Treatment of catatonia with electroconvulsive therapy in adolescents.

Catatonia is a cluster of abnormalities associated with mood disorders (Singerman and Raheja 1994), schizophrenia, autism (Wing and Shah 2006) and other neurologic, psychiatric, and general medical conditions. Its unique presentation can be clearly defined as syndrome in its own right (Taylor and Fink 2003). The Diagnostic and Statistical Manual of Mental Disorders, Fourth Edition (APA 1994), lists five clinical features of catatonia: motoric immobility; excessive motor activity; extreme negativism or mutism; peculiarities of voluntary movement; and echolalia or echopraxia. The symptoms of catatonia fall under three categories: retarded, excited, and malignant. The retarded form presents itself with symptoms such as stupor and repetitive movements, the excited form exhibits agitation and delirium (Fink 2009), and the malignant form is marked by autonomic instability, including hypertension, elevated heart rate, and rise in temperature, which is often fatal (Singerman and Raheja 1994; Fink 2009). Overlapping symptoms between neuroleptic malignant syndrome (NMS) and malignant catatonia, and the fact that both respond to electroconvulsive therapy (ECT), has raised questions as to whether there is a relationship between these disorders (Lee 2000; Trollor and Sachdev 1999). One of the difficulties in defining catatonia is that the term has been used to describe an acute and malignant condition in patients with multiple diagnoses, a more chronic condition, and sometimes as a manifestation of a negative outcome of a particular diagnosis. For example Wing and Shah (2006) studied catatonia in children with autism and found symptoms that included: a) increased slowness affecting movement and verbal responses; b) difficulty in initiating and completing actions; c) increased reliance on physical or verbal prompting by others; and d) increased passivity and apparent lack of motivation (Wing and Shah 2000). Since the review by Pataki in 1992, catatonia has been recognized as a condition in its own right with an acute presentation with motor symptoms, delirium, and dysregulation of multiple fundamental life support systems (Pataki et al. 1992). There were 43 reports of pediatric catatonia in the world literature in 1991, and diagnoses in these cases include multiple psychiatric syndromes including mood disorders(Pataki et al. 1992), schizophrenia, and autism (Kinrys and Logan 2001; Cohen et al. 1999). In the adult literature, we were able to identify case reports of catatonia in patients with lupus (Ditmore et al. 1992), hyperthyroidism (Farah and McCall 1995), and during psychiatric illness during pregnancy (Ferrill et al. 1992). It is therefore possible that catatonia may occur during psychiatric complications of other central nervous system conditions. Catatonia occurs in adolescents but its prevalence is unknown, given that it is rare and may often go unrecognized. One group of researchers from France estimated catatonia to occur at an incidence rate of 0.16 per million per year in the general adolescent population and in 0.6% of the inpatient adolescent population (Cohen et al. 1999). However, another study from India found at least two signs of catatonia in 5.5% of their outpatient population (Thakur et al. 2003). This range in prevalence reflects the confusion in the early literature between catatonia as a malignant and life threatening episode, and catatonia as an ongoing form of schizophenia or a chronic condition. The Bush-Francis Catatonia Rating Scale (BFCSR) was first developed in 1996 and can be used to evaluate the severity of its presentation (Fink 2009). The BFCRS 23 item scale is scored from 0 to 3 for each item. In order to facilitate screening for the disorder a shorter version, the Bush-Francis Catatonia Screening Instrument (BFCSI) (14 items) was also developed (Bush et al. 1996). Although the authors suggest that one to four items suggests a positive diagnosis, it is not possible to generate a cut-off score psychometrically given the rarity of the condition and lack of a diagnostic gold standard. The full 23-item BFCRS can be obtained by clinicians for use in practice in the original article (Bush et al. 1996). The BFCRS was later revised (BFCRS-R), shortened to 20 items, and psychometrically tested using Rasch analysis (Wong et al. 2007). This study found catatonia to be a unidimensional diagnosis, again pointing to the validity of catatonia as a diagnosis in its own right. The authors note that because of the rarity of the condition, any rating scale would be insufficient to define a cut-off score for catatonia in a specific population. ECT can be a life-saving and relatively safe treatment for catatonia in adults (Kelly and Zisselman 2000), and a number of individual case studies and chart review studies have reported the successful treatment of catatonia with ECT in adolescents beginning in 1999 (Zaw et al. 1999), with individual and case series of similar results following since that time (Singerman and Raheja 1994; Wing and Shah 2006). This is true for catatonia associated with schizophrenia (Baeza et al. 2004), mood disorders (Singerman and Raheja 1994; Pataki et al.1992) and a wide range of other conditions, including epilepsy and encephalitis (Cohen et al. 1999). Cohen et al. (1999) reviewed all English and French reports on