ICNC2018 Abstracts & Symposia Proposals, ICNC 2014

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Everolimus for subependymal giant cell astrocytoma (SEGA) associated with tuberous sclerosis complex (TSC): EXIST-1 long-term efficacy and safety results
David Neal Franz, Elena Belousova, Steven Sparagana, E. Martina Bebin, Michael Frost, Rachel Kuperman, Olaf Witt, Michael H. Kohrman, J. Robert Flamini, Joyce Y. Wu, Paolo Curatolo, Petrus J. de Vries, Elizabeth A. Thiele, Noah Berkowitz, Oezlem Anak, Julie Niolat, Sergiusz Jozwiak

Building: Bourbon Cataratas Convention Centre, Foz do Iguaçu
Room: Iguazu II
Date: 2014-05-08 02:00 PM – 02:15 PM
Last modified: 2014-02-09

Abstract


Introduction

EXIST-1 (NCT00789828), a randomized, double-blind, phase 3 trial, demonstrated that everolimus, an mTOR inhibitor, was superior to placebo for reducing SEGA volume in patients with TSC. Long-term safety and efficacy of everolimus were examined using data available 11-January-2013.

Methods

Patients (median age, 9.5years [range, 0.8-26.6years]) received 4.5mg/m2/day oral everolimus (n=78; titrated to a target trough 5-15ng/mL) or placebo (n=39). SEGA response rate (primary endpoint) was defined as proportion of patients with ≥50% reduction in sum of volumes of all target SEGA (≥1 SEGA ≥1 cm in longest diameter) versus baseline. Adverse events (AEs) were monitored at every visit.

Results

Everolimus was superior to placebo for SEGA response rate (34.6% vs 0.0% P˂0.0001; original cut-off, 02-March-2011). Following positive results for original cut-off, placebo patients were offered open-label everolimus in the study’s extension phase. As of 11-January-2013, 111 patients received ≥1 dose of everolimus and were included in this extension analysis. Median treatment duration of everolimus was 29.3months and SEGA response rate was 48.6% (95% confidence interval [CI], 39.0-58.3%). As of 11-January-2013, median time to SEGA progression was not reached; however, SEGA progressions were observed for 9 (8.1%) patients. Most AEs continued to be grade 1 or 2. The most frequent serious adverse events occurring in more than 3% of patients were pneumonia (10.8%), pyrexia (4.5%), gastroenteritis (3.6%), and convulsion (3.6%).

Conclusion

Everolimus continued to reduce SEGA volume with no new safety concerns.


Keywords


tuberous sclerosis complex; subependymal giant cell astrocytoma; everolimus; mTOR inhibition

References



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