Classification issues in catatonia

Kahlbaum described catatonia as a disorder in which mood syndromes were the primary features and the characteristic symptoms were the motor signs. In the present study, we examined the relationship between motor features and other syndromes of psychosis, the clinical validity of Kahlbaum's concept of catatonia, its relationship to schizophrenia and mood disorder, and its nosological position in relation to DSM-III-R, DSM-IV and Leonhard's classification of endogenous psychoses. Patients with Kahlbaum's catatonia differed from patients with schizophrenia or mood disorder in various demographic and clinical variables. Positive and negative motor syndromes, although interrelated, showed a different correlational pattern with other psychotic syndromes. Catatonia did not appear to fit into any particular nosological category, although this issue largely depends on whether schizophrenia and mood disorders are broadly or restrictively defined. When definitions are more restrictive as in Leonhard's system, catatonia seems to be better accommodated as a “third psychosis”, i.e. described by the concept of cycloid psychosis.

     

Catatonia in pediatric obsessive–compulsive disorder: report of two cases

Catatonia is a psychomotor syndrome which may occur in a wide variety of medical, neurological, and psychiatric conditions. In pediatrics, this condition is rare and is associated with high morbidity and mortality if not correctly diagnosed and treated. Catatonia in obsessive–compulsive disorder is an infrequent association that has been understudied and underdiagnosed. To add to the knowledge on this unusual clinical presentation, two pediatric patients are reported and discussed together with the other two cases described in the literature. These four cases in total of catatonia associated with OCD confirm that it is a relationship that is infrequently reported, possibly because of lack of awareness in clinicians that catatonia can also be caused by OCD, and because the similarity between some catatonic signs and some compulsive phenomena may compound the identification of the former. Most cases of catatonia in this small series seemed to have responded to the optimization of the treatment for OCD. This highlights the clinical importance of an accurate diagnosis of catatonia when associated with OCD.

     

The many varieties of catatonia

About 10 % of patients with severe acute psychiatric illness exhibit a cluster of motor signs (mutism, negativism, rigidity, posturing, stereotypy, staring, etc.) that are identified as the syndrome of catatonia. Catatonia responds to sedative anticonvulsant treatment (barbiturates, benzodiazepines) and to electroconvulsive therapy. These treatments raise seizure thresholds. The commonality in response indicates that catatonia, malignant catatonia, neuroleptic malignant syndrome, toxic serotonin syndrome, delirious mania, catatonic excitement, benign stupor, and oneirophrenia are best evaluated as diverse manifestations of one syndrome for clinical and neuroscience research purposes.

     

The Catatonia Conundrum: Evidence of Psychomotor Phenomena as a Symptom Dimension in Psychotic Disorders

To provide a rational basis for reconceptualizing catatonia in Diagnostic and Statistical Manual of Mental Disorders (Fifth Edition), we briefly review historical sources, the psychopathology of catatonia, and the relevance of catatonic schizophrenia in contemporary practice and research. In contrast to Kahlbaum, Kraepelin and others (Jaspers, Kleist, and Schneider) recognized the prevalence of motor symptoms in diverse psychiatric disorders but concluded that the unique pattern and persistence of certain psychomotor phenomena defined a “catatonic” subtype of schizophrenia, based on intensive long-term studies. The enduring controversy and confusion that ensued underscores the fact that the main problem with catatonia is not just its place in Diagnostic and Statistical Manual of Mental Disorders but rather its lack of conceptual clarity. There still are no accepted principles on what makes a symptom catatonic and no consensus on which signs and symptoms constitute a catatonic syndrome. The resulting heterogeneity is reflected in treatment studies that show that stuporous catatonia in any acute disorder responds to benzodiazepines or electroconvulsive therapy, whereas catatonia in the context of chronic schizophrenia is phenomenologically different and less responsive to either modality. Although psychomotor phenomena are an intrinsic feature of acute and especially chronic schizophrenia, they are insufficiently recognized in practice and research but may have significant implications for treatment outcome and neurobiological studies. While devising a separate category of catatonia as a nonspecific syndrome has heuristic value, it may be equally if not more important to re-examine the psychopathological basis for defining psychomotor symptoms as catatonic and to re-establish psychomotor phenomena as a fundamental symptom dimension or criterion for both psychotic and mood disorders.     

