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Clinical Trial Outcome Measurements in Lennox-Gastaut Syndrome and Their Application to the Development of Targeted Treatments

  • Pediatric Neurology (A Yeshokumar, Section Editor)
  • Published:
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Abstract

Purpose of Review

Lennox-Gastaut syndrome (LGS) is an epileptic encephalopathy that is challenging to treat, with >80% of patients remaining refractory to therapy. Patient-oriented clinical outcome measures are essential for treatment guidance and the development of new therapies. Here, we provide a comprehensive review of outcome measures used in clinical trials targeting patients with LGS, in order to inform future studies, develop, and implement targeted patient and caregiver-relevant outcome measures.

Recent Findings

Published clinical trials that include, or specifically target, individuals with LGS are reviewed, and primary and secondary outcome measurements are examined. Outcome measures are grouped and discussed as (1) seizure-related, (2) cognitive, (3) functional, (4) quality of life, and (5) psychosocial for both the LGS patient and caregiver. In patients with LGS, seizure frequency is the most common outcome measured by clinicians, while quality of life and psychosocial effect of treatments are often most important to families.

Summary

Therefore, a disconnect exists between outcome measurements used in clinical trials and those rated important by families. Furthermore, cognitive, functional, and psychosocial outcome measures are often insufficiently sensitive for many LGS patients, and thus, using better targeted measures may result in an increased availability of treatment options.

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Papers of particular interest, published recently, have been highlighted as:

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      Acknowledgements

      We would like to acknowledge Dr. Tracy Dixon- Salazar and the LGS Foundation for their inspiration and support of this review.

      Funding

      Dr. Gofshteyn is supported by the NIH-NINDS K12 Award Number NS066274-10.

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      Authors and Affiliations

      1. Charles E. Schmidt College of Medicine at, Florida Atlantic University, Boca Raton, FL, USA

        Victoria Danan BA

      2. Division of Child Neurology, Nationwide Children’s Hospital and The Ohio State University College of Medicine, Columbus, OH, USA

        Patel Anup MD & Vidaurre Jorge MD

      3. Division of Pediatric Neurology, Weill Cornell Medicine, New York City, NY, USA

        Beal Jules MD, Kerry Gao BS & Jacqueline S Gofshteyn MD

      4. Department of Neurology, UT Health Austin Pediatric Neurosciences at Dell Children’s, University of Texas at Austin, Austin, TX, USA

        Freedman Daniel DO

      5. Department of Child Neurology, The Medical University of South Carolina, Charleston, SC, USA

        Bhatia Sonal MD

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      Danan, V., Anup, P., Jules, B. et al. Clinical Trial Outcome Measurements in Lennox-Gastaut Syndrome and Their Application to the Development of Targeted Treatments. Curr Treat Options Neurol 23, 30 (2021). https://doi.org/10.1007/s11940-021-00687-z

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