An investigation of the middle and late behavioural phenotypes of Mucopolysaccharidosis Type-III / E. M. CROSS in Journal of Neurodevelopmental Disorders, 6-1 (December 2014)  

Public ISBD [article]  inJournal of Neurodevelopmental Disorders > 6-1 (December 2014) . - p.46 Titre : An investigation of the middle and late behavioural phenotypes of Mucopolysaccharidosis Type-III Type de document : Texte imprimé et/ou numérique Auteurs : E. M. CROSS, Auteur ; S. GRANT, Auteur ; S. JONES, Auteur ; B. W. BIGGER, Auteur ; J. E. WRAITH, Auteur ; L. V. MAHON, Auteur ; M. LOMAX, Auteur ; D. J. HARE, Auteur Article en page(s) : p.46 Langues : Anglais (eng) Mots-clés : Behavioural phenotype  Mps iii  Mucopolysaccharidosis  Sanfilippo syndrome Index. décimale : PER Périodiques Résumé : BACKGROUND: Mucopolysaccharidosis type-III (MPS III) is an autosomal recessive lysosomal storage disorder. It causes progressive physical and cognitive decline and has been linked to increased incidences of behavioural problems. METHODS: Data on the behaviour and adaptive skills of 20 children with MPS III and 25 children with intellectual disability (ID) (17 included in analysis) were gathered via parental report questionnaire. The frequencies of different types of behaviour displayed by children with MPS III and children with ID were compared across two age categories. RESULTS: The total frequency of challenging behaviours displayed by children aged 2-9 years with MPS III and ID was not significantly different. Behaviours associated with hyperactivity, orality, unusual body movements and inattention were seen significantly more frequently in 2-9 year olds with MPS III than in those with ID. Children aged 10-15 years with MPS III showed significantly fewer problem behaviours than a contrasting group with ID. The frequency of challenging behaviours displayed by children with MPS III and their adaptive skills was found to decrease with age. CONCLUSIONS: Behaviours relating to hyperactivity, orality, unusual body movements and inattention are part of the behavioural phenotype of the middle phase of MPS III. The late phase of MPS III is associated with low rates of problem behaviour and loss of adaptive skills. Therefore, families with a child with MPS III may benefit from a different type of clinical service when the child is aged 2-9 years, than when aged 10-15 years. En ligne : http://dx.doi.org/10.1186/1866-1955-6-46 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=347 [article] An investigation of the middle and late behavioural phenotypes of Mucopolysaccharidosis Type-III [Texte imprimé et/ou numérique] / E. M. CROSS, Auteur ; S. GRANT, Auteur ; S. JONES, Auteur ; B. W. BIGGER, Auteur ; J. E. WRAITH, Auteur ; L. V. MAHON, Auteur ; M. LOMAX, Auteur ; D. J. HARE, Auteur . - p.46. Langues : Anglais (eng) in Journal of Neurodevelopmental Disorders > 6-1 (December 2014) . - p.46 Mots-clés : Behavioural phenotype  Mps iii  Mucopolysaccharidosis  Sanfilippo syndrome Index. décimale : PER Périodiques Résumé : BACKGROUND: Mucopolysaccharidosis type-III (MPS III) is an autosomal recessive lysosomal storage disorder. It causes progressive physical and cognitive decline and has been linked to increased incidences of behavioural problems. METHODS: Data on the behaviour and adaptive skills of 20 children with MPS III and 25 children with intellectual disability (ID) (17 included in analysis) were gathered via parental report questionnaire. The frequencies of different types of behaviour displayed by children with MPS III and children with ID were compared across two age categories. RESULTS: The total frequency of challenging behaviours displayed by children aged 2-9 years with MPS III and ID was not significantly different. Behaviours associated with hyperactivity, orality, unusual body movements and inattention were seen significantly more frequently in 2-9 year olds with MPS III than in those with ID. Children aged 10-15 years with MPS III showed significantly fewer problem behaviours than a contrasting group with ID. The frequency of challenging behaviours displayed by children with MPS III and their adaptive skills was found to decrease with age. CONCLUSIONS: Behaviours relating to hyperactivity, orality, unusual body movements and inattention are part of the behavioural phenotype of the middle phase of MPS III. The late phase of MPS III is associated with low rates of problem behaviour and loss of adaptive skills. Therefore, families with a child with MPS III may benefit from a different type of clinical service when the child is aged 2-9 years, than when aged 10-15 years. En ligne : http://dx.doi.org/10.1186/1866-1955-6-46 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=347

Autism and chronic ill health: an observational study of symptoms and diagnoses of central sensitivity syndromes in autistic adults / S. GRANT in Molecular Autism, 13 (2022)  

