Catatonia among adolescents with Down syndrome: a review and 2 case reports

Catatonia is a relatively common condition with an estimated prevalence of 0.6% to 17% among youth with psychiatric disorders. Certain patient groups, such as those with autism, may be at a particularly high risk for catatonia. Most of the youth with catatonia are males with a diagnosis of a bipolar disorder. We describe here 2 adolescent females, both with Down syndrome, who presented with catatonia not accompanied by significant affective or psychotic symptoms or with a general medical condition. Both patients had functioned well until the onset of catatonic symptoms. In the current classification system used in the Diagnostic and Statistical Manual of Mental Disorders, Fourth Edition, catatonia is described in association with schizophrenia, as a specifier of affective disorders or secondary to general medical conditions. The cases described here highlight the problem with this classification system when patients fail to meet any of the 3 diagnostic categories under which catatonia is currently described.     

Catatonia as the presenting symptom in systemic lupus erythematosus

Catatonia is a syndrome of physical and behavioral abnormalities that can result from psychiatric, neurological, or medical illness. Although systemic lupus erythematosus (SLE) is commonly known to cause neurological and psychiatric manifestations, it has only rarely been reported to cause the catatonic syndrome. In nearly all previously reported cases, the diagnosis of catatonia was reported in patients with an established diagnosis of lupus. We report a case in which a woman with no known medical history presented with catatonia that did not respond to standard treatment with benzodiazepines, suffered a long and complicated hospital course, and was eventually diagnosed with lupus. With initiation of treatment for lupus, her symptoms of catatonia remitted. This case illustrates the importance of considering medical causes in the diagnosis and treatment of psychiatric disorders, especially the catatonic syndrome.     

Catatonia in childhood and adolescence

Child and adolescent catatonia has been poorly investigated. A literature review was undertaken to clarify phenomenology, diagnosis, etiology, and treatment as well as ethical problems of catatonia in childhood and adolescence. Although there are no accepted standardized criteria for catatonia in childhood and adolescence, catatonic features described by child psychiatrists are similar to Diagnostic and Statistical Manual of Mental Disorders (4th edn; DSM-IV) criteria for catatonia. With respect to etiology, the motor and behavioral symptoms that are part of catatonia bear some similarities with those seen in autism. Several case reports suggest an association between catatonia and general medical conditions. Certain drugs abused by youngsters as well as prescribed medicine can induce catatonia. Regarding catatonic cases originally diagnosed as schizophrenia, it is unclear whether all of these cases should be identified as schizophrenia or whether some of them are pervasive developmental disorders that develop psychic features in adolescence. Environmental changes preceding the onset of catatonia in patients with mood disorder play a possibly important role. Examples that suggest stress-induced catatonia, although rare, also exist. A few patients exhibit features of malignant catatonia, some without taking neuroleptics and others having taken them. Benzodiazepines and electroconvulsive therapy are considered to be effective treatments for catatonic youngsters.     

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.

     

Catatonia in a Psychiatric Intensive Care Facility: Incidence and Response to Benzodiazepines

This study was performed to establish the incidence of catatonia in a psychiatric intensive care unit, to test the Bush-Francis Catatonia Screening Instrument (BFCSI) and to assess the response of catatonic signs to benzodiazepines. During a 12-month period all patients admitted to a psychiatric intensive care unit were screened for catatonic signs using the BFCSI. Patients with catatonia were further assessed with the Bush-Francis Catatonia Rating Scale (BFCRS), the Modified Rogers Scale (MRS), and scales for associated psychotic and parkinsonian symptoms. They were treated with oral lorazepam or parenteral clonazepam and their responses evaluated daily. Neuroleptics were stopped for at least 3 days. Twenty four patients met the DSM IV criteria for catatonia, giving an incidence of 15% with a significantly higher proportion of non-Europeans. The most common associated diagnosis was schizophrenia (54%). Twenty two patients completed the benzodiazepine trial. All showed significant responses after 3 days of treatment. Sixteen (16/22, 73%) had full remission within 6 days, most within 2 to 4 days. Partial responders (n = 6) all had schizophrenia and were more likely to have longer pre-trial catatonic episodes. We find the BFCSI a simple and reliable tool to screen for catatonia, and our data attest to the efficacy of benzodiazepines in the treatment of catatonia.     

