Autism Symptoms in Children and Young Adults With Fragile X Syndrome, Angelman Syndrome, Tuberous Sclerosis Complex, and Neurofibromatosis Type 1: A Cross-Syndrome Comparison
ObjectiveThe etiology of autism spectrum disorder (ASD) remains unclear, due to genetic heterogeneity and heterogeneity in symptoms across individuals. This study compares ASD symptomatology between monogenetic syndromes with a high ASD prevalence, in order to reveal syndrome specific vulnerabilitie...
Ausführliche Beschreibung
Gespeichert in:
| Autor*in: |
Kyra Lubbers [verfasserIn] Eefje M. Stijl [verfasserIn] Bram Dierckx [verfasserIn] Doesjka A. Hagenaar [verfasserIn] Leontine W. ten Hoopen [verfasserIn] Jeroen S. Legerstee [verfasserIn] Pieter F. A. de Nijs [verfasserIn] André B. Rietman [verfasserIn] Kirstin Greaves-Lord [verfasserIn] Manon H. J. Hillegers [verfasserIn] Gwendolyn C. Dieleman [verfasserIn] Sabine E. Mous [verfasserIn] ENCORE Expertise Center [verfasserIn] Rianne Oostenbrink [verfasserIn] Karen C.G.B. Bindels-de Heus [verfasserIn] Marie-Claire Y. de Wit [verfasserIn] Henriëtte A. Mol [verfasserIn] Ype Elgersma [verfasserIn] |
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| Format: |
E-Artikel |
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| Sprache: |
Englisch |
| Erschienen: |
2022 |
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| Schlagwörter: |
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| Übergeordnetes Werk: |
In: Frontiers in Psychiatry - Frontiers Media S.A., 2010, 13(2022) |
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| Übergeordnetes Werk: |
volume:13 ; year:2022 |
| Links: |
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| DOI / URN: |
10.3389/fpsyt.2022.852208 |
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| Katalog-ID: |
DOAJ031247717 |
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| 245 | 1 | 0 | |a Autism Symptoms in Children and Young Adults With Fragile X Syndrome, Angelman Syndrome, Tuberous Sclerosis Complex, and Neurofibromatosis Type 1: A Cross-Syndrome Comparison |
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| 520 | |a ObjectiveThe etiology of autism spectrum disorder (ASD) remains unclear, due to genetic heterogeneity and heterogeneity in symptoms across individuals. This study compares ASD symptomatology between monogenetic syndromes with a high ASD prevalence, in order to reveal syndrome specific vulnerabilities and to clarify how genetic variations affect ASD symptom presentation.MethodsWe assessed ASD symptom severity in children and young adults (aged 0-28 years) with Fragile X Syndrome (FXS, n = 60), Angelman Syndrome (AS, n = 91), Neurofibromatosis Type 1 (NF1, n = 279) and Tuberous Sclerosis Complex (TSC, n = 110), using the Autism Diagnostic Observation Schedule and Social Responsiveness Scale. Assessments were part of routine clinical care at the ENCORE expertise center in Rotterdam, the Netherlands. First, we compared the syndrome groups on the ASD classification prevalence and ASD severity scores. Then, we compared individuals in our syndrome groups with an ASD classification to a non-syndromic ASD group (nsASD, n = 335), on both ASD severity scores and ASD symptom profiles. Severity scores were compared using MANCOVAs with IQ and gender as covariates.ResultsOverall, ASD severity scores were highest for the FXS group and lowest for the NF1 group. Compared to nsASD, individuals with an ASD classification in our syndrome groups showed less problems on the instruments' social domains. We found a relative strength in the AS group on the social cognition, communication and motivation domains and a relative challenge in creativity; a relative strength of the NF1 group on the restricted interests and repetitive behavior scale; and a relative challenge in the FXS and TSC groups on the restricted interests and repetitive behavior domain.ConclusionThe syndrome-specific strengths and challenges we found provide a frame of reference to evaluate an individual's symptoms relative to the larger syndromic population and to guide treatment decisions. Our findings support the need for personalized care and a dimensional, symptom-based diagnostic approach, in contrast to a dichotomous ASD diagnosis used as a prerequisite for access to healthcare services. Similarities in ASD symptom profiles between AS and FXS, and between NF1 and TSC may reflect similarities in their neurobiology. Deep phenotyping studies are required to link neurobiological markers to ASD symptomatology. | ||
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10.3389/fpsyt.2022.852208 doi (DE-627)DOAJ031247717 (DE-599)DOAJ202fd10196a84dc78ef4070ab5f15967 DE-627 ger DE-627 rakwb eng RC435-571 Kyra Lubbers verfasserin aut Autism Symptoms in Children and Young Adults With Fragile X Syndrome, Angelman Syndrome, Tuberous Sclerosis Complex, and Neurofibromatosis Type 1: A Cross-Syndrome Comparison 2022 Text txt rdacontent Computermedien c rdamedia Online-Ressource cr rdacarrier ObjectiveThe etiology of autism spectrum disorder (ASD) remains unclear, due to genetic heterogeneity and heterogeneity in symptoms across individuals. This study compares ASD symptomatology between monogenetic syndromes with a high ASD prevalence, in order to reveal syndrome specific vulnerabilities and to clarify how genetic variations affect ASD symptom presentation.MethodsWe assessed ASD symptom severity in children and young adults (aged 0-28 years) with Fragile X Syndrome (FXS, n = 60), Angelman Syndrome (AS, n = 91), Neurofibromatosis Type 1 (NF1, n = 279) and Tuberous Sclerosis Complex (TSC, n = 110), using the Autism Diagnostic Observation Schedule and Social Responsiveness Scale. Assessments were part of routine clinical care at the ENCORE expertise center in Rotterdam, the Netherlands. First, we compared the syndrome groups on the ASD classification prevalence and ASD severity scores. Then, we compared individuals in our syndrome groups with an ASD classification to a non-syndromic ASD group (nsASD, n = 335), on both ASD severity scores and ASD symptom profiles. Severity scores were compared using MANCOVAs with IQ and gender as covariates.ResultsOverall, ASD severity scores were highest for the FXS group and lowest for the NF1 group. Compared to nsASD, individuals with an ASD classification in our syndrome groups showed less problems on the instruments' social domains. We found a relative strength in the AS group on the social cognition, communication and motivation domains and a relative challenge