The clinical similarity between both syndromes isdemonstrated in our cases. On the level of pathophysiol- 1996), as is the case in neuroleptic malignant ogy however, catatonia and NMS are quite different, syndrome (NMS). NMS shares with catatonia a with catatonia rather being a cortical psychomotor number of symptoms e.g. immobility, rigidity and syndrome and NMS a subcortical motor disorder. stupor (Koch et al., 2000). Moreover, treatment Similarities can be explained by means of well-known strategies applied to catatonic patients may also be models of basal ganglia function. The nosological prob- beneficial to NMS patients. Therefore, some have lem, however, can only be resolved when the concept of suggested that NMS and catatonia are variants of the same disorder (Fink and Taylor, 2001).
Key words : Catatonia ; neuroleptic malignant syn- We will report on five cases of catatonia and drome ; neuroleptics ; benzodiazepines ; fronto- NMS and discuss the issue of classification of both Case reports
Introduction
The concept of catatonia, literally meaning “to stretch tight”, goes back to the original description This 55-year old man sustained a severe trau- by Karl Ludwig Kahlbaum in 1874. In his mono- matic brain injury. He was in a coma for one week graph Kahlbaum characterized catatonia as a motor after which he was left with an organic brain syn- disorder representing a phase in an illness that pro- drome. Due to a severe behavioral disorder treat- gresses from mania, depression and psychosis to a ment with multiple neuroleptics was necessary. final end stage of dementia (Kahlbaum, 1974 ; Fink One year after his head injury he came to the and Taylor, 2001). Due to the ideas raised by emergency room for an erysipelas for which antibi- Kraepelin and Bleuler, the concept of catatonia was otic treatment was started. At that moment he was embedded into the classification of schizophrenia treated with risperdone and pimozide. On admis- (Pfuhlmann and Stöber, 2001). Although numerous sion he did not respond adequately, was extremely authors raised arguments against this view, the idea apathetic and his consciousness was found to be of catatonia as a subtype of schizophrenia persisted decreased. Generalised muscular rigidity was throughout the 20th century and was adopted in all found, as well as elevated body temperature. Levels DSM versions, including DSM-IV. Nevertheless, of creatin kinase were also raised, supporting a apart from its association with psychotic disorders, diagnosis of neuroleptic malignant syndrome.
catatonia has been described in general medical Neuro-imaging demonstrated aspecific and mild conditions (medical catatonia), and most frequently akinesia, bradykinesia, hypokinesie, hyperkinesias, rigidity, Negativism, posturing, grimacing, stereotypies, psychosis, maniccatalepsy, gegenhalten, paratonia, primitive reflexes, persevera- behaviour, agitation, withdrawal, regression, mannerisms, rituals,tions, impulsivity, tics utilization behaviour, imitation behaviour, echopraxia, automaticobedience Proposed diagnostic criteria for catatonia, as proposed by Fink and Taylor (2003) A. Immobility, mutism, or stupor of at least 1 hour’s duration, B. In the absence of immobility, mutism, or stupor, at least two associated with at least one of the following : observed or elicited on two or more occasions.
– automatic obedience– posturing– negativism– gegenhalten– ambitendencyobserved or elicited on two or more occasions Neuroleptic treatment was stopped and a treatment treatment by means of doxycyclin. The patient regimen with 10 mg t.i.d. bromocriptine, 100 mg recovered over a period of three weeks.
t.i.d. dantrolene and 2 mg clonazepam IV was start-ed. The creatin kinase level normalized and the clinical image improved gradually over a period oftwo of weeks. The patient was transferred to apsychiatric ward. The ultimate diagnosis was A 49-year old woman with a previous history neuroleptic malignant syndrome in a patient with of alcohol abuse and depressive episodes was admitted for investigation of subacute dementia. The clinical picture consisted of bradyphrenia and hypokinesia, a fine tremor of the upper limbs,decreased facial expression, disinhibited primitive A 35-year-old man with known bipolar disorder reflexes, diminished attention and memory.
was admitted to the intensive care unit for Perseverations, neologisms, stereotypies and echo- auto-intoxication with alprazolam, paroxetine and ing were notably present. Comprehension could lithium. The episode was complicated by renal insufficiency, aspiration pneumonia, development Serum tests did not show any abnormality.
of pressure wounds, bruises and rib fractures. After Investigation for infectious and auto-immune caus- recovery he was admitted to the psychiatric ward es remained negative. The CSF did not show signs where an episode of psychotic depression was of central nervous system inflammation. Magnetic diagnosed. He was treated with haloperidol, which resonance imaging of the brain was normal, as well was switched to risperidone later on.