Idiopathic recurrent catatonia needs maintenance lorazepam: case report and review

OBJECTIVES: Catatonia as a phenomenon has been well described with either a schizophrenic illness, severe mood disorders or periodic catatonia disorder. We aim to report a patient who had recurrent catatonia that responded to and required lorazepam for maintenance. METHODS: We describe the case of a 28 year old woman who had a history of recurrent catatonia that was unresponsive to most anti-psychotics, but who responded to high doses of lorazepam and needed long-term lorazepam for maintenance. RESULTS AND CONCLUSION: Our patient met the criteria for a diagnosis of idiopathic catatonic disorder. Response to lorazepam suggests that a certain group of patients may require long-term treatment with lorazepam, especially those who may have down-regulation of GABA-A receptors.     

Schizophrenia with prominent catatonic features ('catatonic schizophrenia'): I. Demographic and clinical correlates in the chronic phase

This study set out to determine the frequency of catatonic syndrome in chronic schizophrenia and its association with sociodemographic, clinical, and treatment variables. A cross-sectional assessment of a randomly selected cohort of patients (n=225; mean age=42+/-7 years; mean length of illness=20.4+/-7.5 years) with DSM-IV schizophrenia was employed using standard rating instruments for catatonia, drug-induced extrapyramidal symptoms (EPS), and psychotic, depressive, and obsessive-compulsive symptoms. Using a rather narrow definition of catatonia [the presence of four or more signs/symptoms with at least one having a score '2' or above on the Bush-Francis Catatonia Rating Scale (BFCRS)], 72 subjects (32%) met the criteria for the catatonia group (mean number of catatonic signs/symptoms=5.9+/-2.0; mean sum score of 8.7+/-3.4 on the BFCRS). The frequency distribution of catatonic signs/symptoms in the catatonic group and in the whole sample was very similar, with mannerisms, grimacing, stereotypes, posturing, and mutism being the most frequent. In the logistic regression analysis, catatonic subjects had a significantly earlier age of onset, more negative symptoms, and were more likely to receive benzodiazepines than their noncatatonic counterparts. In multiple regression analysis, the severity of catatonia as indicated by the sum score of BFCRS was predicted only by earlier age of onset and negative symptoms. Using relatively narrow criteria, this study confirmed that, if methodically assessed, catatonic signs and symptoms are prevalent in patients with chronic schizophrenia. Catatonia can be differentiated from EPS. Catatonic features indicate a generally poor prognosis in the chronic phase of schizophrenia.     

Memantine and catatonia: a case report and literature review

Catatonia is a movement disorder with various possible etiologies. The majority of cases are associated with an underlying mood or psychotic disorder, while others are caused by medical conditions. Currently, benzodiazepines are the first-line psychopharmacologic agents in the treatment of catatonia. However, several cases have been reported in which treatment with memantine proved to be effective. We present the case of a 92-year-old female with major depressive disorder and associated catatonic symptoms. In this case, the patient's symptoms remitted quickly after the initiation of memantine. We review the possible causes of catatonia and pharmacologic treatments for the condition and highlight the possible benefits of N-methylD-aspartic acid receptor antagonists such as memantine in the treatment of catatonia.     

Response of catatonia to risperidone: two case reports

The present study describes two patients, both of Yemenite origin, with catatonic schizophrenia who responded to treatment with risperidone. One had a long history of psychiatric disorder, whereas the other was a first-episode, drug-naive patient. Our observation agrees with previous reports on the use of risperidone and other novel neuroleptic agents in the treatment of catatonia of different etiologies.     

