Multiyear Evaluation of Maintenance Electroconvulsive Therapy in an Adult With Autism Spectrum Disorder, Catatonia, and Challenging Behavior.

ABSTRACT: We report the case of a 30-year-old man diagnosed with autism spectrum disorder who received electroconvulsive therapy (ECT) over a 4-year period to treat catatonia associated with life-threatening self-injury, aggression, major depression, and associated self-care, daily living, and communication skills deficits. A systematic schedule of maintenance ECT (m-ECT) was associated with elimination of challenging behavior, catatonic and depressive symptom remission, removal of protective equipment, and reduced dosages of psychotropic medications.

Treatment of Severe Self-Injurious Behavior in Autism Spectrum Disorder by Neuromodulation.

An increasing number of case reports and series document the safe and effective use of electroconvulsive therapy (ECT) in children, adolescents, and young adults with autism spectrum disorder who engage in severe, intractable, repetitive self-injurious behavior (SIB) without environmental or operant function. Although the treatment is very effective for such patients, they typically remain highly dependent on frequent maintenance ECT (M-ECT) to maintain suppression of the SIB achieved during the acute course. Some patients receive M-ECT as frequently as once every 5 days. Such a regimen is quite burdensome for the patient and the patient's family, and the long-term effects of such regimens, starting as early as childhood, are unknown. In this review, we explore the expanding literature supporting the use of ECT for suppressing severe SIB associated with autism spectrum disorder. We also focus on the possible development of alternate nonconvulsive focal forms of brain stimulation, which might replace frequent M-ECT or reduce how frequently a patient needs to receive it. Although there are scarce clinical data currently available supporting these latter treatments, future studies are clearly indicated.

Catatonia in an adolescent with velo-cardio-facial syndrome.

Velo-cardio-facial syndrome (VCFS) is the most common microdeletion syndrome in humans and is probably the most frequent genetic cause of psychosis currently known. Many psychiatric disorders have been reported to occur in people with VCFS including, but not limited to schizophrenia, unipolar and bipolar mood disorders (with or without psychotic features), schizoaffective disorder, psychosis NOS, social phobia, generalized and separation anxiety, obsessive-compulsive disorder, autism spectrum disorder, cognitive impairment, and ADHD. This report describes the psychiatric onset and development of catatonia in an adolescent female with VCFS that was undiagnosed until 15 years of age. Catatonia may be a relatively common presentation in people with VCFS with treatment-refractory psychiatric manifestations.

Youth with severe mental illness and complex non-somatic motor abnormalities: conflicting conceptualizations and unequal treatment.

Two emerging diagnostic concepts promote distinct treatments for youth with acute-onset motor abnormalities and severe concurrent psychiatric symptoms: Pediatric acute-onset neuropsychiatric syndrome (PANS) and pediatric catatonia. Both have institutional approval in parts of Europe and in the USA, meriting an unconditional comparison of supporting evidence. Here we report results of qualitative and quantitative analyses of literature and Swedish National Registry Data suggesting that (1) catatonic patients are liable to fulfilling diagnostic criteria for PANS, (2) three conservatively assessed PANS case-reports present with possible unrecognized catatonia, (3) lithium and electroconvulsive therapy usage frequencies in Swedish minors (exclusively recommended for severe mental illness) are strongly intercorrelated and unequally distributed across Swedish counties, (4) established severe mental disorders are rarely overtly considered amongst PANS-specific research and (5) best-available evidence treatments appear markedly superior for pediatric catatonia compared to PANS in both childhood and adolescence. Prioritizing treatments for pediatric catatonia in concerned subjects could markedly improve treatment outcomes.

Electroconvulsive therapy for psychotropic-refractory bipolar affective disorder and severe self-injury and aggression in an 11-year-old autistic boy.

