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. 2023 Mar 1;18:42. doi: 10.1186/s13023-023-02626-4

Table 1.

PICOS criteria for inclusion and exclusion of studies

PICOS Inclusion Exclusion
Population Patients with LGS, including clearly defined “probable” LGS Other DEEs
Intervention Studies assessing the overall burden of illness of LGS without the impact of an intervention Studies assessing the impact of interventionsa
Comparator N/A
Outcomes

Prevalence/incidence

Economic burden

Direct and indirect costs

Resource use

Patient and caregiver HRQoL and broader patient-reported outcomes

Mortality

Study designs/publication type

Publications reporting original data, including:

Healthcare insurance claims data

Chart reviews

Registry studies

Electronic healthcare databases studies

Epidemiologic surveys

Observational studies

Economic evaluations if they contain original data on resource utilisation and HRQoL

In vitro and in vivo studies

Preclinical studies

Case reports and case series

Clinical studies reporting only efficacy and safety data

Economic evaluations reporting on specific interventionsb

Reviewsc, SLRsc, comments, letters, editorials and press releases

Limits Searches of conferences in Embase were limited to the previous 3 years (conferences 2018-current)d None

HRQoL, health-related quality of life; LGS, Lennox–Gastaut syndrome; N/A, non-applicable; SLR, systematic literature review

aThe focus is on the overall burden as opposed to the impact of specific interventions

bEconomic evaluations reporting on specific interventions without any original data on e.g HRQoL (utilities) and healthcare resource utilisation will be excluded, however, the reference lists will be searched to ensure all relevant studies are captured in this SLR.

cReviews and SLRs will be excluded from final inclusion but the reference lists will be searched to ensure all relevant studies are captured in this SLR.

dThe rationale for excluding conference abstracts > 3 years is that this should be sufficient time for the data to be published in peer-review journals if they are of sufficient importance and quality. The rationale for including conference abstracts ≤3 years is to include the most up-to-date data.