Electroconvulsive Treatment for Catatonia in Autism Spectrum Disorders.

Catatonia has been increasingly recognized in people with autism spectrum disorders (ASD). Assessment, diagnosis, and treatments are reviewed and illustrated with 2 new case vignettes. The use of electroconvulsive treatment (ECT) is recommended in patients who fail to respond to medical treatments, including a trial of lorazepam or another benzodiazepine. The importance of maintenance ECT is discussed. There is an urgent need for prospective studies of catatonia in ASD and for controlled treatment trials.

Electroconvulsive Therapy for Catatonia in Children and Adolescents.

Catatonia may be more common in children and adolescents than previously thought. A boost for the recognition of pediatric catatonia comes from changes in Diagnostic and Statistical Manual of Mental Disorders, 5th edition, facilitating the diagnosis in a wide range of pediatric and adult patients with associated developmental and autistic spectrum disorders; and schizophrenic, affective, and medical disorders. The current status, assessment, and treatment of pediatric catatonia are described. Two case vignettes illustrate diagnostic assessment and treatment. Theories modeling the mechanism of catatonia are reviewed, including a vagal theory.

Neuroleptic Malignant Syndrome/Malignant Catatonia in Child Psychiatry: Literature Review and a Case Series.

OBJECTIVE: To describe the presentation of neuroleptic malignant syndrome (NMS) and malignant catatonia (MC) in children and adolescents. BACKGROUND: NMS and MC are life-threatening, neuropsychiatric syndromes, associated with considerable morbidity and mortality. NMS is diagnosed when there is a recent history of treatment with an antipsychotic (AP) medication, while MC is diagnosed when the symptoms resemble NMS but without a history of exposure to an AP agent. Some authorities believe that apart from the history of exposure to an AP medication, the two conditions are identical. The symptoms of NMS/MC include severe agitation, behavior disregulation, motor and speech changes, self-injury and aggression, autonomic instability, and a range of psychiatric symptoms (affective, anxiety, or psychotic symptoms). Patients may be misdiagnosed with another disorder leading to extensive tests and a delay in treatment. Untreated, the condition may be fatal in 10%-20% of patients, with death sometimes occurring within days of disease onset. METHOD: We describe the presentation and management of five children and adolescents with NMS/MC. CONCLUSION: MC and NMS are life-threatening medical emergencies, which if diagnosed promptly, can be successfully treated with known effective treatments (benzodiazepines and/or electroconvulsive therapy).

Catatonia in Ugandan children with nodding syndrome and effects of treatment with lorazepam: a pilot study.

BACKGROUND: Nodding syndrome (NS) is a severe neuropsychiatric syndrome of an unknown etiology affecting children and adolescents mostly in Eastern Africa. Symptoms of NS and catatonia seem to overlap. We investigated the presence and types of catatonic symptoms in NS and their response to one or two doses of lorazepam, the first-line treatment for catatonia. METHODS: A cross-sectional descriptive study with systematic assessment of catatonia in 33 patients with NS using a modified version of the Bush Francis Catatonia Rating Scale. Sixteen patients met criteria for catatonia and were observed in an open and uncontrolled study to examine the effects of one or two doses of lorazepam in them. RESULTS: Sixteen of 33 patients with NS had an average of 5 catatonia symptoms and met criteria for catatonia. The highest scores were found for mutism, staring, poor eating/drinking, stupor, and grimacing. Excitement, rigidity, negativism and impulsivity had lower scores. None of the children had echolalia or echopraxia. In 6 children, there was a reduction of more than 50% in catatonia ratings, representing a positive response to lorazepam. Three out of six children whose catatonia ratings did not change after the first dose, responded after administration of a second double dose. There were no unusual or critical side-effects. CONCLUSIONS: About half of a selected sample of children with NS met criteria for catatonia. Catatonia scores decreased in most patients after one or two doses of lorazepam. Larger, longer, and controlled studies are warranted to assess the prevalence of catatonia in NS and to assess the use of lorazepam in NS through its effects on catatonia. TRIAL REGISTRATION: ClinicalTrials.gov NCT02462109 Date of formal registration: June 2, 2015.

A clinical review of the treatment of catatonia.

Catatonia is a severe motor syndrome with an estimated prevalence among psychiatric inpatients of about 10%. At times, it is life-threatening especially in its malignant form when complicated by fever and autonomic disturbances. Catatonia can accompany many different psychiatric illnesses and somatic diseases. In order to recognize the catatonic syndrome, apart from thorough and repeated observation, a clinical examination is needed. A screening instrument, such as the Bush-Francis Catatonia Rating Scale, can guide the clinician through the neuropsychiatric examination. Although severe and life-threatening, catatonia has a good prognosis. Research on the treatment of catatonia is scarce, but there is overwhelming clinical evidence of the efficacy of benzodiazepines, such as lorazepam, and electroconvulsive therapy.