in creativity; a relative strength of the NF1 group on the restricted interests and repetitive behavior scale; and a relative challenge in the FXS and TSC groups on the restricted interests and repetitive behavior domain.ConclusionThe syndrome-specific strengths and challenges we found provide a frame of reference to evaluate an individual's symptoms relative to the larger syndromic population and to guide treatment decisions. Our findings support the need for personalized care and a dimensional, symptom-based diagnostic approach, in contrast to a dichotomous ASD diagnosis used as a prerequisite for access to healthcare services. Similarities in ASD symptom profiles between AS and FXS, and between NF1 and TSC may reflect similarities in their neurobiology. Deep phenotyping studies are required to link neurobiological markers to ASD symptomatology. Fragile X Syndrome Angelman Syndrome Tuberous Sclerosis Complex Neurofibromatosis Type 1 autism spectrum disorder autistic traits Psychiatry Kyra Lubbers verfasserin aut Kyra Lubbers verfasserin aut Eefje M. Stijl verfasserin aut Eefje M. Stijl verfasserin aut Eefje M. Stijl verfasserin aut Bram Dierckx verfasserin aut Bram Dierckx verfasserin aut Bram Dierckx verfasserin aut Doesjka A. Hagenaar verfasserin aut Doesjka A. Hagenaar verfasserin aut Doesjka A. Hagenaar verfasserin aut Doesjka A. Hagenaar verfasserin aut Leontine W. ten Hoopen verfasserin aut Leontine W. ten Hoopen verfasserin aut Leontine W. ten Hoopen verfasserin aut Jeroen S. Legerstee verfasserin aut Jeroen S. Legerstee verfasserin aut Jeroen S. Legerstee verfasserin aut Pieter F. A. de Nijs verfasserin aut Pieter F. A. de Nijs verfasserin aut Pieter F. A. de Nijs verfasserin aut André B. Rietman verfasserin aut André B. Rietman verfasserin aut André B. Rietman verfasserin aut Kirstin Greaves-Lord verfasserin aut Kirstin Greaves-Lord verfasserin aut Kirstin Greaves-Lord verfasserin aut Kirstin Greaves-Lord verfasserin aut Manon H. J. Hillegers verfasserin aut Manon H. J. Hillegers verfasserin aut Manon H. J. Hillegers verfasserin aut Gwendolyn C. Dieleman verfasserin aut Gwendolyn C. Dieleman verfasserin aut Gwendolyn C. Dieleman verfasserin aut Sabine E. Mous verfasserin aut Sabine E. Mous verfasserin aut Sabine E. Mous verfasserin aut ENCORE Expertise Center verfasserin aut Rianne Oostenbrink verfasserin aut Karen C.G.B. Bindels-de Heus verfasserin aut Marie-Claire Y. de Wit verfasserin aut Henriëtte A. Mol verfasserin aut Ype Elgersma verfasserin aut In Frontiers in Psychiatry Frontiers Media S.A., 2010 13(2022) (DE-627)631498796 (DE-600)2564218-2 16640640 nnns volume:13 year:2022 https://doi.org/10.3389/fpsyt.2022.852208 kostenfrei https://doaj.org/article/202fd10196a84dc78ef4070ab5f15967 kostenfrei https://www.frontiersin.org/articles/10.3389/fpsyt.2022.852208/full kostenfrei https://doaj.org/toc/1664-0640 Journal toc kostenfrei GBV_USEFLAG_A SYSFLAG_A GBV_DOAJ SSG-OLC-PHA GBV_ILN_20 GBV_ILN_22 GBV_ILN_23 GBV_ILN_24 GBV_ILN_31 GBV_ILN_39 GBV_ILN_40 GBV_ILN_60 GBV_ILN_62 GBV_ILN_63 GBV_ILN_65 GBV_ILN_69 GBV_ILN_73 GBV_ILN_74 GBV_ILN_95 GBV_ILN_105 GBV_ILN_110 GBV_ILN_151 GBV_ILN_161 GBV_ILN_170 GBV_ILN_206 GBV_ILN_213 GBV_ILN_230 GBV_ILN_285 GBV_ILN_293 GBV_ILN_602 GBV_ILN_2014 GBV_ILN_4012 GBV_ILN_4037 GBV_ILN_4112 GBV_ILN_4125 GBV_ILN_4126 GBV_ILN_4249 GBV_ILN_4305 GBV_ILN_4306 GBV_ILN_4307 GBV_ILN_4313 GBV_ILN_4322 GBV_ILN_4323 GBV_ILN_4324 GBV_ILN_4325 GBV_ILN_4338 GBV_ILN_4367 GBV_ILN_4700 AR 13 2022 |
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10.3389/fpsyt.2022.852208 doi (DE-627)DOAJ031247717 (DE-599)DOAJ202fd10196a84dc78ef4070ab5f15967 DE-627 ger DE-627 rakwb eng RC435-571 Kyra Lubbers verfasserin aut Autism Symptoms in Children and Young Adults With Fragile X Syndrome, Angelman Syndrome, Tuberous Sclerosis Complex, and Neurofibromatosis Type 1: A Cross-Syndrome Comparison 2022 Text txt rdacontent Computermedien c rdamedia Online-Ressource cr rdacarrier ObjectiveThe etiology of autism spectrum disorder (ASD) remains unclear, due to genetic heterogeneity and heterogeneity in symptoms across individuals. This study compares ASD symptomatology between monogenetic syndromes with a high ASD prevalence, in order to reveal syndrome specific vulnerabilities and to clarify how genetic variations affect ASD symptom presentation.MethodsWe assessed ASD symptom severity in children and young adults (aged 0-28 years) with Fragile X Syndrome (FXS, n = 60), Angelman Syndrome (AS, n = 91), Neurofibromatosis Type 1 (NF1, n = 279) and Tuberous Sclerosis Complex (TSC, n = 110), using the Autism Diagnostic Observation Schedule and Social Responsiveness Scale. Assessments were part of routine clinical care at the ENCORE expertise center in Rotterdam, the Netherlands. First, we compared the syndrome groups on the ASD classification prevalence and ASD severity scores. Then, we compared individuals in our syndrome groups with an ASD classification to a non-syndromic ASD group (nsASD, n = 335), on both ASD severity scores and ASD symptom profiles. Severity scores were compared using MANCOVAs with IQ and gender as covariates.ResultsOverall, ASD severity scores were highest for the FXS group and lowest for the NF1 group. Compared to nsASD, individuals with an ASD classification in our syndrome groups showed less problems on the instruments' social domains. We found a relative strength in the AS group on the social cognition, communication and motivation domains and a relative challenge in creativity; a relative strength of the NF1 group on the restricted interests and repetitive behavior scale; and a relative challenge in the FXS and TSC groups on the restricted interests and repetitive behavior domain.ConclusionThe syndrome-specific strengths and challenges we found provide a frame of reference to evaluate an individual's symptoms relative to the larger syndromic population and to guide treatment decisions. Our findings support the need for personalized care and a dimensional, symptom-based diagnostic approach, in contrast to a dichotomous ASD diagnosis used as a prerequisite for access to healthcare services. Similarities in ASD symptom profiles between AS and FXS, and between NF1 and TSC may reflect similarities in their neurobiology. Deep phenotyping studies are required to link neurobiological markers to ASD symptomatology. Fragile X Syndrome Angelman Syndrome Tuberous Sclerosis Complex Neurofibromatosis Type 1 autism spectrum disorder autistic traits Psychiatry Kyra Lubbers verfasserin aut Kyra Lubbers