2026-05-30T07:32:16Zhttps://riuma.uma.es/rest/oai/requestoai:riuma.uma.es:10630/395742026-02-03T11:01:54Zcom_10630_2254col_10630_37953
RIUMA. Repositorio Institucional de la Universidad de Málaga
author
Romero-Galisteo, Rita Pilar
author
González-Sánchez, Manuel
author
Costa, Lara
author
Brandao, Rita
author
Ramalhete, Catia
author
Leao, Carla
author
Jacobsohn, Lia
2025-07-30T08:40:50Z
2025-07-30T08:40:50Z
2022-07
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.
https://hdl.handle.net/10630/39574
10.1016/j.ejpn.2022.06.003
Objective
The aim of the study was to identify and characterize outcome measures for objective and subjective assessment in persons with Rett syndrome (RS).
Methods
A 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.
Results
Twenty 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.
Conclusions
The 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.
eng
Rett, Síndrome de
Outcome measurement instruments in Rett syndrome: A systematic review
journal article
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URL
https://riuma.uma.es/bitstreams/1a033fe1-6fb5-44b8-9b81-ccddc4566748/download
File
MD5
552fd2782fe5d3329ba037929b2acee1
1161674
application/pdf
EJPN-REVISION MEDIDAS RESULTADO RETT-COSMISM version RIUMA.pdf