Quality of life outcomes and predictors for youth with intellectual disability and rare diseases

The concept of quality of Life (QoL) has become particularly relevant in the field of intelectual disability (ID). However, the existing scientific literature on QoL of people with ID and rare diseases (RDs) is very limited. Therefore, this study aimed to analyze individual QoL of children and youth...

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Autores: Morán Suárez, María Lucía|||0000-0001-5650-6711, Monsalve González, Asunción, Fontanil, Yolanda, Bacherini, Alice, Balboni, Giulia, Gómez Sánchez, Laura Elisabet
Tipo de recurso: artículo
Fecha de publicación:2025
País:España
Institución:Universidad de Cantabria (UC)
Repositorio:UCrea Repositorio Abierto de la Universidad de Cantabria
Idioma:inglés
OAI Identifier:oai:repositorio.unican.es:10902/39665
Acceso en línea:https://hdl.handle.net/10902/39665
Access Level:acceso abierto
Palabra clave:Intellectual developmental disorder
Fragile X syndrome
West syndrome
Children Adolescents
Young adults
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spelling Quality of life outcomes and predictors for youth with intellectual disability and rare diseasesMorán Suárez, María Lucía|||0000-0001-5650-6711Monsalve González, AsunciónFontanil, YolandaBacherini, AliceBalboni, GiuliaGómez Sánchez, Laura ElisabetIntellectual developmental disorderFragile X syndromeWest syndromeChildren AdolescentsYoung adultsThe concept of quality of Life (QoL) has become particularly relevant in the field of intelectual disability (ID). However, the existing scientific literature on QoL of people with ID and rare diseases (RDs) is very limited. Therefore, this study aimed to analyze individual QoL of children and youth with RDs and to evaluate the impact of personal and contextual factors on their QoL. The sample was composed of 114 Spanish participants with ID and RDs, aged between 4 and 21 years. The KidsLife Scale was applied. This instrument includes 96 items, third-person ormulated, organized across eight QOL domains: physical wellbeing, material wellbeing, emotional wellbeing, interpersonal relationships, social inclusion, rights and personal development. Descriptive statistics were calculated and correlation coefficients and multiple linear regression were computed. The most elevated scores were achieved in physical well-being and material wellbeing, whereas the lowest scores were shown in social inclusion and self-determination domains. Sex, level of support needs, percentage of disability and size of the organization were predictive factors of QoL scores. These outcomes shed light into priority areas and predictors that should be considered in the development of individualized supports, organizational approaches and policy initiatives intended to promote the QoL of children and youth with RDs and ID.This work has been funded by the Spanish Ministry of Economic and Competitiveness (PSI2012-33139); and the Ministry of Science, Innovation and Universities; and the State Research Agency (PID2019-105737RB-100/AEI/10.13039/501100011033).ElsevierUniversidad de Cantabria20252025-01-01journal articlehttp://purl.org/coar/resource_type/c_6501NAhttp://purl.org/coar/version/c_be7fb7dd8ff6fe43info:eu-repo/semantics/articlehttps://hdl.handle.net/10902/39665Research in Developmental Disabilities, 2025, 164, 105071reponame:UCrea Repositorio Abierto de la Universidad de Cantabriainstname:Universidad de Cantabria (UC)Inglésengopen accesshttp://purl.org/coar/access_right/c_abf2Attribution-NonCommercial-NoDerivatives 4.0 Internationalhttp://creativecommons.org/licenses/by-nc-nd/4.0/info:eu-repo/semantics/openAccessoai:repositorio.unican.es:10902/396652026-06-02T12:39:31Z
dc.title.none.fl_str_mv Quality of life outcomes and predictors for youth with intellectual disability and rare diseases
title Quality of life outcomes and predictors for youth with intellectual disability and rare diseases
spellingShingle Quality of life outcomes and predictors for youth with intellectual disability and rare diseases
Morán Suárez, María Lucía|||0000-0001-5650-6711
Intellectual developmental disorder
Fragile X syndrome
West syndrome
Children Adolescents
Young adults
title_short Quality of life outcomes and predictors for youth with intellectual disability and rare diseases