verfasserin aut Eefje M. Stijl verfasserin aut Eefje M. Stijl verfasserin aut Eefje M. Stijl verfasserin aut Bram Dierckx verfasserin aut Bram Dierckx verfasserin aut Bram Dierckx verfasserin aut Doesjka A. Hagenaar verfasserin aut Doesjka A. Hagenaar verfasserin aut Doesjka A. Hagenaar verfasserin aut Doesjka A. Hagenaar verfasserin aut Leontine W. ten Hoopen verfasserin aut Leontine W. ten Hoopen verfasserin aut Leontine W. ten Hoopen verfasserin aut Jeroen S. Legerstee verfasserin aut Jeroen S. Legerstee verfasserin aut Jeroen S. Legerstee verfasserin aut Pieter F. A. de Nijs verfasserin aut Pieter F. A. de Nijs verfasserin aut Pieter F. A. de Nijs verfasserin aut André B. Rietman verfasserin aut André B. Rietman verfasserin aut André B. Rietman verfasserin aut Kirstin Greaves-Lord verfasserin aut Kirstin Greaves-Lord verfasserin aut Kirstin Greaves-Lord verfasserin aut Kirstin Greaves-Lord verfasserin aut Manon H. J. Hillegers verfasserin aut Manon H. J. Hillegers verfasserin aut Manon H. J. Hillegers verfasserin aut Gwendolyn C. Dieleman verfasserin aut Gwendolyn C. Dieleman verfasserin aut Gwendolyn C. Dieleman verfasserin aut Sabine E. Mous verfasserin aut Sabine E. Mous verfasserin aut Sabine E. Mous verfasserin aut ENCORE Expertise Center verfasserin aut Rianne Oostenbrink verfasserin aut Karen C.G.B. Bindels-de Heus verfasserin aut Marie-Claire Y. de Wit verfasserin aut Henriëtte A. Mol verfasserin aut Ype Elgersma verfasserin aut In Frontiers in Psychiatry Frontiers Media S.A., 2010 13(2022) (DE-627)631498796 (DE-600)2564218-2 16640640 nnns volume:13 year:2022 https://doi.org/10.3389/fpsyt.2022.852208 kostenfrei https://doaj.org/article/202fd10196a84dc78ef4070ab5f15967 kostenfrei https://www.frontiersin.org/articles/10.3389/fpsyt.2022.852208/full kostenfrei https://doaj.org/toc/1664-0640 Journal toc kostenfrei GBV_USEFLAG_A SYSFLAG_A GBV_DOAJ SSG-OLC-PHA GBV_ILN_20 GBV_ILN_22 GBV_ILN_23 GBV_ILN_24 GBV_ILN_31 GBV_ILN_39 GBV_ILN_40 GBV_ILN_60 GBV_ILN_62 GBV_ILN_63 GBV_ILN_65 GBV_ILN_69 GBV_ILN_73 GBV_ILN_74 GBV_ILN_95 GBV_ILN_105 GBV_ILN_110 GBV_ILN_151 GBV_ILN_161 GBV_ILN_170 GBV_ILN_206 GBV_ILN_213 GBV_ILN_230 GBV_ILN_285 GBV_ILN_293 GBV_ILN_602 GBV_ILN_2014 GBV_ILN_4012 GBV_ILN_4037 GBV_ILN_4112 GBV_ILN_4125 GBV_ILN_4126 GBV_ILN_4249 GBV_ILN_4305 GBV_ILN_4306 GBV_ILN_4307 GBV_ILN_4313 GBV_ILN_4322 GBV_ILN_4323 GBV_ILN_4324 GBV_ILN_4325 GBV_ILN_4338 GBV_ILN_4367 GBV_ILN_4700 AR 13 2022 |
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10.3389/fpsyt.2022.852208 doi (DE-627)DOAJ031247717 (DE-599)DOAJ202fd10196a84dc78ef4070ab5f15967 DE-627 ger DE-627 rakwb eng RC435-571 Kyra Lubbers verfasserin aut Autism Symptoms in Children and Young Adults With Fragile X Syndrome, Angelman Syndrome, Tuberous Sclerosis Complex, and Neurofibromatosis Type 1: A Cross-Syndrome Comparison 2022 Text txt rdacontent Computermedien c rdamedia Online-Ressource cr rdacarrier ObjectiveThe etiology of autism spectrum disorder (ASD) remains unclear, due to genetic heterogeneity and heterogeneity in symptoms across individuals. This study compares ASD symptomatology between monogenetic syndromes with a high ASD prevalence, in order to reveal syndrome specific vulnerabilities and to clarify how genetic variations affect ASD symptom presentation.MethodsWe assessed ASD symptom severity in children and young adults (aged 0-28 years) with Fragile X Syndrome (FXS, n = 60), Angelman Syndrome (AS, n = 91), Neurofibromatosis Type 1 (NF1, n = 279) and Tuberous Sclerosis Complex (TSC, n = 110), using the Autism Diagnostic Observation Schedule and Social Responsiveness Scale. Assessments were part of routine clinical care at the ENCORE expertise center in Rotterdam, the Netherlands. First, we compared the syndrome groups on the ASD classification prevalence and ASD severity scores. Then, we compared individuals in our syndrome groups with an ASD classification to a non-syndromic ASD group (nsASD, n = 335), on both ASD severity scores and ASD symptom profiles. Severity scores were compared using MANCOVAs with IQ and gender as covariates.ResultsOverall, ASD severity scores were highest for the FXS group and lowest for the NF1 group. Compared to nsASD, individuals with an ASD classification in our syndrome groups showed less problems on the instruments' social domains. We found a relative strength in the AS group on the social cognition, communication and motivation domains and a relative challenge in creativity; a relative strength of the NF1 group on the restricted interests and repetitive behavior scale; and a relative challenge in the FXS and TSC groups on the restricted interests and repetitive behavior domain.ConclusionThe syndrome-specific strengths and challenges we found provide a frame of reference to evaluate an individual's symptoms relative to the larger syndromic population and to guide treatment decisions. Our findings support the need for personalized care and a dimensional, symptom-based diagnostic approach, in contrast to a dichotomous ASD diagnosis used as a prerequisite for access to healthcare services. Similarities in ASD symptom profiles between AS and FXS, and between NF1 and TSC may reflect similarities in their neurobiology. Deep phenotyping studies are required to link neurobiological markers to ASD symptomatology. Fragile X Syndrome Angelman Syndrome Tuberous Sclerosis Complex Neurofibromatosis Type 1 autism spectrum disorder autistic traits Psychiatry Kyra Lubbers verfasserin aut Kyra Lubbers verfasserin aut Eefje M. Stijl verfasserin aut Eefje M. Stijl verfasserin aut Eefje M. Stijl verfasserin aut Bram Dierckx verfasserin aut Bram Dierckx verfasserin aut Bram Dierckx verfasserin aut Doesjka A. Hagenaar verfasserin aut Doesjka A. Hagenaar verfasserin aut Doesjka A. Hagenaar verfasserin aut Doesjka A. Hagenaar verfasserin aut Leontine W. ten Hoopen verfasserin aut Leontine W. ten Hoopen verfasserin aut Leontine W. ten Hoopen verfasserin aut Jeroen S. Legerstee verfasserin aut Jeroen S. Legerstee verfasserin aut Jeroen S. Legerstee verfasserin aut Pieter F. A. de Nijs verfasserin aut Pieter F. A. de Nijs verfasserin aut Pieter F. A. de Nijs verfasserin aut André B. Rietman verfasserin aut André B. Rietman verfasserin aut André B. Rietman verfasserin aut Kirstin Greaves-Lord verfasserin aut Kirstin Greaves-Lord verfasserin aut Kirstin Greaves-Lord verfasserin aut Kirstin Greaves-Lord verfasserin aut Manon H. J. Hillegers verfasserin aut Manon H. J. Hillegers verfasserin aut Manon H. J. Hillegers verfasserin aut Gwendolyn