RT Journal Article
T1 Outcome measurement instruments in Rett syndrome: A systematic review
A1 Romero-Galisteo, Rita Pilar
A1 González-Sánchez, Manuel
A1 Costa, Lara
A1 Brandao, Rita
A1 Ramalhete, Catia
A1 Leao, Carla
A1 Jacobsohn, Lia
K1 Rett, Síndrome de
AB ObjectiveThe aim of the study was to identify and characterize outcome measures for objective and subjective assessment in persons with Rett syndrome (RS).MethodsA systematic review was conducted consulting the EBSCO, Cochrane, Web of Science, Scielo, MEDLINE and PsycINFO databases for published studies describing the use of patient-reported outcome measures (PROMs) and other outcome measures in persons with RS. Validation studies and observational studies were included. The PROMs were first described, and then the measurement properties were evaluated using predefined criteria according to the COnsensus-based Standards for the selection of health Measurement Instruments (COSMIN). The outcome measures were then grouped according to the International Classification of Functioning, Disability and Health (ICF) to establish a relationship between outcome measures and ICF domains.ResultsTwenty out of 2327 articles were appraised, and seventeen different outcome measures were identified and described. Ten outcome measures corresponded to evaluation questionnaires, while the remaining seven assessed functional outcomes: walking distance, physical activity level and ability to interact visually. A relation between these outcome measures that assess RS and the ICF allows understanding that most of the instruments (fifteen) include the assessment of activity limitations.ConclusionsThe findings of this study seem to be promising for their use by clinicians and researchers, although they have methodological limitations. The accuracy and quality of these individual outcome measures should continue to be assessed in an attempt to gather a consensus on the best tools used in RS.
PB Elsevier
YR 2022
FD 2022-07
LK https://hdl.handle.net/10630/39574
UL https://hdl.handle.net/10630/39574
LA eng
NO R.P. Romero-Galisteo, M. González-Sánchez, L. Costa, R. Brandão, C. Ramalhete, C. Leão, L. Jacobsohn, Outcome measurement instruments in Rett syndrome: A systematic review, European Journal of Paediatric Neurology, Volume 39, 2022, Pages 79-87, ISSN 1090-3798, https://doi.org/10.1016/j.ejpn.2022.06.003.
DS RIUMA. Repositorio Institucional de la Universidad de Málaga
RD 23 ene 2026