Table 1 Summary of the eligibility criteria for both SLRs
Criterion | Inclusion criteria | Exclusion criteria |
|---|---|---|
Population | Studies of patients with mitochondrial disease with associated seizures were eligible. Studies reporting data for caregivers of these patients were also eligible Specifically, patients with the following mitochondrial diseases were eligible for inclusion in the SLR: Leigh’s syndrome POLG1 mutation related disorders: AHS MCHS MEMSA SCAE PEO MIRAS SANDO arPEO (includingC10orf2/Twinkle) adPEO MELAS MERRF RARS2 mutation related disorders: Pontocerebellar hypoplasia type 6 PDHC deficiency Studies with mixed populations (> 75% included in the list above) were also eligible In the original SLRs, the terminology used to describe the population was ‘mitochondrial disease with epilepsy’; however, for the update SLRs this was changed to ‘mitochondrial disease with associated seizures’. Although there was a change in terminology, the eligible patient population and study selection were consistent | Animal/in vitro studies Patients with other conditions Patients with mitochondrial diseases who did not have epilepsy |
Interventions | No restrictions | |
Comparators | No restrictions | |
Outcomes | HRQoL and utilities SLR Studies reporting data from HRQoL tools were eligible for inclusion, including: Adult's Attitudes to Children with Epilepsy: Visual Analogue Scale DISABKIDS (Epilepsy Module) ELDQOL ECQ EFA GEOS-YP HARCES CHEQOL-25 Modified Impact of Epilepsy Schedule ICI IPES ICND NeuroQol Newcastle Mitochondrial Disease Questionnaire PedsQL PedsQL Epilepsy Module PESQ PROMIS Paediatric/Parent Proxy Profile: PROMIS Paediatric/Parent Proxy Profile 25 PROMIS Paediatric/Parent Proxy Profile 37 PROMIS Paediatric/Parent Proxy Profile 49 QOLCE: QOLCE 16 QOLCE 55 G-QOLCE QOLIE: QOLIE 31 QOLIE 89 QOLIE-AD-48 QOLPES SF-36 Studies reporting data from the following health state utility tools were eligible for inclusion: AQoL-6D for adolescents AQoL for adults: AQoL-4D AQoL-6D AQoL-8D CHU9D EQ-5D EQ-5D-Y: EQ-5D-Y Proxy Version 1 EQ-5D-Y Proxy Version 2 EQ-5D-Y Interviewer Administered Proxy Version 1 EQ-5D-Y Interviewer Administered Proxy Version 2 HUI HUI2 HUI3 SF-6D 15D 16D 17D AHUM HSCS-PS QWB Costs and healthcare resource use SLR Studies reporting data on the following types of costs and healthcare resource utilisation were eligible for inclusion: Direct monetary costs associated with mitochondrial disease with associated seizures specifically: Direct medical costs Direct non-medical costs Indirect monetary costs, including on caregivers, specifically: Impact on work or education Days lost, e.g. work or education Resource use: Any non-monetary resource use data | Economic evaluation outcomes, e.g. QALYs / ICERs, were ineligible |
Study designs and categories of articles | No restrictions on study design providing eligible outcomes were reported | Categories of articles excluded: Opinion pieces Letters Editorials Systematic reviews Systematic reviews published since 2016 were used for reference checking only Studies published as abstracts or conference presentations were not eligible for inclusion |
Limits | Limit to English language only | Non-English language articles |