M. Bluett-Duncan , D. Astill , R. Charbak , J. Clayton-Smith , S. Cole , P.A. Cook , J. Cozens , K. Keely , J. Morris , R. Mukherjee , E. Murphy , P. Turnpenny , J. Williams , A.G. Wood , L.M. Yates , R.L. Bromley
{"title":"胎儿丙戊酸钠谱系障碍儿童和成人的神经发育结果：ConcePTION项目的贡献","authors":"M. Bluett-Duncan , D. Astill , R. Charbak , J. Clayton-Smith , S. Cole , P.A. Cook , J. Cozens , K. Keely , J. Morris , R. Mukherjee , E. Murphy , P. Turnpenny , J. Williams , A.G. Wood , L.M. Yates , R.L. Bromley","doi":"10.1016/j.ntt.2023.107292","DOIUrl":null,"url":null,"abstract":"<div><h3>Aim</h3><p>To describe the neurodevelopmental phenotype of older children and adults with a diagnosis of Fetal Valproate Spectrum Disorder (FVSD).</p></div><div><h3>Methods</h3><p>In this cross-sectional study, 90 caregivers were recruited and completed a series of questionnaires regarding the neurodevelopmental outcomes of 146 individuals aged 7–37 years (M = 18.1), including individuals with a formal diagnosis of FVSD (<em>n</em> = 99), individuals exposed to Valproate but without an FVSD diagnosis (<em>n</em> = 24), and individuals not exposed to Valproate (<em>N</em> = 23). The mean dose of valproate exposure for individuals with an FVSD diagnosis was 1470 mg/day.</p></div><div><h3>Results</h3><p>Individuals with a diagnosis of FVSD showed significantly higher levels of moderate (43.4%) and severe (14.4%) cognitive impairment than other groups (<em>p</em> = 0.003), high levels of required formal educational support (77.6%), and poorer academic competence than individuals not exposed to Valproate (<em>p</em> = 0.001). Overall psychosocial problems (<em>p =</em> 0.02), internalising problems (<em>p</em> = 0.05) and attention problems (<em>p</em> = 0.001), but not externalising problems, were elevated in individuals with a diagnosis of FVSD. Rates of neurodevelopmental disorders, particularly autistic spectrum disorders (62.9%) and sensory problems (80.6%) are particularly central to the FVSD phenotype. There was no evidence of a statistical dose-dependent effect, possibly due to the high mean dose of exposure having a uniformly negative impact across the sample. Individuals with FVSD had required a significant number of health and child development services.</p></div><div><h3>Interpretation</h3><p>Children and young adults with a diagnosis of FVSD are at an increased risk of a range of altered neurodevelopmental outcomes, highlighting the need for a multidisciplinary approach to clinical management across the lifespan.</p></div>","PeriodicalId":19144,"journal":{"name":"Neurotoxicology and teratology","volume":"100 ","pages":"Article 107292"},"PeriodicalIF":2.6000,"publicationDate":"2023-09-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Neurodevelopmental outcomes in children and adults with Fetal Valproate Spectrum Disorder: A contribution from the ConcePTION project\",\"authors\":\"M. Bluett-Duncan , D. Astill , R. Charbak , J. Clayton-Smith , S. Cole , P.A. Cook , J. Cozens , K. Keely , J. Morris , R. Mukherjee , E. Murphy , P. Turnpenny , J. Williams , A.G. Wood , L.M. Yates , R.L. Bromley\",\"doi\":\"10.1016/j.ntt.2023.107292\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<div><h3>Aim</h3><p>To describe the neurodevelopmental phenotype of older children and adults with a diagnosis of Fetal Valproate Spectrum Disorder (FVSD).</p></div><div><h3>Methods</h3><p>In this cross-sectional study, 90 caregivers were recruited and completed a series of questionnaires regarding the neurodevelopmental outcomes of 146 individuals aged 7–37 years (M = 18.1), including individuals with a formal diagnosis of FVSD (<em>n</em> = 99), individuals exposed to Valproate but without an FVSD diagnosis (<em>n</em> = 24), and individuals not exposed to Valproate (<em>N</em> = 23). The mean dose of valproate exposure for individuals with an FVSD diagnosis was 1470 mg/day.