Lorazepam, fluoxetine and packing therapy in an adolescent with pervasive developmental disorder and catatonia

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Abstract

Packing therapy is an adjunct symptomatic treatment used for autism and/or catatonia. Here, we report the case of a 15-year-old boy with pervasive developmental disorder who developed catatonia. At admission, catatonic symptoms were severe and the patient required a feeding tube. Lorazepam up to 15 mg/day moderately improved the catatonic symptoms. On day 36 we added fluoxetine and on day 62 we added packing therapy (twice per week, 10 sessions). After three packing sessions, the patient showed a significant clinical improvement (P < 0.001). At discharge (day 96), he was able to return to his special education program. Although we do not consider packing as a psychodynamic treatment, this case challenges the concept of embodied self that has opened new perspectives on a dialogue between psychoanalysis and neuroscience. Indeed, better body representation following packing sessions, as shown in patient’s drawing, paralleled clinical improvement, and supports the concept of embodied self. This concept may serve as a link between psychoanalysis and attachment theory, developmental psychology with the early description of “sense of self”, and cognitive neurosciences that more and more support the concept of embodied cognition. Further clinical studies are necessary to clarify the efficacy and underlying mechanism of packing treatment and to understand how patient’s experience may illustrate the concept of embodied self.

Introduction

The concept of embodied self has open new perspectives on a dialogue between psychoanalysis and neurosciences (Fonagy and Target, 2007, Gallese, 2007). In this report, we aimed to discuss how packing therapy that was given to an adolescent with autism and catatonia, and the clinical response over time, illustrate the concept of embodied self. Before summarizing and discussing the case report, a brief overview on packing therapy, on sensory integration and catatonia is warranted.

Packing therapy is based on multisensory (tactile, cenesthesic and proprioceptive) stimulations. Ross et al. (1988) conducted a national survey which demonstrated that it was rarely used in modern American psychiatry. By reviewing its use in 46 hospitalized psychiatric patients, they concluded that the treatment was safe and had interesting and useful effects that go beyond the concept of simple restraint.

The overall treatment encompasses a series of two sessions per week over a minimum one-month period. Usually, each session lasts one hour; however, the session time can be expanded to two hours depending on the patient’s response. Sessions are conducted under the supervision of a psychomotricien1 and at least two members of the patient’s care team are present (Cohen et al., 2009, Delion, 2007). First, the patient is wrapped in damp sheets (cold phase). Then, the patient is covered up with a rescue cover and a dry blanket, and the body spontaneously warms up (warm phase). The head of the patient remains free from the wrapping, which allows for communication through visual and auditory channels. Cardiac and respiratory frequencies and blood pressure are monitored before and after the session to detect adverse cardio-vascular effects and/or adverse autonomous reactions. At the end of the session, the patient is asked to draw or model with clay in order to provide non-verbal avenues to express feelings and explore body representations. Throughout the session, the patient’s comments and clinicians’ relevant observations (e.g. clinical signs, body image, and cenesthesic sensations) are carefully recorded by one of the participants (Cohen et al., 2009). Packing is used as an adjunct treatment in two main indications: catatonia (Cohen et al., 2009) and behavioral disturbances occurring in autism or mental retardation (Lobry et al., submitted for publication; Goeb et al., 2009). Recent controversies have emerged based on: (i) the absence of an evidence-based study to support the treatment; (ii) the possible absence of free consent in individuals with poor communication skills; and (iii) erroneous association of the theoretical background of packing with psychoanalysis, despite the fact patient’s experience of packing may contribute to the psychodynamic metapsychology of the self (Delion, 2007). This final point is crucial, as there is strong disagreement between parents associations and psychodynamic theory in the field of autism in France (Chamak and Cohen, 2003) and abroad (Rhode, 2008).

In our view packing therapy is better understood as a sensory-integration approach as described by Ayres (2005) or Bullinger (Kloeckner et al., 2009). Sensory integration is the hierarchical organization of the somatic sensations that serve as foundations for the individual’s perceptions, behaviors and learning. The greatest potential for the development of sensory integration occurs within an adaptation response, which is a purposeful, goal-directed response to a sensory experience. Auditory, vestibular, proprioceptive, tactile and visual senses are progressively integrated as a body percept, and are rooted in different psychosomatic functions such as the coordination of the two sides of the body, motor planning, activity level, attention span and emotional stability. Sensory integration dysfunction (SID) results in a wide variety of developmental disorders (Bundy, 2005, Bundy et al., 2007). Considering the poor sensory processing observed in Autism Spectrum Disorders, SID is viewed as a core deficit on which treatment interventions should be focused (Bauman, 2005, Greespan et al., 2008, Kloeckner et al., 2009). Individuals with autism who can express themselves have also reported the importance of sensory processing (Chamak et al., 2008). In addition to learning disorders and severe developmental disorders, SID may contribute to other clinical symptoms such as catatonia without entirely explaining the cognitive dysfunction (Cohen et al., 2009).