We report the successful use of electroconvulsive therapy in a 11-year-old boy with autism and a 4-year history of psychotropic-resistant bipolar affective disorder associated with dangerous episodes of self-injurious and aggressive behaviors placing his caregivers and himself at significant safety risk. Extensive behavioral and medication interventions in both inpatient and outpatient settings had been ineffective, and the boy was at risk for acute physical injury and restrictive out-of-home placement. An acute course of eight bilateral electroconvulsive therapies resulted in significant mood stabilization and significant improvement of self-injury and aggression. Maintenance electroconvulsive therapy and psychotropic interventions were then pursued.

Mask Compliance Training for Individuals With Intellectual and Developmental Disabilities.

Since the arrival of the novel coronavirus, recommendations for public masking have emerged to decrease infection rates. For a variety of reasons, tolerating wearing a mask is challenging for many individuals with intellectual and developmental disabilities (IDDs). Therefore, we evaluated behavioral strategies to promote compliance with wearing a mask with six hospitalized individuals diagnosed with IDDs. One participant was compliant with wearing the mask for extended durations during baseline while engaging in various activities (e.g., academics, leisure). For the other five individuals, engagement in activities alone was ineffective. Blocking mask removal, reinforcement for mask wearing, and noncontingent access to preferred activities or competing stimuli were then evaluated using a changing-criterion design in which the duration participants were required to tolerate the mask gradually increased. Increases in compliance with mask wearing were achieved with all participants; however, the terminal duration was attained for only four of the five individuals. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1007/s40617-021-00583-7.

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.

ECT for self-injury in an autistic boy.

OBJECTIVE: Self-injurious behavior presents a significant challenge in autism, and first-line psychopharmacological and behavioral interventions have limited efficacy in some patients. These intractable cases may be responsive to electroconvulsive therapy. CLINICAL PICTURE: This article presents an eight-year-old boy with autism, mental retardation, prominent mood lability and a five-year history of extreme self-injurious behavior towards his head, averaging 109 self-injurious attempts hourly. The patient was at high risk for serious head trauma, and required usage of bilateral arm restraints and protective equipment (i.e., padding on shoulders, arms, and legs). All areas of daily functioning were profoundly impacted by dangerous self-injury. TREATMENT: Fifteen bilateral ECT treatments resulted in excellent mood stabilization and reduction of self-injury to 19 attempts hourly, and maintenance ECT was pursued. The patient was able to return to developmentally-appropriate educational and social activities. CONCLUSION: ECT should be considered in the treatment algorithm of refractory cases of severe self-injury in autism.

Catatonia and autism: a historical review, with implications for electroconvulsive therapy.

Current autism research is historically separated from catatonia and other childhood psychotic disorders, although catatonia and autism share several common symptoms (mutism, echolalia, stereotypic speech and repetitive behaviors, posturing, grimacing, rigidity, mannerisms, and purposeless agitation). Electroconvulsive therapy (ECT) effectively treats catatonia and catatonia-related conditions of intractable compulsions, tics, and self-injury in people with autism. We assess the incidence of catatonic symptoms in autism, examine emerging ECT indications in people with autism and related developmental disorders, and encourage ethical debate and legal-administrative action to assure equal access to ECT for people with autism.

Non-suicidal self-injury and electroconvulsive therapy: Outcomes in adolescent and young adult populations.

BACKGROUND: Electroconvulsive (ECT) therapy is a highly effective treatment for severe depression. Although the clear majority of patients respond to ECT, not all do, and we still lack good predictors for ECT outcome, especially in adolescents and young adults. One clinical variable that has been associated with reduced likelihood of ECT antidepressant response in adults is comorbid borderline personality disorder. As self-injurious behavior is often a feature of borderline personality disorder, we hypothesized that adolescent and young adult patients with a history of non-suicidal self-injury (NSSI), who were being treated for major depression with ECT, would have a poorer response than patients without such a history. METHODS: We conducted a retrospective chart review of 48 patients treated with ECT for depression at The Johns Hopkins Hospital between the ages of 14 and 25. RESULTS: Initial analyses showed that the presence of NSSI was not associated with ECT outcomes. However, sub-group analyses suggested that it was associated with response to ECT and overall remission among female patients. Specifically, the results suggested that in adolescent and young adult female ECT patients, the presence of NSSI was associated with lower odds of response (OR: 0.04; 95% CI: 0.0004, 0.81, p = 0.03) and remission (OR: 0.09; 95% CI: 0.0000, 0.81, p = 0.03), and a greater mean number of treatments (5.83; 95% CI: 0.27, 11.39, p = 0.04) compared with patients without NSSI. CONCLUSIONS: Clearly, the finding that NSSI may be associated with poorer ECT outcomes among female patients needs to be replicated. Nonetheless, our data suggest caution when considering an adolescent or young adult woman for a course of ECT.