as the evaluation of cerebral blood flow by means One month after his suicide attempt he was of SPECT-scan. The EEG showed an increase of admitted to the neurological ward with progressive slow wave activity. Screening for paraneoplastic dyspnoea, a slight cough, swallowing problems and disorders also remained negative, except for some apathy. On clinical examination a bilateral marked aspecific sequellar micronodules in the lungs and cogwheel rigidity, especially at the upper limbs, 2 biliar cysts in the liver, as seen on CT.
was found as well as additional axial rigidity. He In the absence of any clear organic cause this also had little facial expression, a bulbar dysarthria picture was diagnosed as a catatonic episode prob- with marked salivation and swallowing problems.
ably due to an underlying psychotic depression. A Imaging of the brain was normal. Levels of creatin treatment with 4 mg q.i.d. intravenously lorazepam kinase were elevated (1100 U/L). The diagnosis of was rapidly successful and the patient was trans- neuroleptic malignant syndrome was made. His ferred to the psychiatric ward. The diagnosis of neuroleptic treatment was stopped and 10 mg t.i.d.
psychotic depression was confirmed. She was bromocriptine, 100 mg t.i.d. dantrolen and 1 mg treated with an SSRI and is doing well since. The t.i.d. lorazepam were started, along with antibiotic original hypothesis of dementia was not retained.
CATATONIA AND NEUROLEPTIC MALIGNANT SYNDROME Discussion
This 66-year old woman had a previous history Three out of our 5 presented patients suffered of recurrent depressive episodes and psychosis, for from NMS, while two were diagnosed as catatonic, which she had been treated with electro-convulsive in one patient there were arguments for both albeit therapy. She was admitted to a psychiatric ward in a sequence of events. As is clear from these because of paranoid delusions, and a treatment cases, there are many similarities between NMS with neuroleptics was started. She developed swal- and catatonia. On a symptomatic level, both condi- lowing problems and shortly thereafter a pneumo- tions share the occurrence of specific symptoms nia was diagnosed. As her consciousness deterio- like akinesia, muscle rigidity, stupor and mutism.
rated and respiratory parameters worsened she was Clinical similarities between catatonia and NMS temporarily admitted to the intensive care ward. At have also been confirmed by Koch et al. (2000), that time a generalised increase of muscle tone with who found that most of their patients meeting cogwheel rigidity and hypokinesia was noted.
criteria for NMS, simultaneously met clinical Based on this clinical picture and the elevated and research criteria for catatonia. On the other serum levels of creatin kinase a diagnosis of neuro- hand, there are also clear differences. Behavioral leptic malignant syndrome was suspected.
symptoms are more prominent in catatonia, but She was also treated with 4 mg q.i.d. intra- symptoms of autonomic dysfunction are character- venously lorazepam and the clinical picture cleared in three weeks. The patient remained emotionally NMS and catatonia also share similarities in unstable, with crying fits, and showed a very treatment strategies. As was demonstrated in our negativistic attitude as well as bizarre somatoform case reports, benzodiazepines are considered delusions. She was transferred to the psychiatric standard therapy in both conditions. In the NMS, ward for further treatment. The final diagnosis was dantrolene and dopamine receptor agonists are also NMS and catatonia due to psychotic depression.
useful. In resistant cases, electroconvulsive therapyis shown to be effective in both conditions (Koch etal., 2000). Most cases of NMS and catatonia are now readily treatable, if adequately recognized.
In an excellent review on the subject of NMS A 67-year old woman, known with a history of and catatonia, Northoff (2002) proposes an inter- psychosis and alcohol and benzodiazepine abuse, esting hypothesis explaining both the similarities was admitted to the neurology department because and the differences between both entities. The pre- of motor and cognitive deterioration. On clinical sented evidence from functional neuro-imaging, testing generalized muscle rigidity, diminished neurophysiology, neurochemical and neurophar- reflexes, mutism and negativistic behaviour were macological data, converges to a model of predom- inant cortical dysfunction in catatonia, which is thus to be regarded as a cortical “psychomotor syn- except for a cortico-subcortical atrophy and some drome”, while NMS is to be considered a subcorti- aspecific white matter changes. Based on the histo- cal “motor disorder”. The predominant areas of ry taking a diagnosis of progressive dementia was functional disturbance in catatonia are supposed to suspected and the hypothesis of dementia with be the medial orbitofrontal cortex and the posterior parietal cortex. The former is specifically involved After a ten days treatment with diazepam, in affective and behavioral symptoms, while the risperidone and rivastigmine the patient’s situation latter is involved in spatial characteristics of move- improved substantially with increase of verbal out- ment, and hence perhaps in adequate termination of put and better cooperation. A brief episode of pul- ongoing movements. The focus of dysfunction in monary infection induced a recurrence of the same NMS is supposed to be the striatum, especially the symptomatology and the diagnosis of catatonia was posterior part, involved in initiation of movements, made based on the presence of negativism, mutism, and to a lesser extent the ventral and anterior parts rigidity, hypokinesia and oppositional behaviour.
of the striatum, involved in cognitive and behav- Because of this catatonic picture, she was treated ioral control. Moreover, in catatonia a functional with lorazepam IV and additionally with amytripti- down-regulation of the cortical GABA system was line, levodopa. The clinical signs improved gradu- found, especially in the right posterior parietal ally although episodes of oppositional behaviour cortex, and the right lateral orbitofrontal cortex persisted and stereotypic movements occurred.