C. Dieleman verfasserin aut Gwendolyn C. Dieleman verfasserin aut Gwendolyn C. Dieleman verfasserin aut Sabine E. Mous verfasserin aut Sabine E. Mous verfasserin aut Sabine E. Mous verfasserin aut ENCORE Expertise Center verfasserin aut Rianne Oostenbrink verfasserin aut Karen C.G.B. Bindels-de Heus verfasserin aut Marie-Claire Y. de Wit verfasserin aut Henriëtte A. Mol verfasserin aut Ype Elgersma verfasserin aut In Frontiers in Psychiatry Frontiers Media S.A., 2010 13(2022) (DE-627)631498796 (DE-600)2564218-2 16640640 nnns volume:13 year:2022 https://doi.org/10.3389/fpsyt.2022.852208 kostenfrei https://doaj.org/article/202fd10196a84dc78ef4070ab5f15967 kostenfrei https://www.frontiersin.org/articles/10.3389/fpsyt.2022.852208/full kostenfrei https://doaj.org/toc/1664-0640 Journal toc kostenfrei GBV_USEFLAG_A SYSFLAG_A GBV_DOAJ SSG-OLC-PHA GBV_ILN_20 GBV_ILN_22 GBV_ILN_23 GBV_ILN_24 GBV_ILN_31 GBV_ILN_39 GBV_ILN_40 GBV_ILN_60 GBV_ILN_62 GBV_ILN_63 GBV_ILN_65 GBV_ILN_69 GBV_ILN_73 GBV_ILN_74 GBV_ILN_95 GBV_ILN_105 GBV_ILN_110 GBV_ILN_151 GBV_ILN_161 GBV_ILN_170 GBV_ILN_206 GBV_ILN_213 GBV_ILN_230 GBV_ILN_285 GBV_ILN_293 GBV_ILN_602 GBV_ILN_2014 GBV_ILN_4012 GBV_ILN_4037 GBV_ILN_4112 GBV_ILN_4125 GBV_ILN_4126 GBV_ILN_4249 GBV_ILN_4305 GBV_ILN_4306 GBV_ILN_4307 GBV_ILN_4313 GBV_ILN_4322 GBV_ILN_4323 GBV_ILN_4324 GBV_ILN_4325 GBV_ILN_4338 GBV_ILN_4367 GBV_ILN_4700 AR 13 2022 |
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10.3389/fpsyt.2022.852208 doi (DE-627)DOAJ031247717 (DE-599)DOAJ202fd10196a84dc78ef4070ab5f15967 DE-627 ger DE-627 rakwb eng RC435-571 Kyra Lubbers verfasserin aut Autism Symptoms in Children and Young Adults With Fragile X Syndrome, Angelman Syndrome, Tuberous Sclerosis Complex, and Neurofibromatosis Type 1: A Cross-Syndrome Comparison 2022 Text txt rdacontent Computermedien c rdamedia Online-Ressource cr rdacarrier ObjectiveThe etiology of autism spectrum disorder (ASD) remains unclear, due to genetic heterogeneity and heterogeneity in symptoms across individuals. This study compares ASD symptomatology between monogenetic syndromes with a high ASD prevalence, in order to reveal syndrome specific vulnerabilities and to clarify how genetic variations affect ASD symptom presentation.MethodsWe assessed ASD symptom severity in children and young adults (aged 0-28 years) with Fragile X Syndrome (FXS, n = 60), Angelman Syndrome (AS, n = 91), Neurofibromatosis Type 1 (NF1, n = 279) and Tuberous Sclerosis Complex (TSC, n = 110), using the Autism Diagnostic Observation Schedule and Social Responsiveness Scale. Assessments were part of routine clinical care at the ENCORE expertise center in Rotterdam, the Netherlands. First, we compared the syndrome groups on the ASD classification prevalence and ASD severity scores. Then, we compared individuals in our syndrome groups with an ASD classification to a non-syndromic ASD group (nsASD, n = 335), on both ASD severity scores and ASD symptom profiles. Severity scores were compared using MANCOVAs with IQ and gender as covariates.ResultsOverall, ASD severity scores were highest for the FXS group and lowest for the NF1 group. Compared to nsASD, individuals with an ASD classification in our syndrome groups showed less problems on the instruments' social domains. We found a relative strength in the AS group on the social cognition, communication and motivation domains and a relative challenge in creativity; a relative strength of the NF1 group on the restricted interests and repetitive behavior scale; and a relative challenge in the FXS and TSC groups on the restricted interests and repetitive behavior domain.ConclusionThe syndrome-specific strengths and challenges we found provide a frame of reference to evaluate an individual's symptoms relative to the larger syndromic population and to guide treatment decisions. Our findings support the need for personalized care and a dimensional, symptom-based diagnostic approach, in contrast to a dichotomous ASD diagnosis used as a prerequisite for access to healthcare services. Similarities in ASD symptom profiles between AS and FXS, and between NF1 and TSC may reflect similarities in their neurobiology. Deep phenotyping studies are required to link neurobiological markers to ASD symptomatology. Fragile X Syndrome Angelman Syndrome Tuberous Sclerosis Complex Neurofibromatosis Type 1 autism spectrum disorder autistic traits Psychiatry Kyra Lubbers verfasserin aut Kyra Lubbers verfasserin aut Eefje M. Stijl verfasserin aut Eefje M. Stijl verfasserin aut Eefje M. Stijl verfasserin aut Bram Dierckx verfasserin aut Bram Dierckx verfasserin aut Bram Dierckx verfasserin aut Doesjka A. Hagenaar verfasserin aut Doesjka A. Hagenaar verfasserin aut Doesjka A. Hagenaar verfasserin aut Doesjka A. Hagenaar verfasserin aut Leontine W. ten Hoopen verfasserin aut Leontine W. ten Hoopen verfasserin aut Leontine W. ten Hoopen verfasserin aut Jeroen S. Legerstee verfasserin aut Jeroen S. Legerstee verfasserin aut Jeroen S. Legerstee verfasserin aut Pieter F. A. de Nijs verfasserin aut Pieter F. A. de Nijs verfasserin aut Pieter F. A. de Nijs verfasserin aut André B. Rietman verfasserin aut André B. Rietman verfasserin aut André B. Rietman verfasserin aut Kirstin Greaves-Lord verfasserin aut Kirstin Greaves-Lord verfasserin aut Kirstin Greaves-Lord verfasserin aut Kirstin Greaves-Lord verfasserin aut Manon H. J. Hillegers verfasserin aut Manon H. J. Hillegers verfasserin aut Manon H. J. Hillegers verfasserin aut Gwendolyn C. Dieleman verfasserin aut Gwendolyn C. Dieleman verfasserin aut Gwendolyn C. Dieleman verfasserin aut Sabine E. Mous verfasserin aut Sabine E. Mous verfasserin aut Sabine E. Mous verfasserin aut ENCORE Expertise Center verfasserin aut Rianne Oostenbrink verfasserin aut Karen C.G.B. Bindels-de Heus verfasserin aut Marie-Claire Y. de Wit verfasserin aut Henriëtte A. Mol verfasserin aut Ype Elgersma verfasserin aut In Frontiers in Psychiatry Frontiers Media S.A., 2010 13(2022) (DE-627)631498796 (DE-600)2564218-2 16640640 nnns volume:13 year:2022 https://doi.org/10.3389/fpsyt.2022.852208 kostenfrei https://doaj.org/article/202fd10196a84dc78ef4070ab5f15967 kostenfrei https://www.frontiersin.org/articles/10.3389/fpsyt.2022.852208/full kostenfrei https://doaj.org/toc/1664-0640 Journal toc kostenfrei GBV_USEFLAG_A SYSFLAG_A GBV_DOAJ SSG-OLC-PHA GBV_ILN_20 GBV_ILN_22 GBV_ILN_23 GBV_ILN_24 GBV_ILN_31 GBV_ILN_39 GBV_ILN_40 GBV_ILN_60 GBV_ILN_62 GBV_ILN_63 GBV_ILN_65 GBV_ILN_69 GBV_ILN_73 GBV_ILN_74 GBV_ILN_95 GBV_ILN_105 GBV_ILN_110 GBV_ILN_151 GBV_ILN_161 GBV_ILN_170 GBV_ILN_206 GBV_ILN_213 GBV_ILN_230 GBV_ILN_285 GBV_ILN_293 GBV_ILN_602 GBV_ILN_2014 GBV_ILN_4012 GBV_ILN_4037 GBV_ILN_4112 GBV_ILN_4125 GBV_ILN_4126 GBV_ILN_4249 GBV_ILN_4305 GBV_ILN_4306 GBV_ILN_4307 GBV_ILN_4313 GBV_ILN_4322 GBV_ILN_4323 GBV_ILN_4324 GBV_ILN_4325 GBV_ILN_4338 GBV_ILN_4367 GBV_ILN_4700 AR 13 2022 |