Vagal intimations for catatonia and electroconvulsive therapy.

OBJECTIVES: Catatonia is currently viewed as a unique syndrome that consists of specific motor signs responding to benzodiazepines and electroconvulsive therapy (ECT). Advances in catatonia may provide a new window into the mechanism of ECT. Findings on catatonia are updated and related to the mechanism of ECT. METHODS: Selective literature review. RESULTS: There are several putative models and mechanisms of catatonia concerning motor circuitry dysfunction, abnormal neurotransmitters, epilepsy, genetic risk factors, and endocrine and immune dysfunction. Fear and vagal nerve models are presented casting catatonia in an evolutionary-based autonomic neural substrate of social behaviors, engagement, and disengagement according to perceived level of danger. Benzodiazepines and ECT are thought to have autonomic and vagal effects. CONCLUSIONS: Advances in catatonia provide a new window into the mechanism of ECT. Fear and vagal nerve models call for further anatomical, functional, and clinical studies on the vagal nerve and for further studies on the use of anticholinergic medications in catatonia, on vagal function in catatonia, and on the effects of benzodiazepines, ECT, and induced seizures on cholinergic and vagal function.

Stability of intellectual functioning during maintenance electroconvulsive therapy.

We describe the stability of neuropsychologic testing in a 16-year-old boy with cerebellar dysgenesis who received 61 acute and maintenance electroconvulsive therapy treatments for malignant catatonia. Measures of nonverbal intelligence and visual memory before treatment onset and after 61 electroconvulsive therapy treatments indicated no evidence of decline in intellectual functioning and acute or delayed memory. This case offers further support for the safety and efficacy of maintenance electroconvulsive therapy in both pediatric and developmentally disabled populations.

Presence of GAD65 autoantibodies in the serum of children with autism or ADHD.

Antibodies against glutamic acid decarboxylase 65 (GAD65) have been detected in the serum of patients with several neurological disorders. The presence of antibodies against GAD65 has not yet been examined in the serum of patients with neurodevelopmental disorders such as autism or attention-deficit/hyperactivity disorder (ADHD). In this study, GAD65 antibodies and total IgG were assayed in the serum of normal subjects and patients diagnosed with autism or ADHD. GAD65 antibodies were detected in the serum of 15% of children with autism (N = 20), 27% of children with ADHD (N = 15) and of none of the controls (N = 14). The serum of 60% of autistic and 53% of ADHD patients reacted with Purkinje neurons in mouse cerebellum. Serum from 20% of ADHD patients reacted also with the cells in the molecular and granule cell layers and cells in the vicinity of the Purkinje neurons. No association was found between the titer of GAD65 antibodies and total IgG levels, and presence of seizures or mental retardation. None of the ADHD patients were diagnosed with mental retardation. Serum anti-GAD65 antibodies may be a common marker of subgroups of patients with autism and ADHD. Reactions of serum antibodies with the cells in the cerebellum in these patients suggest direct effects on brain function. The subgroup of children with autism and ADHD that tests positive for GAD65 antibodies needs further characterization in a larger study.

When is electroconvulsive therapy appropriate for children and adolescents?

The indications for electroconvulsive therapy in children and adolescents are similar to those in adults, including severe affective, psychotic and catatonic pathology that has proven refractory to psychotropic medications and causes significant functional impairment. ECT may be indicated as well in specific pediatric neurological conditions. Multiple published reports demonstrate the safety and efficacy of ECT in pediatric patients with a wide range of psychopathology. ECT has also been successfully used in youth with autism and other neurodevelopmental disabilities who present with catatonic deterioration. However, resistance and stigma persist regarding the use of ECT in children and adolescents in both the professional and lay communities, creating barriers to pediatric ECT access. We argue that the use of ECT in children and adolescents is appropriate for specific clinical indications, and urge removal of impediments to ECT access in this population.

Tics as signs of catatonia: electroconvulsive therapy response in 2 men.

OBJECTIVES: Tics have rarely been described in catatonia although tics are sudden and nonrhythmic variants of stereotypic or repetitive movement abnormalities that are considered cardinal symptoms of catatonia. We describe 2 men with tics and self-injurious behavior, who met criteria for catatonia. One patient met criteria for autism. CASE REPORTS: We reported 2 new cases and performed a literature review using PubMed to identify other cases of tics that were treated with electroconvulsive therapy. Tics along with other catatonic symptoms and self-injurious behavior responded to electroconvulsive therapy in 2 men. Eight other patients with tics that were treated with electroconvulsive therapy were found in the literature. Catatonia was recognized in 4 of the 8 patients. Two patients met criteria for autism. CONCLUSIONS: Tics, with or without self-injurious behavior, may be signs of catatonia. Patients with tics or Tourette syndrome warrant assessment for catatonia. If catatonia is present, electroconvulsive therapy provides a safe but rarely used alternative to pharmacotherapy, psychosurgery, or invasive brain stimulation in the treatment of tics and Tourette 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.

