Sleep disturbances and behavior in Smith-Magenis syndrome

Background: The Smith-Magenis syndrome (SMS) shows a collection of neurodevelopmental problems including mild to moderate intellectual disability, change-related anxiety, impulsivity, speech delay, Attention-Deficit/Hyperactivity Disorder (ADH) and sleep disturbances. Sleep disorders, when present,...

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Autores: Garayzábal, Elena, Hidalgo, Irene, Miranda de Souza, Ana Luiza Decanini [UNESP], da Silva, Nathani Cristina [UNESP], Giacheti, Celia Maria [UNESP], Pinato, Luciana [UNESP]
Formato: artículo
Estado:Versión publicada
Fecha de publicación:2022
País:Brasil
Recursos:Universidade Estadual Paulista (UNESP)
Repositorio:Repositório Institucional da UNESP
Idioma:inglés
OAI Identifier:oai:repositorio.unesp.br:11449/241989
Acesso em linha:http://dx.doi.org/10.1016/j.ridd.2022.104286
http://hdl.handle.net/11449/241989
Access Level:acceso abierto
Palavra-chave:Behavior
Neurodevelopmental disorder
Sleep
Smith Magenis syndrome
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spelling Sleep disturbances and behavior in Smith-Magenis syndromeBehaviorNeurodevelopmental disorderSleepSmith Magenis syndromeBackground: The Smith-Magenis syndrome (SMS) shows a collection of neurodevelopmental problems including mild to moderate intellectual disability, change-related anxiety, impulsivity, speech delay, Attention-Deficit/Hyperactivity Disorder (ADH) and sleep disturbances. Sleep disorders, when present, have been treated in several populations with consecutive improvements in cognitive and behavioral aspects. Aims: To better understand the existing relationships between sleep disturbances and behavioral problems in SMS syndrome this study describes the sleep and behavior problems in the SMS and explores the possible relation between both. Methods and procedures: 17 individuals with SMS (50% males; 11.2 ± 4.9 years old) and 12 individuals with typical development (50% male; 11.1 ± 4.4 years old) were investigated using the Sleep Disturbance Scale for Children and the Child Behavior Checklist. Results: A high percentage (60%) of individuals with SMS have an indication of sleep disorders, being the most frequent disorders the sleep-wake transition disorders, and disorders of initiating and maintaining sleep with sleep latency higher than acceptable and total sleep time below acceptable. More than 94% of the SMS group presented clinical or borderline scores on the total behavioral problems scale. The most common behavioral problems were Externalizing Problems, Thought and Attention, ADH and Aggressive problems. There was a positive correlation between disorders of initiating and maintaining sleep, sleep-wake transition disorders, disorders of arousal, disorders of excessive somnolence and behavioral problems. Conclusions and implications: The worse the sleep disturbances investigated, the more severe the behavioral problems characteristics reinforcing the importance to address the sleep problems in the treatment of SMS individuals.Coordenação de Aperfeiçoamento de Pessoal de Nível Superior (CAPES)Departamento de Lingüística General Facultad de Filosofía y Letras Universidad Autónoma de MadridDepartamento de Filología Española Facultad de Filosofía y Letras Universidad Autónoma de MadridFacultad de Ciencias de la Salud Universidad de Castilla La ManchaCentro Universitario de Educación Superior (CUNIMAD)Department of Speech Language and Hearing Sciences São Paulo State University (UNESP), SPDepartment of Speech Language and Hearing Sciences São Paulo State University (UNESP), SPCAPES: 001Universidad Autónoma de MadridUniversidad de Castilla La ManchaCentro Universitario de Educación Superior (CUNIMAD)Universidade Estadual Paulista (UNESP)2023-03-02T06:29:02Z2023-03-02T06:29:02Z2022-09-01info:eu-repo/semantics/publishedVersioninfo:eu-repo/semantics/articlehttp://dx.doi.org/10.1016/j.ridd.2022.104286Research in Developmental Disabilities, v. 128.1873-33790891-4222http://hdl.handle.net/11449/24198910.1016/j.ridd.2022.1042862-s2.0-85132963818Scopusreponame:Repositório Institucional da UNESPinstname:Universidade Estadual Paulista (UNESP)instacron:UNESPengResearch in Developmental Disabilitiesinfo:eu-repo/semantics/openAccessGarayzábal, ElenaHidalgo, IreneMiranda de Souza, Ana Luiza Decanini [UNESP]da Silva, Nathani Cristina [UNESP]Giacheti, Celia Maria [UNESP]Pinato, Luciana [UNESP]2024-08-09T17:40:01Zoai:repositorio.unesp.br:11449/241989Repositório InstitucionalPUBhttp://repositorio.unesp.br/oai/requestrepositoriounesp@unesp.bropendoar:29462024-08-09T17:40:01Repositório Institucional da UNESP - Universidade Estadual Paulista (UNESP)false
dc.title.none.fl_str_mv Sleep disturbances and behavior in Smith-Magenis syndrome
title Sleep disturbances and behavior in Smith-Magenis syndrome