| allfieldsSound |
10.3389/fpsyt.2022.852208 doi (DE-627)DOAJ031247717 (DE-599)DOAJ202fd10196a84dc78ef4070ab5f15967 DE-627 ger DE-627 rakwb eng RC435-571 Kyra Lubbers verfasserin aut Autism Symptoms in Children and Young Adults With Fragile X Syndrome, Angelman Syndrome, Tuberous Sclerosis Complex, and Neurofibromatosis Type 1: A Cross-Syndrome Comparison 2022 Text txt rdacontent Computermedien c rdamedia Online-Ressource cr rdacarrier ObjectiveThe etiology of autism spectrum disorder (ASD) remains unclear, due to genetic heterogeneity and heterogeneity in symptoms across individuals. This study compares ASD symptomatology between monogenetic syndromes with a high ASD prevalence, in order to reveal syndrome specific vulnerabilities and to clarify how genetic variations affect ASD symptom presentation.MethodsWe assessed ASD symptom severity in children and young adults (aged 0-28 years) with Fragile X Syndrome (FXS, n = 60), Angelman Syndrome (AS, n = 91), Neurofibromatosis Type 1 (NF1, n = 279) and Tuberous Sclerosis Complex (TSC, n = 110), using the Autism Diagnostic Observation Schedule and Social Responsiveness Scale. Assessments were part of routine clinical care at the ENCORE expertise center in Rotterdam, the Netherlands. First, we compared the syndrome groups on the ASD classification prevalence and ASD severity scores. Then, we compared individuals in our syndrome groups with an ASD classification to a non-syndromic ASD group (nsASD, n = 335), on both ASD severity scores and ASD symptom profiles. Severity scores were compared using MANCOVAs with IQ and gender as covariates.ResultsOverall, ASD severity scores were highest for the FXS group and lowest for the NF1 group. Compared to nsASD, individuals with an ASD classification in our syndrome groups showed less problems on the instruments' social domains. We found a relative strength in the AS group on the social cognition, communication and motivation domains and a relative challenge in creativity; a relative strength of the NF1 group on the restricted interests and repetitive behavior scale; and a relative challenge in the FXS and TSC groups on the restricted interests and repetitive behavior domain.ConclusionThe syndrome-specific strengths and challenges we found provide a frame of reference to evaluate an individual's symptoms relative to the larger syndromic population and to guide treatment decisions. Our findings support the need for personalized care and a dimensional, symptom-based diagnostic approach, in contrast to a dichotomous ASD diagnosis used as a prerequisite for access to healthcare services. Similarities in ASD symptom profiles between AS and FXS, and between NF1 and TSC may reflect similarities in their neurobiology. Deep phenotyping studies are required to link neurobiological markers to ASD symptomatology. Fragile X Syndrome Angelman Syndrome Tuberous Sclerosis Complex Neurofibromatosis Type 1 autism spectrum disorder autistic traits Psychiatry Kyra Lubbers verfasserin aut Kyra Lubbers verfasserin aut Eefje M. Stijl verfasserin aut Eefje M. Stijl verfasserin aut Eefje M. Stijl verfasserin aut Bram Dierckx verfasserin aut Bram Dierckx verfasserin aut Bram Dierckx verfasserin aut Doesjka A. Hagenaar verfasserin aut Doesjka A. Hagenaar verfasserin aut Doesjka A. Hagenaar verfasserin aut Doesjka A. Hagenaar verfasserin aut Leontine W. ten Hoopen verfasserin aut Leontine W. ten Hoopen verfasserin aut Leontine W. ten Hoopen verfasserin aut Jeroen S. Legerstee verfasserin aut Jeroen S. Legerstee verfasserin aut Jeroen S. Legerstee verfasserin aut Pieter F. A. de Nijs verfasserin aut Pieter F. A. de Nijs verfasserin aut Pieter F. A. de Nijs verfasserin aut André B. Rietman verfasserin aut André B. Rietman verfasserin aut André B. Rietman verfasserin aut Kirstin Greaves-Lord verfasserin aut Kirstin Greaves-Lord verfasserin aut Kirstin Greaves-Lord verfasserin aut Kirstin Greaves-Lord verfasserin aut Manon H. J. Hillegers verfasserin aut Manon H. J. Hillegers verfasserin aut Manon H. J. Hillegers verfasserin aut Gwendolyn C. Dieleman verfasserin aut Gwendolyn C. Dieleman verfasserin aut Gwendolyn C. Dieleman verfasserin aut Sabine E. Mous verfasserin aut Sabine E. Mous verfasserin aut Sabine E. Mous verfasserin aut ENCORE Expertise Center verfasserin aut Rianne Oostenbrink verfasserin aut Karen C.G.B. Bindels-de Heus verfasserin aut Marie-Claire Y. de Wit verfasserin aut Henriëtte A. Mol verfasserin aut Ype Elgersma verfasserin aut In Frontiers in Psychiatry Frontiers Media S.A., 2010 13(2022) (DE-627)631498796 (DE-600)2564218-2 16640640 nnns volume:13 year:2022 https://doi.org/10.3389/fpsyt.2022.852208 kostenfrei https://doaj.org/article/202fd10196a84dc78ef4070ab5f15967 kostenfrei https://www.frontiersin.org/articles/10.3389/fpsyt.2022.852208/full kostenfrei https://doaj.org/toc/1664-0640 Journal toc kostenfrei GBV_USEFLAG_A SYSFLAG_A GBV_DOAJ SSG-OLC-PHA GBV_ILN_20 GBV_ILN_22 GBV_ILN_23 GBV_ILN_24 GBV_ILN_31 GBV_ILN_39 GBV_ILN_40 GBV_ILN_60 GBV_ILN_62 GBV_ILN_63 GBV_ILN_65 GBV_ILN_69 GBV_ILN_73 GBV_ILN_74 GBV_ILN_95 GBV_ILN_105 GBV_ILN_110 GBV_ILN_151 GBV_ILN_161 GBV_ILN_170 GBV_ILN_206 GBV_ILN_213 GBV_ILN_230 GBV_ILN_285 GBV_ILN_293 GBV_ILN_602 GBV_ILN_2014 GBV_ILN_4012 GBV_ILN_4037 GBV_ILN_4112 GBV_ILN_4125 GBV_ILN_4126 GBV_ILN_4249 GBV_ILN_4305 GBV_ILN_4306 GBV_ILN_4307 GBV_ILN_4313 GBV_ILN_4322 GBV_ILN_4323 GBV_ILN_4324 GBV_ILN_4325 GBV_ILN_4338 GBV_ILN_4367 GBV_ILN_4700 AR 13 2022 |
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Kyra Lubbers @@aut@@ Eefje M. Stijl @@aut@@ Bram Dierckx @@aut@@ Doesjka A. Hagenaar @@aut@@ Leontine W. ten Hoopen @@aut@@ Jeroen S. Legerstee @@aut@@ Pieter F. A. de Nijs @@aut@@ André B. Rietman @@aut@@ Kirstin Greaves-Lord @@aut@@ Manon H. J. Hillegers @@aut@@ Gwendolyn C. Dieleman @@aut@@ Sabine E. Mous @@aut@@ ENCORE Expertise Center @@aut@@ Rianne Oostenbrink @@aut@@ Karen C.G.B. Bindels-de Heus @@aut@@ Marie-Claire Y. de Wit @@aut@@ Henriëtte A. Mol @@aut@@ Ype Elgersma @@aut@@ |