精神科急性入院患者中紧张症的识别和治疗

目的回顾性分析精神科急性入院患者中紧张症的识别和治疗情况。方法对2017年10月1日至2018年10月1日我院重症精神病房急性入院患者1020例的病历资料进行回顾性分析,根据《美国精神障碍诊断与统计手册第5版》(Diagnostic and Statistical Manual of Mental Disorders,5th Edition,DSM-5)紧张症的诊断标准进行诊断,参照Bush-Francis紧张症评定量表(Bush-Francis catatonia rating scale,BFCRS)的症状定义归类确诊病例的临床表现,并比较不同治疗方案的疗效。结果确诊紧张症91例,占调查患者的8.92%。81例(89.01%)紧张症患者的基础疾病为精神分裂症。患者最常见的紧张症症状为缄默(81.32%)、兴奋(72.53%)、冲动性(71.43%)、静止/呆滞(65.93%)和作态(51.65%),另外先占观念(92.30%)和怪异念头(58.24%)也较常见。仅有7例(7.69%)患者的病历资料中曾提及“(亚)木僵”、“紧张症/综合征”等词。30例患者仅用抗精神病药治疗,治愈率为83.33%(25例),仅用抗精神病药治疗者与抗精神病药联合苯二氮 类和/或改良电休克治疗者的治愈率差异无统计学意义(X^2=1.75,P=0.63)。1例患者在肌注氟哌啶醇后出现恶性综合征。结论精神科急性入院患者中紧张症并不少见,但识别率低。抗精神病药能有效治疗紧张症,但存在出现恶性综合征的风险。     

Clinical and cerebral blood flow changes in catatonic patients treated with ECT

Objective: To determine changes in clinical manifestations and cerebral blood flow (CBF) before and after administration of ECT to patients with catatonia due to schizophrenia or mood disorders. Methods: A sample of nine patients who met DSM-IV criteria for catatonia was studied. Patients received between 5 and 15 sessions (thrice per week) of ECT. Severity of catatonia was measured with the Modified Rogers Scale (MRS). Changes in CBF were measured with a brain single positron emission computer tomography (SPECT) that was performed 1 week before the first ECT and 1 week after the last ECT. Results: Catatonia was due to schizophrenia in five patients and mood disorders in four patients. There were no significant clinical and brain SPECT differences between these two groups before treatment. Pre- vs. post-ECT comparisons showed significant reduction of catatonic symptoms in both groups. However, patients with mood disorders needed less ECT sessions and showed greater clinical improvement. Brain SPECT showed significant increase in CBF in parietal, temporal, and occipital regions in patients with mood disorder and no significant changes in patients with schizophrenia. Conclusions: These results support the efficacy of ECT for treatment of catatonic patients, especially secondary to mood disorders, which seem to be correlated with improvements in CBF.     

Catatonia: prediction of outcome

Of 250 patients diagnosed as having catatonia, 40% of the 214 for whom follow-up information was available had at some time recovered completely. Although certain features of history and mental status significantly predicted recovery, or its absence, many factors differed from those used in studies of mixed subtypes of schizophrenia. Most motor symptoms correlated poorly with outcome: in particular, those symptoms classically associated with catatonia (catalepsy, posturing, staring, mutism, rigidity, grimacing, negativism) proved poor predictors of outcome. A seven-factor scale used in previous studies allowed refinement of prognosis, but the majority of cases were bunched in the middle of the scale, leaving relatively few cases in the good- and poor-prognosis groups. Duration of illness from onset of prodromal symptoms to hospitalization correlated with outcome as strongly as did the seven-factor scale and provided a more useful distribution of cases among the prognostic groups. Catatonia, a syndrome that has long been associated with schizophrenia, has a prognosis generally better than other subtypes of schizophrenia. The presence of catatonic symptoms should be grounds for separately reporting these patients in studies of treatment and outcome.     