Genetic predisposition and environmental causes in periodic and systematic catatonia

The nosological heterogeneity of catatonic schizophrenia was the focus of a family study involving 139 catatonic patients. The clinical dichotomization of catatonia into periodic catatonia and systematic catatonia revealed good inter-rater reliability and stability of the diagnoses at follow-up. Analysis of the cumulative morbidity risk among first-degree relatives gave an excessive familial aggregation of homogenous psychoses in periodic catatonia with a risk of 26.9 %. The mode of inheritance was consistent with an autosomal dominant model. In contrast, first-degree relatives of probands with systematic catatonia had a morbidity risk of 4.6 %. Subsequent investigations pointed to a potential association of systematic catatonia to exposure to midgestational infections in the index cases and linked these sporadic disorders to disturbances of fetal neurodevelopment. The periodic catatonia, an unequivocally genetically transmitted illness, is at present under investigation in a genome-wide linkage analysis.

     

Catatonia Is not Schizophrenia: Kraepelin's Error and the Need to Recognize Catatonia as an Independent Syndrome in Medical Nomenclature

Catatonia is a motor dysregulation syndrome described by Karl Kahlbaum in 1874. He understood catatonia as a disease of its own. Others quickly recognized it among diverse disorders, but Emil Kraepelin made it a linchpin of his concept of dementia praecox. Eugen Bleuler endorsed this singular association. During the 20th century, catatonia has been considered a type of schizophrenia. In the 1970s, American authors identified catatonia in patients with mania and depression, as a toxic response, and in general medical and neurologic illnesses. It was only occasionally found in patients with schizophrenia. When looked for, catatonia is found in 10% or more of acute psychiatric admissions. It is readily diagnosable, verifiable by a lorazepam challenge test, and rapidly treatable. Even in its most lethal forms, it responds to high doses of lorazepam or to electroconvulsive therapy. These treatments are not accepted for patients with schizophrenia. Prompt recognition and treatment saves lives. It is time to place catatonia into its own home in the psychiatric classification.     

Catatonic syndrome: From detection to therapy

IntroductionCatatonia is a psychomotor syndrome which can include motor, mental, behavioral and vegetative symptoms. Exclusively associated with schizophrenia until the 1970s, catatonia still remains an under-diagnosed syndrome with significant morbidity and mortality.Literature findingsAs a result of its different forms and developments, catatonic syndrome can be associated with many organic and psychiatric etiologies and confused with a variety of diagnoses. In addition to its organic complications, malignant catatonia can also be extremely severe. Several diagnostic scales are described, those of Bush and Peralta being the most widely used. Despite the recent development of the DSM-5, we can regret the lack of progress in the international classifications concerning both the recognition of the etiological diversity of this syndrome and in the clinical and therapeutic approaches to it. The diagnosis is based solely on clinical data, and needs to be completed by information from paraclinical settings, particularly with respect to detecting organic etiology. The first-line treatment is still based on the use of certain benzodiazepines or benzodiazepine-like agents such as lorazepam, diazepam and zolpidem. If the first or second line fails, or in case of malignant catatonia, electroconvulsive therapy is recommended. For the periodic form, no large-sample study has been performed on long-term treatment. A few case reports suggest the use of lithium in periodic catatonia, specifically to prevent recurrent episodes or at least to extend the inter-episode intervals. Other studies are in favor of the use of benzodiazepines, with disagreement between gradual discontinuation and long-term treatment. Concerning the management of catatonia in patients with schizophrenia, for whom first-line benzodiazepines are often insufficient, certain atypical antipsychotics such as clozapine or quetiapine appear efficient. These data are also applicable to children and adolescents.ConclusionOften neglected by practitioners, catatonic syndrome remains a common entity of which it is important to be aware, especially in case of rapid installation of the symptoms. Diagnostic scales should be used and a lorazepam test should be performed to avoid delaying the diagnosis. Second-line therapy requires further study. This concerns in particular diazepam, anti-NMDA (N-methyl-D-aspartate) and rTMS (repetitive transcranial magnetic stimulation). Some specificities of catatonia, such as the periodic form and cases in patients with schizophrenia, also require further evaluations.     