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<?xml version="1.0" encoding="UTF-8"?><collection xmlns="http://www.loc.gov/MARC21/slim"><record><leader>01000caa a22002652 4500</leader><controlfield tag="001">DOAJ031247717</controlfield><controlfield tag="003">DE-627</controlfield><controlfield tag="005">20230501193915.0</controlfield><controlfield tag="007">cr uuu---uuuuu</controlfield><controlfield tag="008">230226s2022 xx |||||o 00| ||eng c</controlfield><datafield tag="024" ind1="7" ind2=" "><subfield code="a">10.3389/fpsyt.2022.852208</subfield><subfield code="2">doi</subfield></datafield><datafield tag="035" ind1=" " ind2=" "><subfield code="a">(DE-627)DOAJ031247717</subfield></datafield><datafield tag="035" ind1=" " ind2=" "><subfield code="a">(DE-599)DOAJ202fd10196a84dc78ef4070ab5f15967</subfield></datafield><datafield tag="040" ind1=" " ind2=" "><subfield code="a">DE-627</subfield><subfield code="b">ger</subfield><subfield code="c">DE-627</subfield><subfield code="e">rakwb</subfield></datafield><datafield tag="041" ind1=" " ind2=" "><subfield code="a">eng</subfield></datafield><datafield tag="050" ind1=" " ind2="0"><subfield code="a">RC435-571</subfield></datafield><datafield tag="100" ind1="0" ind2=" "><subfield code="a">Kyra Lubbers</subfield><subfield code="e">verfasserin</subfield><subfield code="4">aut</subfield></datafield><datafield tag="245" ind1="1" ind2="0"><subfield code="a">Autism Symptoms in Children and Young Adults With Fragile X Syndrome, Angelman Syndrome, Tuberous Sclerosis Complex, and Neurofibromatosis Type 1: A Cross-Syndrome Comparison</subfield></datafield><datafield tag="264" ind1=" " ind2="1"><subfield code="c">2022</subfield></datafield><datafield tag="336" ind1=" " ind2=" "><subfield code="a">Text</subfield><subfield code="b">txt</subfield><subfield code="2">rdacontent</subfield></datafield><datafield tag="337" ind1=" " ind2=" "><subfield code="a">Computermedien</subfield><subfield code="b">c</subfield><subfield code="2">rdamedia</subfield></datafield><datafield tag="338" ind1=" " ind2=" "><subfield code="a">Online-Ressource</subfield><subfield code="b">cr</subfield><subfield code="2">rdacarrier</subfield></datafield><datafield tag="520" ind1=" " ind2=" "><subfield code="a">ObjectiveThe etiology of autism spectrum disorder (ASD) remains unclear, due to genetic heterogeneity and heterogeneity in symptoms across individuals. This study compares ASD symptomatology between monogenetic syndromes with a high ASD prevalence, in order to reveal syndrome specific vulnerabilities and to clarify how genetic variations affect ASD symptom presentation.MethodsWe assessed ASD symptom severity in children and young adults (aged 0-28 years) with Fragile X Syndrome (FXS, n = 60), Angelman Syndrome (AS, n = 91), Neurofibromatosis Type 1 (NF1, n = 279) and Tuberous Sclerosis Complex (TSC, n = 110), using the Autism Diagnostic Observation Schedule and Social Responsiveness Scale. Assessments were part of routine clinical care at the ENCORE expertise center in Rotterdam, the Netherlands. First, we compared the syndrome groups on the ASD classification prevalence and ASD severity scores. Then, we compared individuals in our syndrome groups with an ASD classification to a non-syndromic ASD group (nsASD, n = 335), on both ASD severity scores and ASD symptom profiles. Severity scores were compared using MANCOVAs with IQ and gender as covariates.ResultsOverall, ASD severity scores were highest for the FXS group and lowest for the NF1 group. Compared to nsASD, individuals with an ASD classification in our syndrome groups showed less problems on the instruments' social domains. We found a relative strength in the AS group on the social cognition, communication and motivation domains and a relative challenge in creativity; a relative strength of the NF1 group on the restricted interests and repetitive behavior scale; and a relative challenge in the FXS and TSC groups on the restricted interests and repetitive behavior domain.ConclusionThe syndrome-specific strengths and challenges we found provide a frame of reference to evaluate an individual's symptoms relative to the larger syndromic population and to guide treatment decisions. Our findings support the need for personalized care and a dimensional, symptom-based diagnostic approach, in contrast to a dichotomous ASD diagnosis used as a prerequisite for access to healthcare services. Similarities in ASD symptom profiles between AS and FXS, and between NF1 and TSC may reflect similarities in their neurobiology. 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Stijl</subfield><subfield code="e">verfasserin</subfield><subfield code="4">aut</subfield></datafield><datafield tag="700" ind1="0" ind2=" "><subfield code="a">Eefje M. Stijl</subfield><subfield code="e">verfasserin</subfield><subfield code="4">aut</subfield></datafield><datafield tag="700" ind1="0" ind2=" "><subfield code="a">Eefje M. 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Kyra Lubbers misc RC435-571 misc Fragile X Syndrome misc Angelman Syndrome misc Tuberous Sclerosis Complex misc Neurofibromatosis Type 1 misc autism spectrum disorder misc autistic traits misc Psychiatry Autism Symptoms in Children and Young Adults With Fragile X Syndrome, Angelman Syndrome, Tuberous Sclerosis Complex, and Neurofibromatosis Type 1: A Cross-Syndrome Comparison |
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RC435-571 Autism Symptoms in Children and Young Adults With Fragile X Syndrome, Angelman Syndrome, Tuberous Sclerosis Complex, and Neurofibromatosis Type 1: A Cross-Syndrome Comparison Fragile X Syndrome Angelman Syndrome Tuberous Sclerosis Complex Neurofibromatosis Type 1 autism spectrum disorder autistic traits |
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Kyra Lubbers Eefje M. Stijl Bram Dierckx Doesjka A. Hagenaar Leontine W. ten Hoopen Jeroen S. Legerstee Pieter F. A. de Nijs André B. Rietman Kirstin Greaves-Lord Manon H. J. Hillegers Gwendolyn C. Dieleman Sabine E. Mous ENCORE Expertise Center Rianne Oostenbrink Karen C.G.B. Bindels-de Heus Marie-Claire Y. de Wit Henriëtte A. Mol Ype Elgersma |
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10.3389/fpsyt.2022.852208 |
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autism symptoms in children and young adults with fragile x syndrome, angelman syndrome, tuberous sclerosis complex, and neurofibromatosis type 1: a cross-syndrome comparison |
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RC435-571 |
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Autism Symptoms in Children and Young Adults With Fragile X Syndrome, Angelman Syndrome, Tuberous Sclerosis Complex, and Neurofibromatosis Type 1: A Cross-Syndrome Comparison |