Does recurrent catatonia manifest in a similar fashion in all the episodes of mood disorder? A case series with literature review

Catatonia, originally conceptualised by Kahlbaum in 1868, is a neuropsychiatric condition that has been found to occur concomitantly with several organic and psychiatric conditions. Starting from the era of Kraepelin and Bleuler, this condition was faultily linked with schizophrenia alone; however, over time, greater associations have been found between catatonia and mood disorders. Despite the availability of several reports supporting this finding, there is a relative paucity of studies that specifically focus on catatonia to be the first symptom manifestation heralding a subsequent mood episode. In addition, there is scant literature to determine whether there are specific presentations of catatonia that show greater associations with mood disorders and whether these signs and symptoms recur in a stereotypical fashion in the subsequent mood episodes in the lifetime of an individual. We hereby report two cases with a diagnosis of mood disorders (bipolar disorder and recurrent depressive disorder) who had catatonia as the initial symptom not only at presentation but also at subsequent episodes. The report emphasises that recurrent catatonia can be the initial clinical manifestation of an underlying mood episode, which appears otherwise masked behind the catatonic presentation. These catatonic symptoms can be interestingly similar in all the subsequent episodes. A detailed clinical evaluation is thus warranted after catatonia has been duly treated to provide a holistic management.     

Catatonia in autistic disorder: A sign of comorbidity or variable expression?

Catatonia, once solely attributed to schizophrenia, is now thought to be associated with many disorders. Autistic disorder shares some symptoms with catatonia, namely, mutism, echopraxia/echolalia, and sterotypes. Catatonia in autism may therefore be a variant of the autistic condition. However, organic deficits and psychiatric disorders, such as bipolar disorder, have also been deficits and psychiatric disorders, such as bipolar disorder, have also been linked with the manifestation of catatonia. Individuals with autism presenting with these comorbid conditions may therefore be at increased risk for catatonia. Little is written of the association of autism and catatonia to clarify the possibility of catatonia as a variant or a sign of a comorbid condition. The authors discuss three autistic patients and suggest specific etiologies for the symptoms of catatonia which presented in these cases. The therapeutic and diagnostic importance of comorbid disorders in autism is stressed.     

Clinical relevance of chronic catatonic schizophrenia in children and adolescents: evidence from a prospective naturalistic study

The paper examines the phenomenology, diagnosis, and course of catatonia in children and adolescents. From 1993 to 2003, 21 boys and 9 girls, aged 12 to 18 years, were admitted for a catatonic syndrome (0.6% of the total inpatient population). Phenomenology and associated diagnoses were similar to those reported in the adult literature but relative frequency differed, with schizophrenia being the most frequent diagnosis. Comparison of patients with schizophrenia (n=17) to those with other diagnoses (n=13) showed that the two groups differed in terms of sex ratio, type of onset and phenomenology of catatonic symptoms, duration of hospitalization, and severity at discharge. Using discriminant function analysis, the combination of three clinical variables--male gender, duration of catatonic episode, and severity at discharge--correctly classified 100% of cases in the schizophrenia group. Catatonia is an infrequent but severe condition in young people, and is usually associated with schizophrenia. There is a need for research in the field of catatonic schizophrenia in adolescents as it appears to be a clinically relevant but understudied subgroup.     

Catatonia and consultation-liaison psychiatry study of 12 cases

Nowadays, catatonia is no more considered as a subtype of schizophrenia. Catatonia seems more frequently associated to mood disorders as well as general medical conditions. It is sometimes difficult to associate formally a medical etiology to this syndrome. But we found, in the literature, three groups of associated general medical conditions: neurological disorders, drug induced and toxic induced conditions, metabolic conditions. We present a prospective study of 12 clinical cases of catatonia due to general medical conditions we realized in the Consultation-Liaison Psychiatry Department of the University Hospital of LILLE (France) during a period of 5 months. We find coherent data with the literature. However, our results suggest that if medical conditions precipitate the catatonia syndrome, they are rarely its only etiology. We think that if somatic factors are co-morbid with psychiatric conditions they do not necessarily predominate as the target of treatment. The treatment of the catatonia must be a priority and remain symptomatic, to allow in parallel the specific treatment for the somatic disorder or the psychiatric disorder.     