ECT Has Much to Offer Our Patients: It Should Wot Be Ignored

ECT is an effective treatment for severe mental disorders, including major depression, delusional depression, bipolar disorder, manic delirium, schizophrenia, malignant catatonia, and neuroleptic malignant syndrome. It reduces suicidality, melancholia, catatonia, aggression, and excitement. Age is no barrier, with ECT successful in children, adolescents, and the old-old. It is effective when other treatments have failed.

It is a safe treatment. The mortality rate is less than that associated with normal pregnancies. The fractures, panic, and amnesia that marked early treatments have now been modified. Even amnesia, which is often described as the main objection of patients, and the relapse rates are now comparable to the effects of medicines.

It is an efficient treatment, costing less for a course of treatment than conventional new medicines and hospitalization.

National and international psychiatric societies ignore ECT in their educational programmes. I believe that the leaders have a duty to our patients to provide a forum and educational opportunities for their members for all interventions that may relieve mental disorders. To ignore ECT, an effective treatment, is to do a disservice to our patients, and to abrogate the Hippocratic Oath to which many practitioners subscribe.     

Retrospective chart review of catatonia in child and adolescent psychiatric patients

Ghaziuddin N, Dhossche D, Marcotte K. Retrospective chart review of catatonia in child and adolescent psychiatric patients. Objective: Identify the frequency of catatonia among at‐risk children and adolescents receiving psychiatric treatment. Method: Subjects were children and adolescents (<18 years), who had received psychiatric treatment at a University Hospital during 2004–2009, and were diagnosed with disorders with known risk for catatonia or displayed symptoms suggestive of catatonia. Approval was obtained from the Investigational Review Board (IRB). The first 101 (n = 101) subjects were selected among 570 subjects identified by psychiatric diagnoses: any pervasive developmental disorder, psychosis‐NOS (Not Otherwise Specified), intermittent explosive disorder, mental retardation, catatonia and neuroleptic malignant syndrome. Subjects met study‐defined criteria for catatonia, if they had three or more of the following symptoms: unexplained agitation/excitement, disturbed or unusual movements, reduced movements, repetitive or stereotyped movements, or reduced or loss of speech. Results: Eighteen (17.8%) subjects, among a group suspected to be at a higher risk for catatonia, met the study‐defined criteria for this syndrome. However, only two subjects had been diagnosed by their treatment providers. Higher rates of intellectual disability and aggression were found among the group that met study‐criteria. Conclusion: We concluded that catatonia is under recognized and undertreated among children and adolescents receiving psychiatric treatment.     

Catatonia in autism: implications across the life span

Background  There is increasing evidence that catatonia is an important source of impairment in adolescents and adults with autism. Aim  Review of the evaluation, diagnosis, differential diagnosis, and treatment of catatonia in autism. Method  Presentation and discussion of a case-vignette spanning early childhood to adulthood. Results  Autistic and catatonic symptoms overlap, yet catatonia is diagnosable in about one of seven adolescents and young adults with autism. Case-reports suggest that benzodiazepines and electroconvulsive therapy are effective treatments in the acute and maintenance phase for people with autism who develop catatonia. Conclusions  Catatonia should be assessed in people with autism when there is an obvious and marked deterioration in movement, vocalizations, pattern of activities, self-care, and practical skills. Benzodiazepines and electroconvulsive therapy are favored options for acute and maintenance treatment in these cases. Further studies on the possible biological-genetic overlap between autism and catatonia would be helpful.     

Prevalence and clinical correlations of catatonia in older adults referred to a liaison psychiatry service in a general hospital

Aim

To determine the frequency and clinical correlations of catatonia in older patients referred to a liaison psychiatry service in a general hospital.

Patients and Methods

All patients over 65 years referred to liaison psychiatry were screened for catatonic phenomena with the Bush–Francis Catatonia Screening Instrument (BFCSI) between January and May 2012. Their clinical characteristics and the outcome of treatment were recorded.