title_full Quality of life outcomes and predictors for youth with intellectual disability and rare diseases
title_fullStr Quality of life outcomes and predictors for youth with intellectual disability and rare diseases
title_full_unstemmed Quality of life outcomes and predictors for youth with intellectual disability and rare diseases
title_sort Quality of life outcomes and predictors for youth with intellectual disability and rare diseases
dc.creator.none.fl_str_mv Morán Suárez, María Lucía|||0000-0001-5650-6711
Monsalve González, Asunción
Fontanil, Yolanda
Bacherini, Alice
Balboni, Giulia
Gómez Sánchez, Laura Elisabet
author Morán Suárez, María Lucía|||0000-0001-5650-6711
author_facet Morán Suárez, María Lucía|||0000-0001-5650-6711
Monsalve González, Asunción
Fontanil, Yolanda
Bacherini, Alice
Balboni, Giulia
Gómez Sánchez, Laura Elisabet
author_role author
author2 Monsalve González, Asunción
Fontanil, Yolanda
Bacherini, Alice
Balboni, Giulia
Gómez Sánchez, Laura Elisabet
author2_role author
author
author
author
author
dc.contributor.none.fl_str_mv Universidad de Cantabria
dc.subject.none.fl_str_mv Intellectual developmental disorder
Fragile X syndrome
West syndrome
Children Adolescents
Young adults
topic Intellectual developmental disorder
Fragile X syndrome
West syndrome
Children Adolescents
Young adults
description The concept of quality of Life (QoL) has become particularly relevant in the field of intelectual disability (ID). However, the existing scientific literature on QoL of people with ID and rare diseases (RDs) is very limited. Therefore, this study aimed to analyze individual QoL of children and youth with RDs and to evaluate the impact of personal and contextual factors on their QoL. The sample was composed of 114 Spanish participants with ID and RDs, aged between 4 and 21 years. The KidsLife Scale was applied. This instrument includes 96 items, third-person ormulated, organized across eight QOL domains: physical wellbeing, material wellbeing, emotional wellbeing, interpersonal relationships, social inclusion, rights and personal development. Descriptive statistics were calculated and correlation coefficients and multiple linear regression were computed. The most elevated scores were achieved in physical well-being and material wellbeing, whereas the lowest scores were shown in social inclusion and self-determination domains. Sex, level of support needs, percentage of disability and size of the organization were predictive factors of QoL scores. These outcomes shed light into priority areas and predictors that should be considered in the development of individualized supports, organizational approaches and policy initiatives intended to promote the QoL of children and youth with RDs and ID.
publishDate 2025
dc.date.none.fl_str_mv 2025
2025-01-01
dc.type.none.fl_str_mv journal article
http://purl.org/coar/resource_type/c_6501
NA
http://purl.org/coar/version/c_be7fb7dd8ff6fe43
dc.type.openaire.fl_str_mv info:eu-repo/semantics/article
format article
dc.identifier.none.fl_str_mv https://hdl.handle.net/10902/39665
url https://hdl.handle.net/10902/39665
dc.language.none.fl_str_mv Inglés
eng
language_invalid_str_mv Inglés
language eng
dc.rights.none.fl_str_mv open access
http://purl.org/coar/access_right/c_abf2
Attribution-NonCommercial-NoDerivatives 4.0 International
http://creativecommons.org/licenses/by-nc-nd/4.0/
dc.rights.openaire.fl_str_mv info:eu-repo/semantics/openAccess
rights_invalid_str_mv open access
http://purl.org/coar/access_right/c_abf2
Attribution-NonCommercial-NoDerivatives 4.0 International
http://creativecommons.org/licenses/by-nc-nd/4.0/
eu_rights_str_mv openAccess
dc.publisher.none.fl_str_mv Elsevier
publisher.none.fl_str_mv Elsevier
dc.source.none.fl_str_mv Research in Developmental Disabilities, 2025, 164, 105071
reponame:UCrea Repositorio Abierto de la Universidad de Cantabria
instname:Universidad de Cantabria (UC)
instname_str Universidad de Cantabria (UC)
reponame_str UCrea Repositorio Abierto de la Universidad de Cantabria
collection UCrea Repositorio Abierto de la Universidad de Cantabria
repository.name.fl_str_mv
repository.mail.fl_str_mv
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score 15,81155

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