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).

Psychopharmacology and applied behavioral analysis: tandem treatment of severe problem behaviors in intellectual disability and a case series.

Many individuals with intellectual disability will at some time in their lives engage in problem behaviors that may place them and others at risk, and reduce their opportunities for healthy psychosocial functioning. These behaviors may reach severe proportions in both intensity and frequency, necessitating intervention. Both psychiatrists and behaviorists are often approached regarding negative behaviors in intellectual disability, and each discipline offers key tools in behavioral assessment and resolution. We believe that the coordinated effort of these two disciplines affords the most comprehensive and efficacious method of assessing, understanding and treating a wide range of problem behaviors and associated psychiatric pathology in individuals with various forms of intellectual disability. This paper briefly reviews the background of problem behaviors in intellectual disability and treatment of such disturbances through separate psychiatric and applied behavioral modalities, followed by the proposed coordinated neurobehavioral model. A case series ensues, describing the successful application of the neurobehavioral model to the severe problem behaviors demonstrated by three individuals with intellectual disability related to autism, Cornelia de Lange syndrome and traumatic brain injury.

Autism plus psychosis: a 'one-two punch' risk for tragic violence?

In the recent series of mass murders in Connecticut, Colorado, Norway and elsewhere, a pattern appears to emerge: young men whose social isolation borders on autism apparently become prey to psychotic ideation, and under its influence commit horrific violence. We argue that in some of these tragic cases two concomitant diagnoses may be at play, namely autism and psychosis. Autism itself is not an intrinsically violent disorder, and individuals with autism spectrum disorder (ASD) are no more prone to violent behaviors than the general population. The additional presence of psychotic illness, however, may dramatically change the picture. Based on an examination of historical and contemporary data regarding psychosis and violence in patients without developmental disability we explore three points relevant to the possibility of violence in individuals with comorbid ASD and psychosis. (1) Individuals with ASD have an elevated risk of comorbid psychopathology, including psychosis, which is strongly associated with violence. (2) The content of psychotic ideation has become increasingly violent and lethal in recent decades. (3) It is possible that individuals with ASD are readier than others to act on psychotic impulses. We conclude that there may be a kind of one-two 'vulnerability punch,' giving individuals with ASD a baseline higher risk of comorbid psychiatric illness, not infrequently including psychosis. Recognizing the increased susceptibility of individuals with autism other neurodevelopmental disability to concomitant psychotic illness increases the possibility that they can be correctly identified and treated, mitigating tragic outcomes.

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.

Treatment of cerebellar cognitive affective syndrome with aripiprazole.

This report describes a patient who developed agitation, disorientation, visual hallucinations, inappropriate verbal outbursts, and impaired memory following resection of a choroid plexus papilloma. No medical, neurologic, or metabolic disorders unrelated to the surgery were identified. Five weeks following surgery, treatment with aripiprazole, a partial dopamine agonist, was started to address the delirious state. Improvements in agitation, orientation, memory, and executive functions, as well as a decrease in emotional lability, began within twenty-four hours and continued over the remainder of the inpatient hospitalization. Five months after initial resection, aripiprazole was discontinued without worsening of cognitive or emotional functions. Persistent difficulties with working memory, planning, judgment, and visuospatial skills were noted on neuropsychological examination six months following tumor removal. This case illustrated the therapeutic benefit of aripiprazole for treatment of mental status changes associated with resection of a posterior fossa tumor.

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.

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.