Electroconvulsive therapy for pediatric malignant catatonia with cerebellar dysgenesis.

Electroconvulsive therapy was successfully used to treat malignant catatonia in a 15-year-old male patient with congenital dysgenesis of the left hemisphere of the cerebellum and hypoplasia of the vermis and left pons due to a presumed cerebral vascular accident in utero. The patient experienced significant motor and communication delays with mild cognitive impairment, but was otherwise in good health until age 15 years, when he developed rigidity, posturing, stupor, unresponsiveness, repetitive self-injurious behaviors, and negativism, as well as autonomic abnormalities including profuse diaphoresis and flushing episodes, thus meeting criteria for malignant catatonia. After initial response to lorazepam, the patient required electroconvulsive therapy for resolution of malignant catatonia. The case supports the safe and efficacious usage of electroconvulsive therapy for catatonia in adolescents with cerebellar and other developmental disorders. The role of the cerebellum in catatonia is also reviewed.

Catatonia is hidden in plain sight among different pediatric disorders: a review article.

Over the past two decades, catatonia has been better demarcated in adult psychiatry as a unique syndrome that consists of specific motor signs with a characteristic response to benzodiazepines and electroconvulsive therapy. Pediatric catatonia is considered rare, but may be underdiagnosed, and hence undertreated. Discussed here are the current diagnostic criteria of catatonia in individual cases of children and adolescents diagnosed with childhood disintegrative disorder, Kleine-Levin syndrome, Prader-Willi syndrome, tic disorder, and autoimmune encephalitis, and the effects of benzodiazepines and electroconvulsive therapy. In these cases, catatonia resolved safely once it was recognized and treated properly. Children and adolescents presenting with these disorders should be systematically assessed for catatonia; when the presence of catatonia is confirmed, the use of benzodiazepines and electroconvulsive therapy should be considered. The occurrence of catatonia in such a wide range of child and adolescent disorders supports the view that pediatric catatonia is not so rare, that there are shared elements in the etiology, psychopathology, and pathophysiology of these disorders, and that catatonia is best classified as a unique neurobiologic syndrome.

Self-injury in autism as an alternate sign of catatonia: implications for electroconvulsive therapy.

Multiple reports show the efficacious usage of ECT for catatonia in individuals with autism. There are also a few reports showing that ECT improves self-injury in people with and without autism. In this hypothesis, self-injury in autism and other developmental disorders may be an alternate sign of catatonia, and as such an indication for electroconvulsive therapy. The issue is important because self-injury occurs at an increased rate in autistic and intellectually disabled individuals, but is poorly understood and often difficult to treat with psychological and pharmacological means. Self-injury may be considered a type of stereotypy, a classic symptom of catatonia that is exquisitely responsive to electroconvulsive therapy (ECT). Historical and modern reports further support the association of self-injury, tics and catatonia. Central gamma-aminobutyric acid (GABA) dysfunction may provide an important explanatory link between autism, catatonia and self-injury. Therefore, people with autism and other developmental disorders who develop severe self-injury (with or without concomitant tics) should be assessed for catatonia, and ECT should be considered as a treatment option. Further studies of the utility of ECT as an accepted treatment for catatonia are warranted in the study of self-injury in autism.

Maintenance electroconvulsive therapy in autistic catatonia: a case series review.

The usage of electroconvulsive therapy for the acute resolution of catatonia in autistic children and adults is a novel area that has received increased attention over the past few years. Reported length of the acute ECT course varies among these patients, and there is no current literature on maintenance ECT in autism. The maintenance ECT courses of three patients with autism who developed catatonia are presented. Clinical, research, legal, and administrative implications for ECT treatment in this special population are discussed.

Electroconvulsive therapy for malignant catatonia in adolescence.

Malignant catatonia is a severe form of catatonia which is acute in onset, and is systemically devastating, as it is associated with fever and autonomic instability. Although rarely described in children and adolescents, malignant catatonia should feature prominently in the differential diagnosis of acute pediatric encephalopathies, as it is a treatable syndrome. The case of a 17-year-old adolescent with sickle cell anemia is presented. The patient developed malignant catatonia and responded dramatically to electroconvulsive therapy.