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 | Â |
Comparators | No restrictions | Â |
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 |