| abstract |
ObjectiveThe etiology of autism spectrum disorder (ASD) remains unclear, due to genetic heterogeneity and heterogeneity in symptoms across individuals. This study compares ASD symptomatology between monogenetic syndromes with a high ASD prevalence, in order to reveal syndrome specific vulnerabilities and to clarify how genetic variations affect ASD symptom presentation.MethodsWe assessed ASD symptom severity in children and young adults (aged 0-28 years) with Fragile X Syndrome (FXS, n = 60), Angelman Syndrome (AS, n = 91), Neurofibromatosis Type 1 (NF1, n = 279) and Tuberous Sclerosis Complex (TSC, n = 110), using the Autism Diagnostic Observation Schedule and Social Responsiveness Scale. Assessments were part of routine clinical care at the ENCORE expertise center in Rotterdam, the Netherlands. First, we compared the syndrome groups on the ASD classification prevalence and ASD severity scores. Then, we compared individuals in our syndrome groups with an ASD classification to a non-syndromic ASD group (nsASD, n = 335), on both ASD severity scores and ASD symptom profiles. Severity scores were compared using MANCOVAs with IQ and gender as covariates.ResultsOverall, ASD severity scores were highest for the FXS group and lowest for the NF1 group. Compared to nsASD, individuals with an ASD classification in our syndrome groups showed less problems on the instruments' social domains. We found a relative strength in the AS group on the social cognition, communication and motivation domains and a relative challenge in creativity; a relative strength of the NF1 group on the restricted interests and repetitive behavior scale; and a relative challenge in the FXS and TSC groups on the restricted interests and repetitive behavior domain.ConclusionThe syndrome-specific strengths and challenges we found provide a frame of reference to evaluate an individual's symptoms relative to the larger syndromic population and to guide treatment decisions. Our findings support the need for personalized care and a dimensional, symptom-based diagnostic approach, in contrast to a dichotomous ASD diagnosis used as a prerequisite for access to healthcare services. Similarities in ASD symptom profiles between AS and FXS, and between NF1 and TSC may reflect similarities in their neurobiology. Deep phenotyping studies are required to link neurobiological markers to ASD symptomatology. |
| abstractGer |
ObjectiveThe etiology of autism spectrum disorder (ASD) remains unclear, due to genetic heterogeneity and heterogeneity in symptoms across individuals. This study compares ASD symptomatology between monogenetic syndromes with a high ASD prevalence, in order to reveal syndrome specific vulnerabilities and to clarify how genetic variations affect ASD symptom presentation.MethodsWe assessed ASD symptom severity in children and young adults (aged 0-28 years) with Fragile X Syndrome (FXS, n = 60), Angelman Syndrome (AS, n = 91), Neurofibromatosis Type 1 (NF1, n = 279) and Tuberous Sclerosis Complex (TSC, n = 110), using the Autism Diagnostic Observation Schedule and Social Responsiveness Scale. Assessments were part of routine clinical care at the ENCORE expertise center in Rotterdam, the Netherlands. First, we compared the syndrome groups on the ASD classification prevalence and ASD severity scores. Then, we compared individuals in our syndrome groups with an ASD classification to a non-syndromic ASD group (nsASD, n = 335), on both ASD severity scores and ASD symptom profiles. Severity scores were compared using MANCOVAs with IQ and gender as covariates.ResultsOverall, ASD severity scores were highest for the FXS group and lowest for the NF1 group. Compared to nsASD, individuals with an ASD classification in our syndrome groups showed less problems on the instruments' social domains. We found a relative strength in the AS group on the social cognition, communication and motivation domains and a relative challenge in creativity; a relative strength of the NF1 group on the restricted interests and repetitive behavior scale; and a relative challenge in the FXS and TSC groups on the restricted interests and repetitive behavior domain.ConclusionThe syndrome-specific strengths and challenges we found provide a frame of reference to evaluate an individual's symptoms relative to the larger syndromic population and to guide treatment decisions. Our findings support the need for personalized care and a dimensional, symptom-based diagnostic approach, in contrast to a dichotomous ASD diagnosis used as a prerequisite for access to healthcare services. Similarities in ASD symptom profiles between AS and FXS, and between NF1 and TSC may reflect similarities in their neurobiology. Deep phenotyping studies are required to link neurobiological markers to ASD symptomatology. |
| abstract_unstemmed |
ObjectiveThe etiology of autism spectrum disorder (ASD) remains unclear, due to genetic heterogeneity and heterogeneity in symptoms across individuals. This study compares ASD symptomatology between monogenetic syndromes with a high ASD prevalence, in order to reveal syndrome specific vulnerabilities and to clarify how genetic variations affect ASD symptom presentation.MethodsWe assessed ASD symptom severity in children and young adults (aged 0-28 years) with Fragile X Syndrome (FXS, n = 60), Angelman Syndrome (AS, n = 91), Neurofibromatosis Type 1 (NF1, n = 279) and Tuberous Sclerosis Complex (TSC, n = 110), using the Autism Diagnostic Observation Schedule and Social Responsiveness Scale. Assessments were part of routine clinical care at the ENCORE expertise center in Rotterdam, the Netherlands. First, we compared the syndrome groups on the ASD classification prevalence and ASD severity scores. Then, we compared individuals in our syndrome groups with an ASD classification to a non-syndromic ASD group (nsASD, n = 335), on both ASD severity scores and ASD symptom profiles. Severity scores were compared using MANCOVAs with IQ and gender as covariates.ResultsOverall, ASD severity scores were highest for the FXS group and lowest for the NF1 group. Compared to nsASD, individuals with an ASD classification in our syndrome groups showed less problems on the instruments' social domains. We found a relative strength in the AS group on the social cognition, communication and motivation domains and a relative challenge in creativity; a relative strength of the NF1 group on the restricted interests and repetitive behavior scale; and a relative challenge in the FXS and TSC groups on the restricted interests and repetitive behavior domain.ConclusionThe syndrome-specific strengths and challenges we found provide a frame of reference to evaluate an individual's symptoms relative to the larger syndromic population and to guide treatment decisions. Our findings support the need for personalized care and a dimensional, symptom-based diagnostic approach, in contrast to a dichotomous ASD diagnosis used as a prerequisite for access to healthcare services. Similarities in ASD symptom profiles between AS and FXS, and between NF1 and TSC may reflect similarities in their neurobiology. Deep phenotyping studies are required to link neurobiological markers to ASD symptomatology. |