Results

One hundred and twelve patients over 65 years were referred. Ten (8.9%) met research diagnostic criteria for catatonia and 7 (6.3%) Diagnostic and Statistical Manual of Mental Disorders, 4th Edition criteria for catatonia. All patients presented with the inhibited variant and 5 to 12 catatonic signs. Three patients presented concomitant delirium. The etiology of catatonia was multifactorial, and complications and death were frequent (40% and 20%, respectively). Lorazepam achieved full resolution of catatonia in 50% of patients.

Conclusions

Catatonia in older adults referred to liaison psychiatry in a general hospital is not infrequent and has a multifactorial etiology. The BFCSI is a simple and reliable instrument to detect catatonia in this population. Lorazepam seems to be an effective treatment.     

Long-term maintenance lorazepam for catatonia: a case report

Catatonia is described as a syndrome of motor abnormality associated with the disorder of thought, behavior and emotions. Lorazepam has been shown to be useful in the short-term management of catatonia [Ungvari G.S., Kau L.S., Wai-Kwong T., Shing N.F., The pharmacological treatment of catatonia: an overview. Eur Arch Psychiatry Clin Neurosci 2001;251(suppl 1):31-34; Daniels J., Catatonia: clinical aspects and neurobiological correlates. J Neuropsychiatry Clin Neurosci 2009;21:371-380]. However, there is sparse literature with respect to patients requiring long-term maintenance lorazepam for catatonia. Manjunatha et al. [Manjunatha N., Saddichha S., Khess C.R.J., Idiopathic recurrent catatonia needs maintenance lorazepam: case report and review. Aust NZ J Psychiatry 2007;41:625-627] described a case which required long-term maintenance lorazepam for recurrent catatonia that was unresponsive to most antipsychotics. Gaind et al. [Gaind G.S., Rosebush P.I., Mazurek M.F., Lorazepam treatment of acute and chronic catatonia in two mentally retarded brothers. J Clin Psychiatry 1994;55:20-23] described the use of maintenance lorazepam in a mentally retarded boy with catatonia of 5 years' duration, which improved slowly over a period of 5 months.We present a case of recurrent catatonia, in which symptoms relapsed whenever an attempt was made to taper off lorazepam.     

Benzodiazepines in catatonia associated with systemic lupus erythematosus

Neuropsychiatric disturbances are found in 50-70% of systemic lupus erythematosus (SLE) patients. However, there are rare cases of catatonia being described in SLE. Some studies have shown the effectiveness of high-dose steroid, plasma exchange and electroconvulsive therapy (ECT) in lupus catatonia. Herein are described two SLE patients with catatonia who had good response to i.v. diazepam (i.e. relief of catatonia symptoms). Patient 1, with mild cortical atrophy, had great improvement in catatonia symptoms on i.v. diazepam 150 mg during a period of 5 days. Patient 2, without cortical atrophy, had quick response to i.v. diazepam 10-20 mg. Both patients had no recurrence during 6-month follow up. In conclusion, benzodiazepines may play an important role in the treatment of catatonia associated with SLE if patients refuse ECT treatment.     

Towards the genetic basis of periodic catatonia: pedigree sample for genome scan I and II

In a genome-wide linkage study, we mapped two major susceptibility loci for periodic catatonia, a phenotype with qualitative disturbances of the psychomotor sphere and a morbidity risk of 26.9 % in first-degree relatives of index cases, to chromosome 15q15, and to chromosome 22q13 using nonparametric as well as parametric (autosomal dominant model) analyses. The study included 12 multiplex pedigrees with 135 individuals, among them 57 affected persons. A second genome scan is in progress investigating four families with 21 affected individuals, aiming to confirm linkage results. Age at onset patterns as well as the clinical outcome were similar among affected individuals in both sets of families. Within the pedigrees we observed no physical diseases segregating with periodic catatonia. Under the assumption of genetic homogeneity, the statistical power to detect LOD scores ≥ 2.0 was 98.5 % in the first set of families, and 57.9 % in the second set. Thus, the panel of multiplex pedigrees segregating periodic catatonia seems to represent a homogenous clinical sample, and possesses sufficient statistical power to delineate and confirm linkage to major genetic loci for periodic catatonia.