spellingShingle Sleep disturbances and behavior in Smith-Magenis syndrome
Garayzábal, Elena
Behavior
Neurodevelopmental disorder
Sleep
Smith Magenis syndrome
title_short Sleep disturbances and behavior in Smith-Magenis syndrome
title_full Sleep disturbances and behavior in Smith-Magenis syndrome
title_fullStr Sleep disturbances and behavior in Smith-Magenis syndrome
title_full_unstemmed Sleep disturbances and behavior in Smith-Magenis syndrome
title_sort Sleep disturbances and behavior in Smith-Magenis syndrome
dc.creator.none.fl_str_mv Garayzábal, Elena
Hidalgo, Irene
Miranda de Souza, Ana Luiza Decanini [UNESP]
da Silva, Nathani Cristina [UNESP]
Giacheti, Celia Maria [UNESP]
Pinato, Luciana [UNESP]
author Garayzábal, Elena
author_facet Garayzábal, Elena
Hidalgo, Irene
Miranda de Souza, Ana Luiza Decanini [UNESP]
da Silva, Nathani Cristina [UNESP]
Giacheti, Celia Maria [UNESP]
Pinato, Luciana [UNESP]
author_role author
author2 Hidalgo, Irene
Miranda de Souza, Ana Luiza Decanini [UNESP]
da Silva, Nathani Cristina [UNESP]
Giacheti, Celia Maria [UNESP]
Pinato, Luciana [UNESP]
author2_role author
author
author
author
author
dc.contributor.none.fl_str_mv Universidad Autónoma de Madrid
Universidad de Castilla La Mancha
Centro Universitario de Educación Superior (CUNIMAD)
Universidade Estadual Paulista (UNESP)
dc.subject.por.fl_str_mv Behavior
Neurodevelopmental disorder
Sleep
Smith Magenis syndrome
topic Behavior
Neurodevelopmental disorder
Sleep
Smith Magenis syndrome
description Background: The Smith-Magenis syndrome (SMS) shows a collection of neurodevelopmental problems including mild to moderate intellectual disability, change-related anxiety, impulsivity, speech delay, Attention-Deficit/Hyperactivity Disorder (ADH) and sleep disturbances. Sleep disorders, when present, have been treated in several populations with consecutive improvements in cognitive and behavioral aspects. Aims: To better understand the existing relationships between sleep disturbances and behavioral problems in SMS syndrome this study describes the sleep and behavior problems in the SMS and explores the possible relation between both. Methods and procedures: 17 individuals with SMS (50% males; 11.2 ± 4.9 years old) and 12 individuals with typical development (50% male; 11.1 ± 4.4 years old) were investigated using the Sleep Disturbance Scale for Children and the Child Behavior Checklist. Results: A high percentage (60%) of individuals with SMS have an indication of sleep disorders, being the most frequent disorders the sleep-wake transition disorders, and disorders of initiating and maintaining sleep with sleep latency higher than acceptable and total sleep time below acceptable. More than 94% of the SMS group presented clinical or borderline scores on the total behavioral problems scale. The most common behavioral problems were Externalizing Problems, Thought and Attention, ADH and Aggressive problems. There was a positive correlation between disorders of initiating and maintaining sleep, sleep-wake transition disorders, disorders of arousal, disorders of excessive somnolence and behavioral problems. Conclusions and implications: The worse the sleep disturbances investigated, the more severe the behavioral problems characteristics reinforcing the importance to address the sleep problems in the treatment of SMS individuals.
publishDate 2022
dc.date.none.fl_str_mv 2022-09-01
2023-03-02T06:29:02Z
2023-03-02T06:29:02Z
dc.type.status.fl_str_mv info:eu-repo/semantics/publishedVersion
dc.type.driver.fl_str_mv info:eu-repo/semantics/article
format article
status_str publishedVersion
dc.identifier.uri.fl_str_mv http://dx.doi.org/10.1016/j.ridd.2022.104286
Research in Developmental Disabilities, v. 128.
1873-3379
0891-4222
http://hdl.handle.net/11449/241989
10.1016/j.ridd.2022.104286
2-s2.0-85132963818
url http://dx.doi.org/10.1016/j.ridd.2022.104286
http://hdl.handle.net/11449/241989
identifier_str_mv Research in Developmental Disabilities, v. 128.
1873-3379
0891-4222
10.1016/j.ridd.2022.104286
2-s2.0-85132963818
dc.language.iso.fl_str_mv eng
language eng
dc.relation.none.fl_str_mv Research in Developmental Disabilities
dc.rights.driver.fl_str_mv info:eu-repo/semantics/openAccess
eu_rights_str_mv openAccess
dc.source.none.fl_str_mv Scopus
reponame:Repositório Institucional da UNESP
instname:Universidade Estadual Paulista (UNESP)
instacron:UNESP
instname_str Universidade Estadual Paulista (UNESP)
instacron_str UNESP
institution UNESP
reponame_str Repositório Institucional da UNESP
collection Repositório Institucional da UNESP
repository.name.fl_str_mv Repositório Institucional da UNESP - Universidade Estadual Paulista (UNESP)
repository.mail.fl_str_mv repositoriounesp@unesp.br
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score 15,198674

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