(Northoff et al., 1999), while in NMS a predomi- The final diagnosis was a primary degenerative nance of dopaminergic dysfunction in the striatum dementia, most probably dementia with lewy is suspected. This is reflected in the treatment bodies, accompanied by catatonia, which could be considered inherent to the clinical picture although illustrates and underscores the problem of the nosological status of catatonia. The well-know KAHLBAUM K. L. Catatonia (Die katatonie oder das span- cortico-subcortical model of Alexander and nungsirresein).1973, John Hopkins University DeLong (1986), with parallel loops mediating motor, cognitive and behavioral function allows the KOCH M., CHANDRAGIRI S., RIZVI S., PETRIDES G., description of clinical syndromes with alike symp- FRANCIS A. Catatonic signs in neuroleptic malig-nant syndrome. Comprehensive Psychiatry, 2000, tomatology due to dysfunctions in different parts of 41 (1) : 73-75.
the system, be it cortical or subcortical, a sugges- NORTHOFF G., ECKERT J., FRITZE J. Glutamatergic dys- tion already raised by Cummings (1993).
function in catatonia ? Successful treatment of The issue of nosology can therefore only be three acute akinetic catatonic patients with the resolved by either restricting the concept of catato- NMDA-antagonist amantadine. Journal of nia or broadening it. In a restrictive way catatonia Neurology, Neurosurgery and Psychiatry, 1997, could be defined as a psychomotor disorder in 62 (4) : 404-406.
the context of psychosis or affective disorder. A NORTHOFF G., STEINKE R., CZCERVENKA C., KRAUSE R., broader concept of catatonia would be defined as a ULRICH S., DANOS P., KROPF D., OTTO H., BOGERTS psychomotor syndrome due to any condition, be B. Decreased density of GABA-A receptors in the it medical, pharmacological or psychiatric. As left sensorimotor cortex in akinetic catatonia :investigation of in vivo benzodiazepine receptor follows from the present discussion many of the binding. Journal of Neurology, Neurosurgery and Psychiatry, 1999, 67 (4) : 445-450.
encountered in frontal lobe dysfunction. However, NORTHOFF G. Catatonia and neuroleptic malignant syn- it should be acknowledged that subcortical drome : psychopathology and pathophysiology.
dysfunction, as in NMS, may also lead to similar Journal of Neural Transmission, 2002, 109 :
clinical syndromes. In our view, the concept of catatonia and its relation to fronto-subcortical PERALTA V., CUESTA M. J., SERRANO J. F., MATA I. The dysfunction deserves more attention. The restric- Kahlbaum syndrome : a study of its clinical tion of catatonia to a subtype of schizophrenia is, in validity, nosological status, and relationship with schizophrenia and mood disorder. Comprehensive
Psychiatry, 1997, 38 (1) : 61-67.
FUHLMANN B., STÖBER G. The different conceptions of catatonia : historical overview and critical discus-sion. European Archives of Psychiatry and ALEXANDER G. E., DELONG M. R., STRICK P. L. Parallel Clinical Neuroscience, 2001, 251 (suppl. 1) : I/4-
organization of functionally segregated circuits linking basal ganglia and cortex. Annual Review of Neuroscience, 1986, 9 : 357-381.
AYLOR M. A., FINK M. Catatonia in psychiatric classifi- cation : a home of its own. American Journal of BUSH G., FINK M., PETRIDES G., DOWLING F., FRANCIS A.
Psychiatry, 2003, 160 (7) : 1233-1241.
Catatonia II : treatment with lorazepam and
electroconvulsive therapy. Acta Psychiatrica
Scandinavica, 1996, 93 : 137-143.
CUMMINGS J. L. Frontal-subcortical circuits and human behaviour. Archives of Neurology, 1993, 50 (8) :
873-880.
INK M., TAYLOR M. A. The many varieties of catatonia.
European Archives of Psychiatry and Clinical Neuroscience, 2001, 251 (suppl. 1) : I/8-I/13.

Source: http://www.actaneurologica.be/acta/download/2007-2/04-Vancaester.pdf