Risperidone and lorazepam concomitant use in clonazepam refractory catatonia: a case report

The DSM-IV recognizes catatonia as a subtype of schizophrenia characterized by at least two of the following: motor immobility, excessive motor activity not influenced by external stimuli, and peculiarities of voluntary movement. Catatonia may also occur secondary to mania, depression, or a general medical condition including encephalitis, focal neurological lesions, metabolic disturbances, and drug intoxications and withdrawals. Benzodiazepines remain the first line of treatment; up to 80% of patients respond promptly to Lorazepam challenge; failure to respond to lorazepam may be followed by electroconvulsive therapy. Atypical antipsychotics may be a new alternative in the treatment of catatonia. Successful reduction of the catatonic symptoms has been demonstrated with atypical antipsychotics. A possible mechanism of action for the efficacy of this class of drugs involves the antagonism of the 5-HT2A receptor. We are now reporting a case of treatment response to risperidone in a patient with chronic catatonia resistant to benzodiazepines.     

Electroconvulsive therapy,catatonia, deep vein thrombosis and anticoagulant treatment: a case report

Electroconvulsive therapy (ECT) is considered an effective treatment for pharmacotherapy-resistant severe mental disorders. Catatonia is a complex syndrome characterised by important psychomotor disturbances. Deep vein thrombosis (DVT) and pulmonary embolism (PE) are frequent side effects of prolonged immobility in catatonic patients. Therefore, it is important to resolve the catatonia as soon as possible. ECT is the most effective therapy available and is generally considered a safe procedure. Nevertheless, its use in patients with DVT or PE and anticoagulant treatment remains controversial. We describe a case of a woman in her 40s with a previous diagnosis of bipolar disorder and dysfunctional personality traits. She was hospitalised with persecutory and reference delusions, high emotional lability, anxiety, somatisation and regressive conduct. She later developed catatonic symptoms. No progress was achieved after a month of hospitalisation, despite several pharmacological treatments. She suffered multiple complications of prolonged bedding, such as an extensive DVT of the left common femoral, the external iliac and the common iliac veins. ECT was conducted under treatment with bemiparin. After the third administration, she showed improvement. No major bleeding or PE was developed. The safety of ECT while receiving anticoagulant therapy has been documented, though dosage and type of anticoagulant must be considered. Location of DVT (proximal or distal) may be an important topic to take into account. This report provides further evidence about the efficacy and safety of undergoing ECT in the context of concomitant serious medical conditions, such as DVT and anticoagulant therapy administration.     

Case series: catatonic syndrome in young people.

This article reviews all recent (1977-1997) reports on catatonic adolescents and summarizes the 9 consecutive cases seen at the authors' institution during the past 6 years. Catatonia occurs infrequently in adolescents (0.6% of the inpatient population), but it appears to be a severe syndrome in adolescents of both sexes. Diagnoses associated with catatonia are diverse, including in this series: schizophrenia (n = 6), psychotic depression (n = 1), mania (n = 1), and schizophreniform disorder (n = 1). Two patients had a previous history of pervasive developmental disorder. In the literature, catatonia was also reported in children with organic condition (e.g., epilepsy, encephalitis). Therapeutic management depends on the specific causes, but several points need to be stressed: (1) the frequency of neuroleptic-induced adverse effects; (2) the potential efficacy of sedative drugs on motor signs; (3) the possible use of electroconvulsive therapy; and (4) the necessity to manage family reactions and fears, which are frequent causes of noncooperation. It is concluded that catatonia is an infrequent but severe condition in young people. While symptomatology, etiologies, complications, and treatment are similar to those reported in the adult literature, findings differ with regard to the female-male ratio and the relative frequencies of associated mental disorders.