Severe psychomotor retardationdeveloped over the last year in a 35-year-old man who was living in a residential treatment facility. His diagnoses included atypical autism, moderate mental retardation, and seizure disorder. He stood motionless for hours, often in odd postures. His speech had decreased considerably, and he required assistance in daily living activities. The psychiatric examination did not reveal hallucinations or delusions. His medications included an antiepileptic agent for long-standing grand mal seizures and an atypical antipsychotic for a tentative diagnosis of psychosis not otherwise specified. Trials of antipsychotic medications have had no effect on his motor symptoms.
At this point, catatonia was diagnosed and a trial of lorazepam(Drug information on lorazepam) was started with the dosage titrated over the course of 2 weeks to 4 mg twice a day. The hospital administrators expressed concern that the patient might become dependent on benzodiazepines, but the psychiatrist assured them that lorazepam at the prescribed high dose is an accepted treatment for catatonia. The patient responded well to treatment, with decreased psychomotor slowness, fewer freezing episodes, and increased socialization. Three months after the start of lorazepam, catatonia had resolved. The patient functioned at baseline level again, and he had resumed work at a sheltered workshop. His medications include an antipsychotic, an antiepileptic, and lorazepam (4 mg twice a day). Lorazepam will be tapered and stopped if the patient continues to do well over the next 3 months.
Novel psychological treatment
A new addition to the anticatatonic armamentarium is the psychological method developed by Shah and Wing.15 In brief, the treatment involves keeping the person active, doing what he or she enjoys, using verbal or gentle physical prompts to overcome movement difficulties, and maintaining a predictable structure and routine for each day. The importance of educating caregivers to understand catatonic behavior and to realize that it is not under the control of the patient is paramount. Management techniques for specific issues such as incontinence, freezing in postures, eating problems, and episodes of excitement are specified. This approach can be used in conjunction with medical treatments or when medical treatments fail.
Case 3: Pediatric ECT
A 9-year-old boy with apparently normal development but high familial loading of psychosis was admitted for agitation, anxiety, perplexity, negativism, and psychomotor slowing. Stupor, mutism, fever, and facial flushing developed over the next 2 weeks. Results of extensive medical and psychiatric workups were negative. EEG recordings consistently showed overall slowing but no epileptic spikes. The symptoms satisfied criteria for CDD, which is characterized by massive regression after the age of 2 years but before the age 10 of years, followed by autistic symptoms. In the absence of recommended treatments for CDD, other medical and psychiatric diagnoses-- including encephalitis and malignant catatonia--were considered. After failed trials with anticonvulsants, antiviral medications, and high-dose benzodiazepines, expert opinions regarding an ECT trial were sought; ECT consent was obtained from the caregiver. Stupor, muteness, and refusal to eat and drink improved rapidly during the first course of 7 treatments, but agitation, stereotypies, repetitive speech, and poor level of function remained.
Despite the widely different developmental and recent history, the symptoms of this patient were similar to those of other children with autism and conformed tothe differential diagnosis of CDD. A second ECT course was given because the patient relapsed into stupor and immobility. Again, the most severe catatonic symptoms dissipated, but the patient remained impaired. The patient was discharged and continued with outpatient ECT (once every week or biweekly) for the next 5 months. During that time, the boy slowly returned to baseline function and now attends school and lives at home. There have been no relapses during the past 3 years.
Are autistic and catatonic regression related?
This case illustrates the importance of diagnosing catatonia amidst severe regression of unknown cause in prepubertal children in order to select effective therapies. CDD is considered rare, with a pooled estimate across 4 surveys of 1.7 per 100,000 subjects (95% confidence interval, 0.6-3.8 per 100,000).20 However, it is also likely that an unknown number of cases are not reported because neurologists and other pediatric specialists who are not familiar with the psychiatric classification of CDD label this condition differently (eg, as encephalitis). Textbooks describe CDD as sometimes being associated with known medical conditions, but usually no clear cause is found. Deterioration occurs over the course of weeks or months. Residual symptoms include impaired social interaction, restricted language output, and repetitive behaviors. Follow-up studies have suggested that older age at onset of autistic symptoms, as in CDD, may be associated with worse outcome.21 There is no recommended treatment.
Catatonia has also been reported infrequently in children,22,23 although no systematic studies in this age group have been done. The oldest description of catatonic symptoms (in a 3-year-old child) comes from de Sanctis (1908-1909).24 Lorazepam and ECT were reported to be effective in 2 more recently published reports.22,23
CDD and childhood catatonia are both poorly studied and probably poorly recognized. Catatonia should be studied systematically in children, adolescents, and young adults with psychiatric, neurologic, and developmental disorders--especially autism and Prader-Willi syndrome.25,26 The symptom overlap between autistic regression in CDD and catatonia should be further assessed, and the presence of catatonic symptoms should be assessed during regression in children with autism.27 Most important, catatonia in children seems to respond to the same treatments as in adults.