Onset of Catatonia at Puberty Electroconvulsive Therapy Response in Two Autistic Adolescents Neera Ghaziuddin, MD, Daniel Gih, MD, Virginia Barbosa, MD, Daniel F. Maixner, MD, and Mohammad Ghaziuddin, MD Abstract: Catatonia is a syndrome of motor and behavioral dis- turbance. It is a poorly understood condition, which is underrecognized and may go untreated despite intensive medical workup and numerous unsuccessful medication trials. However, with treatments known to be effective, such as benzodiazepines and/or electroconvulsive therapy, patients may return to their baseline functioning. Autism and catatonia have been previously reported together. We report 2 patients with autism and mental retardation who developed catatonic symptoms at the onset of puberty. Both patients experienced persistent symptoms over several years and presented with a history of motor disturbance, functional decline, and episodic aggression. Both patients were treated with electroconvulsive therapy resulting in a positive response and functional improvement. Catatonia may persist as a chronic condition, lasting over several months or years, if not recognized and treated. Key Words: catatonia, autism, ECT (J ECT 2010;26: 274Y277) C atatonia is a poorly understood but debilitating condition characterized by motor, behavioral, and autonomic symp- toms. It is described in the Diagnostic and Statistical Manual of Mental Disorders, Fourth Edition (DSM-IV ) in association with 3 different diagnoses: general medical conditions, schizo- phrenia, and mood disorders. Under each category, catatonia is described primarily as a psychomotor disturbance that may in- clude increased motor disturbance (usually purposeless) and/or reduced motor activity (immobility, negativism, waxy flexibility, stupor in extreme cases), disturbance of voluntary movements (bizarre or inappropriate postures, repetitive or stereotyped move- ments, prominent grimacing), and echophenomena (mimicking the movements or words of other people). Potential consequences include exhaustion, self-inflicted injury, dehydration, malnutri- tion, and, occasionally, death. Usually associated with affective or psychotic symptoms, catatonia is more common than generally believed. A recent cross- cultural study using standardized criteria found a frequency of 13.6% in India and 9.6% in Wales among psychiatrically hos- pitalized adults. 1 Overall, there is increased awareness of this disorder, the associated risks, and importance of using established treatments. 2 Catatonia can also occur in the setting of autism spectrum disorders. Wing and Shah 3 reported the first systematic study of catatonic symptoms in a larger sample of 506 children with autism spectrum disorders, who were referred to a tertiary re- ferral center in the UK. 4 The authors found that 17% of the sample, ranging in age from 15 to 19 years, displayed what the authors described as Bsevere exacerbation of catatonic features.[ The diagnosis of catatonia was given if there was deterioration of movement resulting in interference with daily functioning, increased slowness affecting movements and action, difficulty in initiating and completing actions, increased reliance on physical or verbal prompting, and increased passivity and apparent lack of motivation. Other authors have pointed out the similarities between catatonia and autism spectrum disorders. 5 Symptoms common to both autism and catatonia include communication difficulties, stereotyped movements, mannerisms, and agitation. Some studies have suggested that 1 in 7 autistic patients may have catatonia and that the diagnosis should be considered in any autistic adolescent or adult presenting with significant func- tional impairment. 6 Both misdiagnosis and missed diagnosis of catatonia have been previously reported. For instance, a case was misdiagnosed for a neurological disorder until catatonia was eventually diag- nosed 15 years later. 7 That catatonia may persist over months or years as a chronic condition was previously described by Kocmur and Vodopivec 7 and Gaind et al 8 and also observed anec- dotally by clinicians who treat patients with autistic disorders. We report 2 adolescents with autism (18 and 16 years at presentation) whose symptoms started around puberty and per- sisted for 5 and 3 years, respectively. The earliest symptoms in each case were motor disturbance associated with a significant change in baseline functioning and increased aggression. Both patients were admitted to our institution for a comprehensive workup, which was followed by treatment with electroconvul- sive therapy (ECT). Fictitious identifiers were used to protect patient confidentiality in accordance with institutional review board guidelines. The MECTA device (MECTA Corporation, Tualatin, Oreg) was used for treating both patients. CASE REPORTS Case 1 LN is an 18-year-old man with diagnoses of congenital sensorineural deafness, mild mental retardation (MR), autism, and reported history of catatonia. His parents presented him for possible treatment with ECT, given the persistent loss in func- tioning despite intensive treatment for the past 5 years. The family approached our institution because LN was from a state where restrictive ECT regulations apply. Birth history was unremarkable. Profound bilateral hearing loss was noted early. LN had delayed developmental milestones, reduced interpersonal relatedness, and preference for parallel CASE REPORT 274 www.ectjournal.com Journal of ECT & Volume 26, Number 4, December 2010 From the Department of Psychiatry, University of Michigan, Ann Arbor, MI. Received for publication November 2, 2009; accepted March 9, 2010. Reprints: Neera Ghaziuddin, MD, Rachel Upjohn Bldg, 4250 Plymouth Rd, Ann Arbor, MI 48109-5734 (e-mail: neerag@umich.edu). Dr Daniel F. Maixner, MD, received funding from the Flinn Foundation. There are no other financial disclosures. Virginia Barbosa, MD, was completing a fellowship at the time of manuscript preparation. She is presently connected with the Cumberland Behavioral Health Center in Lexington, KY. Copyright * 2010 by Lippincott Williams & Wilkins DOI: 10.1097/YCT.0b013e3181de332e Copyright @ 20 Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited. 10