Public ISBD [article]  inMolecular Autism > 13 (2022) . - 7p. Titre : Autism and chronic ill health: an observational study of symptoms and diagnoses of central sensitivity syndromes in autistic adults Type de document : Texte imprimé et/ou numérique Auteurs : S. GRANT, Auteur ; S. NORTON, Auteur ; R. F. WEILAND, Auteur ; Anke M. SCHEEREN, Auteur ; Sander BEGEER, Auteur ; R. A. HOEKSTRA, Auteur Article en page(s) : 7p. Langues : Anglais (eng) Mots-clés : Autism  Central sensitisation  Chronic pain  Fatigue  Fibromyalgia  Sensory processing  Sensory sensitivity Index. décimale : PER Périodiques Résumé : BACKGROUND: Autistic adults, particularly women, are more likely to experience chronic ill health than the general population. Central sensitivity syndromes (CSS) are a group of related conditions that are thought to include an underlying sensitisation of the central nervous system; heightened sensory sensitivity is a common feature. Anecdotal evidence suggests autistic adults may be more prone to developing a CSS. This study aimed to investigate the occurrence of CSS diagnoses and symptoms in autistic adults, and to explore whether CSS symptoms were related to autistic traits, mental health, sensory sensitivity, or gender. METHODS: The full sample of participants included 973 autistic adults (410 men, 563 women, mean age?=?44.6) registered at the Netherlands Autism Register, who completed questionnaires assessing autistic traits, sensory sensitivity, CSS, physical and mental health symptoms. The reliability and validity of the Central Sensitization Inventory (CSI) in an autistic sample was established using exploratory and confirmatory factor analyses. Chi(2) analyses, independent t-tests, hierarchical regression and path analysis were used to analyse relationships between CSS symptoms, autistic traits, measures of mental health and wellbeing, sensory sensitivity, age and gender. RESULTS: 21% of participants reported one or more CSS diagnosis, and 60% scored at or above the clinical cut-off for a CSS. Autistic women were more likely to report a CSS diagnosis and experienced more CSS symptoms than men. Sensory sensitivity, anxiety, age and gender were significant predictors of CSS symptoms, with sensory sensitivity and anxiety fully mediating the relationship between autistic traits and CSS symptoms. LIMITATIONS: Although this study included a large sample of autistic adults, we did not have a control group or a CSS only group. We also could not include a non-binary group due to lack of statistical power. CONCLUSIONS: CSS diagnoses and symptoms appear to be very common in the autistic population. Increased awareness of an association between autism and central sensitisation should inform clinicians and guide diagnostic practice, particularly for women where CSS are common and autism under recognised. En ligne : http://dx.doi.org/10.1186/s13229-022-00486-6 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=459 [article] Autism and chronic ill health: an observational study of symptoms and diagnoses of central sensitivity syndromes in autistic adults [Texte imprimé et/ou numérique] / S. GRANT, Auteur ; S. NORTON, Auteur ; R. F. WEILAND, Auteur ; Anke M. SCHEEREN, Auteur ; Sander BEGEER, Auteur ; R. A. HOEKSTRA, Auteur . - 7p. Langues : Anglais (eng) in Molecular Autism > 13 (2022) . - 7p. Mots-clés : Autism  Central sensitisation  Chronic pain  Fatigue  Fibromyalgia  Sensory processing  Sensory sensitivity Index. décimale : PER Périodiques Résumé : BACKGROUND: Autistic adults, particularly women, are more likely to experience chronic ill health than the general population. Central sensitivity syndromes (CSS) are a group of related conditions that are thought to include an underlying sensitisation of the central nervous system; heightened sensory sensitivity is a common feature. Anecdotal evidence suggests autistic adults may be more prone to developing a CSS. This study aimed to investigate the occurrence of CSS diagnoses and symptoms in autistic adults, and to explore whether CSS symptoms were related to autistic traits, mental health, sensory sensitivity, or gender. METHODS: The full sample of participants included 973 autistic adults (410 men, 563 women, mean age?=?44.6) registered at the Netherlands Autism Register, who completed questionnaires assessing autistic traits, sensory sensitivity, CSS, physical and mental health symptoms. The reliability and validity of the Central Sensitization Inventory (CSI) in an autistic sample was established using exploratory and confirmatory factor analyses. Chi(2) analyses, independent t-tests, hierarchical regression and path analysis were used to analyse relationships between CSS symptoms, autistic traits, measures of mental health and wellbeing, sensory sensitivity, age and gender. RESULTS: 21% of participants reported one or more CSS diagnosis, and 60% scored at or above the clinical cut-off for a CSS. Autistic women were more likely to report a CSS diagnosis and experienced more CSS symptoms than men. Sensory sensitivity, anxiety, age and gender were significant predictors of CSS symptoms, with sensory sensitivity and anxiety fully mediating the relationship between autistic traits and CSS symptoms. LIMITATIONS: Although this study included a large sample of autistic adults, we did not have a control group or a CSS only group. We also could not include a non-binary group due to lack of statistical power. CONCLUSIONS: CSS diagnoses and symptoms appear to be very common in the autistic population. Increased awareness of an association between autism and central sensitisation should inform clinicians and guide diagnostic practice, particularly for women where CSS are common and autism under recognised. En ligne : http://dx.doi.org/10.1186/s13229-022-00486-6 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=459

