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Observable Symptoms of Anxiety in Individuals with Fragile X Syndrome: Parent and Caregiver Perspectives

Caregiver reports, clinical observations, and diagnostic assessments indicate that most individuals with fragile X syndrome experience high levels of chronic anxiety. However, anxiety is a challenging endpoint for outcome measurement in FXS because most individuals cannot reliably report internal em...

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Autores principales: Lozano, Reymundo, Thompson, Talia, Dixon-Weber, Jayne, Erickson, Craig A., Berry-Kravis, Elizabeth, Williams, Sara, Smith, Elizabeth, Frazier, Jean A., Rosselot, Hilary, Farmer, Cristan, Hessl, David
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9498703/
https://www.ncbi.nlm.nih.gov/pubmed/36140827
http://dx.doi.org/10.3390/genes13091660
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author Lozano, Reymundo
Thompson, Talia
Dixon-Weber, Jayne
Erickson, Craig A.
Berry-Kravis, Elizabeth
Williams, Sara
Smith, Elizabeth
Frazier, Jean A.
Rosselot, Hilary
Farmer, Cristan
Hessl, David
author_facet Lozano, Reymundo
Thompson, Talia
Dixon-Weber, Jayne
Erickson, Craig A.
Berry-Kravis, Elizabeth
Williams, Sara
Smith, Elizabeth
Frazier, Jean A.
Rosselot, Hilary
Farmer, Cristan
Hessl, David
author_sort Lozano, Reymundo
collection PubMed
description Caregiver reports, clinical observations, and diagnostic assessments indicate that most individuals with fragile X syndrome experience high levels of chronic anxiety. However, anxiety is a challenging endpoint for outcome measurement in FXS because most individuals cannot reliably report internal emotional or body states. A comprehensive survey of the presence, frequency, and duration of anxiety-related symptoms and questions to elicit open-ended responses was completed by caregivers of 456 individuals with FXS, ages 2–81 years (87 female, 369 male) and 24 female and 2 male FXS self-advocates ages 15–66 years. Caregivers reported classic behavioral indicators of anxiety, such as avoidance, irritability, motor agitation, and physiological symptoms, as well as behavioral features in FXS such as repetitive behavior, aggression, and self-injury. Self-advocate accounts largely paralleled caregiver data. Factor analyses yielded four factors: (1) increased irritability, aggression, and self-injury; (2) increased physical movement, nervous activity, and restlessness; (3) physical and physiological features of anxiety; and (4) internalizing and gastrointestinal symptoms. Caregivers are capable of observing and reporting behaviors that are valid indicators of anxious states that are usually reported in self-report standardized assessments. These results support the development of an anxiety measure for FXS that minimizes problems with rater inference.
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spelling pubmed-94987032022-09-23 Observable Symptoms of Anxiety in Individuals with Fragile X Syndrome: Parent and Caregiver Perspectives Lozano, Reymundo Thompson, Talia Dixon-Weber, Jayne Erickson, Craig A. Berry-Kravis, Elizabeth Williams, Sara Smith, Elizabeth Frazier, Jean A. Rosselot, Hilary Farmer, Cristan Hessl, David Genes (Basel) Article Caregiver reports, clinical observations, and diagnostic assessments indicate that most individuals with fragile X syndrome experience high levels of chronic anxiety. However, anxiety is a challenging endpoint for outcome measurement in FXS because most individuals cannot reliably report internal emotional or body states. A comprehensive survey of the presence, frequency, and duration of anxiety-related symptoms and questions to elicit open-ended responses was completed by caregivers of 456 individuals with FXS, ages 2–81 years (87 female, 369 male) and 24 female and 2 male FXS self-advocates ages 15–66 years. Caregivers reported classic behavioral indicators of anxiety, such as avoidance, irritability, motor agitation, and physiological symptoms, as well as behavioral features in FXS such as repetitive behavior, aggression, and self-injury. Self-advocate accounts largely paralleled caregiver data. Factor analyses yielded four factors: (1) increased irritability, aggression, and self-injury; (2) increased physical movement, nervous activity, and restlessness; (3) physical and physiological features of anxiety; and (4) internalizing and gastrointestinal symptoms. Caregivers are capable of observing and reporting behaviors that are valid indicators of anxious states that are usually reported in self-report standardized assessments. These results support the development of an anxiety measure for FXS that minimizes problems with rater inference. MDPI 2022-09-16 /pmc/articles/PMC9498703/ /pubmed/36140827 http://dx.doi.org/10.3390/genes13091660 Text en © 2022 by the authors. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/).
spellingShingle Article
Lozano, Reymundo
Thompson, Talia
Dixon-Weber, Jayne
Erickson, Craig A.
Berry-Kravis, Elizabeth
Williams, Sara
Smith, Elizabeth
Frazier, Jean A.
Rosselot, Hilary
Farmer, Cristan
Hessl, David
Observable Symptoms of Anxiety in Individuals with Fragile X Syndrome: Parent and Caregiver Perspectives
title Observable Symptoms of Anxiety in Individuals with Fragile X Syndrome: Parent and Caregiver Perspectives
title_full Observable Symptoms of Anxiety in Individuals with Fragile X Syndrome: Parent and Caregiver Perspectives
title_fullStr Observable Symptoms of Anxiety in Individuals with Fragile X Syndrome: Parent and Caregiver Perspectives
title_full_unstemmed Observable Symptoms of Anxiety in Individuals with Fragile X Syndrome: Parent and Caregiver Perspectives
title_short Observable Symptoms of Anxiety in Individuals with Fragile X Syndrome: Parent and Caregiver Perspectives
title_sort observable symptoms of anxiety in individuals with fragile x syndrome: parent and caregiver perspectives
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9498703/
https://www.ncbi.nlm.nih.gov/pubmed/36140827
http://dx.doi.org/10.3390/genes13091660
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