Everolimus and intensive behavioral therapy in an adolescent with tuberous sclerosis complex and severe behavior

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Abstract

Background: Self-injury and aggression have been reported in individuals with TSC (tuberous sclerosis complex), yet few data exist about treatment. Everolimus, an mTOR inhibitor, has been FDA-approved for subependymal giant cell astrocytomas (SEGAs) and renal angiomyolipomas in TSC. However, clinical use of everolimus with direct, real-time observations of self-injury and aggression in an individual with TSC has not been reported. Methods: During an inpatient admission to a neurobehavioral unit, real-time measurements of behaviors and seizures were recorded. An interdisciplinary team used these data to make treatment decisions and applied behavioral and pharmacological treatments, one at a time, in order to evaluate their effects. Results: Aggression and self-injury improved with applied behavioral analysis (ABA), lithium, and asenapine. Improvements in SEGA size, facial angiofibromas, seizures, and the most stable low rates of self-injury were observed during the interval of treatment with everolimus. Conclusion: Mechanism-based treatments in the setting of an evidence-based behavioral and psychopharmacological intervention program may be a model with utility for characterization and treatment of individuals with severe behavior and TSC. © 2013 The Authors.

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APA

Gipson, T. T., Jennett, H., Wachtel, L., Gregory, M., Poretti, A., & Johnston, M. V. (2013). Everolimus and intensive behavioral therapy in an adolescent with tuberous sclerosis complex and severe behavior. Epilepsy and Behavior Case Reports, 1(1), 122–125. https://doi.org/10.1016/j.ebcr.2013.06.004

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