     

Benzodiazepines in the treatment of catatonia

A patient who developed acute catatonia during benzodiazepine withdrawal is discussed. The case prompted us to review the literature on the role of benzodiazepines in the treatment of acute catatonia. Only retrospective and open studies were found which indicate that benzodiazepines do have a beneficial effect. Lorazepam is the most widely studied benzodiazepine and at present is the best treatment option. In the specific case of acute catatonia brought on by benzodiazepine withdrawal the recommended dosage is the same as for acute catatonia caused by something other than benzodiazepine withdrawal.     

L’enseignement du syndrome catatonique en France : enquête nationale et état des lieux

ObjectivesCatatonia is a transnosographic syndrome described by K. Kahlbaum in 1874. Catatonia can be life-threatening due to its complications and in case of malignant catatonia. Safe and effective treatments have been identified (benzodiazepines and electro-convulsive-therapy). The prevalence of this syndrome is important and represents about 10% of inpatients in an acute psychiatric ward. However, this syndrome appears to remain under-diagnosed and poorly known. We were, therefore, interested in the current knowledge of French psychiatry residents and young psychiatrists as well as the education they had received about catatonia.MethodsA questionnaire was submitted to French psychiatry residents and young psychiatrists with fewer than 5 years of experience. It included questions about knowledge of the symptoms, complications, causes and treatments of catatonia. Participants were also asked about their confidence in the management of a patient with catatonia and about the number of catatonic patients they had already met. The type of lecture and teaching about catatonia was also assessed.ResultsAmong the 376 psychiatrists that completed the questionnaire, 37.5% never had received any specific teaching about catatonia. Concerning the 62.5% who benefited from a specific lecture, this was mainly delivered as part of psychiatry DES. Heterogeneity in the education delivery on the French territory had been highlighted. In addition, participants’ knowledge of catatonic syndrome seems incomplete. However, knowledge of catatonic symptoms and first intention treatments was significantly better among respondents who were part of the “with education” group. The confidence in catatonia recognition and management, as well as the number of catatonic patients they met, are also significantly higher in the group “with education”.ConclusionSpecific education seems to improve the knowledge of young doctors and their ability to diagnose and treat catatonic patients. This education remains poorly provided and heterogeneous on the French territory. Catatonia deserves a place in the teaching program of the psychiatry DES, thus to become systematic.     

Lethal catatonia: clinical aspects and therapeutic intervention. A review of the literature

Lethal catatonia continues to occur and represents a nonspecific syndrome associated with diverse organic as well as functional conditions. From this perspective, neuroleptic malignant syndrome may be conceptualized as a neuroleptic-induced toxic or iatrogenic form of organic lethal catatonia. Neuroleptics appear ineffective in the treatment of lethal catatonia and should be stopped whenever this disorder is suspected. Existing data suggest that ECT is a safe and effective treatment for lethal catatonia. ECT also appears effective in the treatment of neuroleptic malignant syndrome.     

Etiopathogenesis of catatonia: generalizations and working hypotheses

Catatonia has been rediscovered over the last 2 decades as a unique syndrome that consists of specific motor signs with a characteristic and uniform response to benzodiazepines and electroconvulsive therapy. Further inquiry into its developmental, environmental, psychological, and biological underpinnings is warranted. In this review, medical catatonia models of motor circuitry dysfunction, abnormal neurotransmitters, epilepsy, genetic risk factors, endocrine dysfunction, and immune abnormalities are discussed. Developmental, environmental, and psychological risk factors for catatonia are currently unknown. The following hypotheses need to be tested: neuroleptic malignant syndrome is a drug-induced form of malignant catatonia; Prader-Willi syndrome is a clinical GABAergic genetic-endocrine model of catatonia; Kleine-Levin syndrome represents a periodic form of adolescent catatonia; and anti-N-methyl-d-aspartate receptor encephalitis is an autoimmune type of catatonia.