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<?xml version="1.0" encoding="UTF-8"?><collection xmlns="http://www.loc.gov/MARC21/slim"><record><leader>01000caa a22002652 4500</leader><controlfield tag="001">DOAJ031247717</controlfield><controlfield tag="003">DE-627</controlfield><controlfield tag="005">20230501193915.0</controlfield><controlfield tag="007">cr uuu---uuuuu</controlfield><controlfield tag="008">230226s2022 xx |||||o 00| ||eng c</controlfield><datafield tag="024" ind1="7" ind2=" "><subfield code="a">10.3389/fpsyt.2022.852208</subfield><subfield code="2">doi</subfield></datafield><datafield tag="035" ind1=" " ind2=" "><subfield code="a">(DE-627)DOAJ031247717</subfield></datafield><datafield tag="035" ind1=" " ind2=" "><subfield code="a">(DE-599)DOAJ202fd10196a84dc78ef4070ab5f15967</subfield></datafield><datafield tag="040" ind1=" " ind2=" "><subfield code="a">DE-627</subfield><subfield code="b">ger</subfield><subfield code="c">DE-627</subfield><subfield code="e">rakwb</subfield></datafield><datafield tag="041" ind1=" " ind2=" "><subfield code="a">eng</subfield></datafield><datafield tag="050" ind1=" " ind2="0"><subfield code="a">RC435-571</subfield></datafield><datafield tag="100" ind1="0" ind2=" "><subfield code="a">Kyra Lubbers</subfield><subfield code="e">verfasserin</subfield><subfield code="4">aut</subfield></datafield><datafield tag="245" ind1="1" ind2="0"><subfield code="a">Autism Symptoms in Children and Young Adults With Fragile X Syndrome, Angelman Syndrome, Tuberous Sclerosis Complex, and Neurofibromatosis Type 1: A Cross-Syndrome Comparison</subfield></datafield><datafield tag="264" ind1=" " ind2="1"><subfield code="c">2022</subfield></datafield><datafield tag="336" ind1=" " ind2=" "><subfield code="a">Text</subfield><subfield code="b">txt</subfield><subfield code="2">rdacontent</subfield></datafield><datafield tag="337" ind1=" " ind2=" "><subfield code="a">Computermedien</subfield><subfield code="b">c</subfield><subfield code="2">rdamedia</subfield></datafield><datafield tag="338" ind1=" " ind2=" "><subfield code="a">Online-Ressource</subfield><subfield code="b">cr</subfield><subfield code="2">rdacarrier</subfield></datafield><datafield tag="520" ind1=" " ind2=" "><subfield code="a">ObjectiveThe etiology of autism spectrum disorder (ASD) remains unclear, due to genetic heterogeneity and heterogeneity in symptoms across individuals. This study compares ASD symptomatology between monogenetic syndromes with a high ASD prevalence, in order to reveal syndrome specific vulnerabilities and to clarify how genetic variations affect ASD symptom presentation.MethodsWe assessed ASD symptom severity in children and young adults (aged 0-28 years) with Fragile X Syndrome (FXS, n = 60), Angelman Syndrome (AS, n = 91), Neurofibromatosis Type 1 (NF1, n = 279) and Tuberous Sclerosis Complex (TSC, n = 110), using the Autism Diagnostic Observation Schedule and Social Responsiveness Scale. Assessments were part of routine clinical care at the ENCORE expertise center in Rotterdam, the Netherlands. First, we compared the syndrome groups on the ASD classification prevalence and ASD severity scores. Then, we compared individuals in our syndrome groups with an ASD classification to a non-syndromic ASD group (nsASD, n = 335), on both ASD severity scores and ASD symptom profiles. Severity scores were compared using MANCOVAs with IQ and gender as covariates.ResultsOverall, ASD severity scores were highest for the FXS group and lowest for the NF1 group. Compared to nsASD, individuals with an ASD classification in our syndrome groups showed less problems on the instruments' social domains. We found a relative strength in the AS group on the social cognition, communication and motivation domains and a relative challenge in creativity; a relative strength of the NF1 group on the restricted interests and repetitive behavior scale; and a relative challenge in the FXS and TSC groups on the restricted interests and repetitive behavior domain.ConclusionThe syndrome-specific strengths and challenges we found provide a frame of reference to evaluate an individual's symptoms relative to the larger syndromic population and to guide treatment decisions. Our findings support the need for personalized care and a dimensional, symptom-based diagnostic approach, in contrast to a dichotomous ASD diagnosis used as a prerequisite for access to healthcare services. Similarities in ASD symptom profiles between AS and FXS, and between NF1 and TSC may reflect similarities in their neurobiology. Deep phenotyping studies are required to link neurobiological markers to ASD symptomatology.</subfield></datafield><datafield tag="650" ind1=" " ind2="4"><subfield code="a">Fragile X Syndrome</subfield></datafield><datafield tag="650" ind1=" " ind2="4"><subfield code="a">Angelman Syndrome</subfield></datafield><datafield tag="650" ind1=" " ind2="4"><subfield code="a">Tuberous Sclerosis Complex</subfield></datafield><datafield tag="650" ind1=" " ind2="4"><subfield code="a">Neurofibromatosis Type 1</subfield></datafield><datafield tag="650" ind1=" " ind2="4"><subfield code="a">autism spectrum disorder</subfield></datafield><datafield tag="650" ind1=" " ind2="4"><subfield code="a">autistic traits</subfield></datafield><datafield tag="653" ind1=" " ind2="0"><subfield code="a">Psychiatry</subfield></datafield><datafield tag="700" ind1="0" ind2=" "><subfield code="a">Kyra Lubbers</subfield><subfield code="e">verfasserin</subfield><subfield code="4">aut</subfield></datafield><datafield tag="700" ind1="0" ind2=" "><subfield code="a">Kyra Lubbers</subfield><subfield code="e">verfasserin</subfield><subfield code="4">aut</subfield></datafield><datafield tag="700" ind1="0" ind2=" "><subfield code="a">Eefje M. 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