Burden of Illness of Lennox–gastaut Syndrome: A Systematic Literature Review
Abstract number :
3.41
Submission category :
16. Epidemiology
Year :
2022
Submission ID :
2205017
Source :
www.aesnet.org
Presentation date :
12/5/2022 12:00:00 PM
Published date :
Nov 22, 2022, 05:27 AM
Authors :
Arturo Benitez, MD, MBA – Takeda Development Center Americas, Inc, Cambridge, MA, USA; Jeannine Roth, M.Sc. Pharm – Takeda Pharmaceuticals International AG, Zurich, Switzerland; J. Scott Andrews, PharmD – Takeda Development Center Americas, Inc, Cambridge, MA, USA; Drishti Shah, PhD – Takeda Development Center Americas, Inc, Cambridge, MA, USA; Emma Butcher, PhD – Oxford PharmaGenesis Ltd, Oxford, UK; J Helen Cross, MB ChB, PhD – UCL Great Ormond Street Institute of Child Health, London, UK; Joseph Sullivan, MD – Department of Neurology University of California, San Francisco, CA, USA
Rationale: Lennox-Gastaut syndrome (LGS) presents a significant challenge to individuals and caregivers. Individual studies have explored the burden of LGS; however, to our knowledge no published reviews have collated information on the clinical, treatment, humanistic, and economic burden of LGS or the impact of the COVID-19 pandemic. This systematic literature review aimed to evaluate the burden of LGS and identify evidence gaps to guide further research.
Methods: Literature searches were performed in MEDLINE, Embase, and the Cochrane Library from inception to March 15, 2022. Supplementary searches included conference proceedings (2017–2022), websites, and review reference lists. Outcomes included clinical symptom burden, treatment burden, comorbidities, care requirements, quality of life, economic burden, healthcare resource use (HCRU), caregiver burden, and impact of the COVID-19 pandemic. Non-interventional studies reporting original primary or subgroup analyses for individuals with LGS were included. Animal studies, case reports, reviews, interventional studies, and non-English articles without abstracts were excluded. Both citations and full texts were screened in duplicate. Included data were summarised narratively. The protocol is registered on PROSPERO (CRD42022317413).
Results: After screening 1,442 de-duplicated records, 112 records were included. Supplementary searches identified 6 further records for inclusion. Clinical symptom burden and treatment burden were frequently reported (in 88 and 61 records, respectively). Fewer records reported on care requirements (n = 16), economic burden (n = 13), comorbidities (n = 8), quality of life (n = 7), or caregiver burden (n = 5). No articles reported on COVID-19 burden. Included records confirmed the high seizure burden experienced by individuals with LGS and reported a range of non-seizure symptoms (e.g., communication problems and physical impairment). In terms of treatment burden, most individuals receive several antiseizure medications (ASMs) to manage their condition; however, these ASMs are associated with adverse events that may lead to discontinuation. The high clinical burden of LGS translates to high costs, owing to HCRU, care requirements, and medication. Evidence on comorbidities, quality of life, and caregiver burden was sparse. Data extraction for additional data points will be completed before the meeting.
Conclusions: LGS is associated with a high burden on individuals and healthcare systems. Economic burden is high owing to healthcare and medication needs, with individuals experiencing a range of clinical symptoms that require management with polytherapy. Novel treatments are needed to address the complex symptoms of LGS, and further research is required to fully understand the burden of LGS, particularly comorbidities, quality of life, caregiver burden, and COVID-19 burden.
Funding: Study funded by Takeda Pharmaceutical Company Limited. Aimee Jones (Oxford PharmaGenesis, Oxford, UK) was involved in designing the protocol, running the searches and abstract screening for the systematic literature review.
Epidemiology