Research Review: Cognitive bias modification of interpretations in youth and its effect on anxiety: a meta-analysis / G. KREBS in Journal of Child Psychology and Psychiatry, 59-8 (August 2018)  

Public ISBD [article]  inJournal of Child Psychology and Psychiatry > 59-8 (August 2018) . - p.831-844 Titre : Research Review: Cognitive bias modification of interpretations in youth and its effect on anxiety: a meta-analysis Type de document : Texte imprimé et/ou numérique Auteurs : G. KREBS, Auteur ; V. PILE, Auteur ; S. GRANT, Auteur ; M. DEGLI ESPOSTI, Auteur ; P. MONTGOMERY, Auteur ; J. Y. F. LAU, Auteur Article en page(s) : p.831-844 Langues : Anglais (eng) Mots-clés : Cognitive bias modification  adolescents  anxiety  children  interpretation bias training Index. décimale : PER Périodiques Résumé : BACKGROUND: Emerging evidence suggests that cognitive bias modification of interpretations (CBM-I) is effective in altering interpretation biases and reducing anxiety in adults. Less is known about the impact of CBM-I in young people, but some recent findings, including a meta-analysis of combined cognitive bias modification of interpretation and attention techniques, have cast doubt on its clinical utility. Given the current debate, this meta-analysis sought to establish the independent effects of CBM-I on interpretations biases and anxiety in youth. METHODS: Studies were identified through a systematic literature search of PsycINFO, Ovid MEDLINE, PsycARTICLES, Web of Science and EMBASE between January 1992 and March 2017. Eligible studies aimed to target interpretation biases; did not combine CBM-I with another intervention; included a control condition; randomly allocated participants to conditions; assessed interpretation bias and/or anxiety as an outcome; included individuals up to age 18; and did not present previously reported data. Reference lists of included articles were checked for further eligible studies, and authors were contacted for unpublished data. RESULTS: We identified 26 studies meeting eligibility criteria that included in the meta-analysis. CBM-I had moderate effects on negative and positive interpretations (g = -0.70 and g = -0.52, respectively) and a small but significant effect on anxiety assessed after training (g = -0.17) and after a stressor (g = -0.34). No significant moderators were identified. CONCLUSIONS: In contrast to previous meta-analytic findings, our results indicate that CBM-I has potential but weak anxiolytic effects in youth. Our findings suggest that it may be premature to disregard the potential value of CBM-I research and further research in this field is warranted. En ligne : http://dx.doi.org/10.1111/jcpp.12809 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=368 [article] Research Review: Cognitive bias modification of interpretations in youth and its effect on anxiety: a meta-analysis [Texte imprimé et/ou numérique] / G. KREBS, Auteur ; V. PILE, Auteur ; S. GRANT, Auteur ; M. DEGLI ESPOSTI, Auteur ; P. MONTGOMERY, Auteur ; J. Y. F. LAU, Auteur . - p.831-844. Langues : Anglais (eng) in Journal of Child Psychology and Psychiatry > 59-8 (August 2018) . - p.831-844 Mots-clés : Cognitive bias modification  adolescents  anxiety  children  interpretation bias training Index. décimale : PER Périodiques Résumé : BACKGROUND: Emerging evidence suggests that cognitive bias modification of interpretations (CBM-I) is effective in altering interpretation biases and reducing anxiety in adults. Less is known about the impact of CBM-I in young people, but some recent findings, including a meta-analysis of combined cognitive bias modification of interpretation and attention techniques, have cast doubt on its clinical utility. Given the current debate, this meta-analysis sought to establish the independent effects of CBM-I on interpretations biases and anxiety in youth. METHODS: Studies were identified through a systematic literature search of PsycINFO, Ovid MEDLINE, PsycARTICLES, Web of Science and EMBASE between January 1992 and March 2017. Eligible studies aimed to target interpretation biases; did not combine CBM-I with another intervention; included a control condition; randomly allocated participants to conditions; assessed interpretation bias and/or anxiety as an outcome; included individuals up to age 18; and did not present previously reported data. Reference lists of included articles were checked for further eligible studies, and authors were contacted for unpublished data. RESULTS: We identified 26 studies meeting eligibility criteria that included in the meta-analysis. CBM-I had moderate effects on negative and positive interpretations (g = -0.70 and g = -0.52, respectively) and a small but significant effect on anxiety assessed after training (g = -0.17) and after a stressor (g = -0.34). No significant moderators were identified. CONCLUSIONS: In contrast to previous meta-analytic findings, our results indicate that CBM-I has potential but weak anxiolytic effects in youth. Our findings suggest that it may be premature to disregard the potential value of CBM-I research and further research in this field is warranted. En ligne : http://dx.doi.org/10.1111/jcpp.12809 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=368

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