Although infrequent in adolescence, catatonia is a severe condition; several deaths have been reported (Ainsworth, 1987; Dimitri et al., 2006). This neuropsychiatric condition severely impedes the patient’s functioning. Catatonic patients face huge impairments in everyday life: waking up, washing themselves, getting dressed, eating, and attempting any activity (Cornic et al., in press). Catatonia occurs in various psychiatric disorders, neurological diseases, intoxications and metabolic conditions (Takaoka and Takata, 2003, Cornic et al., 2007, Lahutte et al., 2008). Its phenomenology encompasses motor (e.g., posturing, catalepsy, waxy flexibility), behavioral (e.g., negativism, mutism, automatic compulsive movements), affective (e.g., involuntary and uncontrollable emotional reactions, affective latency, flat affect, withdrawal), and regressive symptoms (e.g., verbigeration, enuresis and encopresis, echophenomena) (Northoff et al., 1999). Catatonia can occur in young people with history of pervasive developmental disorders (PDD). In these cases, morbidity is often extremely severe and treatment is difficult (Billstedt et al., 2005, Ohta et al., 2006, Wing and Shah, 2000, Kakooza-Mwesige et al., 2008). The recommended treatments are symptomatic and include the use of sedative drugs (e.g., high doses of benzodiazepines) and electroconvulsive therapy (ECT); these treatments offer dramatic and rapid improvement in many cases (Taylor and Fink, 2003, Caroff et al., 2004, Wachtel et al., 2008). But cases with poor improvement have been reported as well (Consoli et al., 2009). When available, treatment of any associated medical condition is required as well (Cornic et al., 2007, Marra et al., 2008).

Catatonia can be viewed as an acute and severe sensory integration dysfunction (SID) state associated with body map disorganization, absence of integration of proprioceptive sensations and failure in motor planning that can explain motor symptoms; in addition, affective symptoms can be viewed as the ultimate product of a failure of sensory integration processes (Cohen et al., 2009). Considering this, we developed a packing therapy for treatment of catatonia that may be used when patients do not respond to high doses of benzodiazepines or when family members are reluctant to accept ECT. In a prospective study on youth catatonia from 1993 to 2007 (Cohen et al., 2005, Cornic et al., 2009), among the 44 patients recruited in a university setting, six adolescents (five males and one female) experienced packing during their stay, including two patients with PDD. Packing therapy appeared to be an effective adjunct treatment in four patients who also received psychotropic medications; overall tolerance and compliance were good (Cohen et al., 2009). However, we failed to find similar case descriptions in the literature.

Here, we report the case of a 15-year-old adolescent (named John) with PDD who developed catatonia by early adolescence and showed a dramatic improvement after adjunction of fluoxetine, packing and lorazepam. This case is noteworthy given (i) the severity of the patient’s symptoms at admission, such as the patient need for tube feeding, (ii) the careful monitoring of catatonic symptoms, (iii) the parents consent to record packing sessions on video before and after improvement (available on website2); and (iv) the dramatic improvement of John’s body representation – as evidenced in drawings – that paralleled clinical improvement.

Section snippets

Family history

There is a family history of bipolar disorder in John’s maternal grandfather, who received ECT and mood stabilizers and recovered. John’s paternal grandmother showed symptoms of psychiatric disorders, but diagnosis was not available.

Early developmental and clinical history

John was the second child of unrelated parents. At birth, John weighed 3.47 kg and was 51 cm long with a 36 cm head circumference. His early development was unremarkable. He walked at 16 months of age, and spoke his first words at 15 months and his first sentences

Therapeutic approach

Therapeutic approaches for catatonia are mainly symptomatic. It is recommended to use high dosage of benzodiazepines, and to perform electroconvulsive therapy (ECT) in cases of resistance or life-threatening condition (Taylor and Fink, 2003, Wachtel et al., 2008). In the case of patient John, benzodiazepines were only moderately efficient despite high doses (lorazepam up to 15 mg/day in a 40 kg-subject), and ECT was not considered because the parents refused this option after two months of

Conclusion

We conclude that, in the case of PDD and catatonia, the sensory-integration approach of packing therapy is a possible and effective adjunct treatment in cases of resistance to high dosage benzodiazepine and as an alternative to ECT. Consent for packing therapy must be collected from patients and parents. Further clinical studies are necessary to clarify the efficacy and underlying mechanism of packing treatment and to understand how patient’s experience may illustrate the concept of embodied

Acknowledgements

The authors thank John’s parents for their implication in the treatment, for trusting us during the prolonged inpatient treatment, and for allowing the publication of John’s case with videos. The authors also thank Bruno Falissard, MD, PhD for his advice regarding statistics.

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