</p></div><div><h3>Results</h3><p>Individuals with a diagnosis of FVSD showed significantly higher levels of moderate (43.4%) and severe (14.4%) cognitive impairment than other groups (<em>p</em> = 0.003), high levels of required formal educational support (77.6%), and poorer academic competence than individuals not exposed to Valproate (<em>p</em> = 0.001). Overall psychosocial problems (<em>p =</em> 0.02), internalising problems (<em>p</em> = 0.05) and attention problems (<em>p</em> = 0.001), but not externalising problems, were elevated in individuals with a diagnosis of FVSD. Rates of neurodevelopmental disorders, particularly autistic spectrum disorders (62.9%) and sensory problems (80.6%) are particularly central to the FVSD phenotype. There was no evidence of a statistical dose-dependent effect, possibly due to the high mean dose of exposure having a uniformly negative impact across the sample. Individuals with FVSD had required a significant number of health and child development services.</p></div><div><h3>Interpretation</h3><p>Children and young adults with a diagnosis of FVSD are at an increased risk of a range of altered neurodevelopmental outcomes, highlighting the need for a multidisciplinary approach to clinical management across the lifespan.</p></div>\",\"PeriodicalId\":19144,\"journal\":{\"name\":\"Neurotoxicology and teratology\",\"volume\":\"100 \",\"pages\":\"Article 107292\"},\"PeriodicalIF\":2.6000,\"publicationDate\":\"2023-09-04\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Neurotoxicology and teratology\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://www.sciencedirect.com/science/article/pii/S0892036223001423\",\"RegionNum\":3,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q3\",\"JCRName\":\"NEUROSCIENCES\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Neurotoxicology and teratology","FirstCategoryId":"3","ListUrlMain":"https://www.sciencedirect.com/science/article/pii/S0892036223001423","RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"NEUROSCIENCES","Score":null,"Total":0}
Neurodevelopmental outcomes in children and adults with Fetal Valproate Spectrum Disorder: A contribution from the ConcePTION project
Aim
To describe the neurodevelopmental phenotype of older children and adults with a diagnosis of Fetal Valproate Spectrum Disorder (FVSD).
Methods
In this cross-sectional study, 90 caregivers were recruited and completed a series of questionnaires regarding the neurodevelopmental outcomes of 146 individuals aged 7–37 years (M = 18.1), including individuals with a formal diagnosis of FVSD (n = 99), individuals exposed to Valproate but without an FVSD diagnosis (n = 24), and individuals not exposed to Valproate (N = 23). The mean dose of valproate exposure for individuals with an FVSD diagnosis was 1470 mg/day.
Results
Individuals with a diagnosis of FVSD showed significantly higher levels of moderate (43.4%) and severe (14.4%) cognitive impairment than other groups (p = 0.003), high levels of required formal educational support (77.6%), and poorer academic competence than individuals not exposed to Valproate (p = 0.001). Overall psychosocial problems (p = 0.02), internalising problems (p = 0.05) and attention problems (p = 0.001), but not externalising problems, were elevated in individuals with a diagnosis of FVSD. Rates of neurodevelopmental disorders, particularly autistic spectrum disorders (62.9%) and sensory problems (80.6%) are particularly central to the FVSD phenotype. There was no evidence of a statistical dose-dependent effect, possibly due to the high mean dose of exposure having a uniformly negative impact across the sample. Individuals with FVSD had required a significant number of health and child development services.
Interpretation
Children and young adults with a diagnosis of FVSD are at an increased risk of a range of altered neurodevelopmental outcomes, highlighting the need for a multidisciplinary approach to clinical management across the lifespan.
期刊介绍：
Neurotoxicology and Teratology provides a forum for publishing new information regarding the effects of chemical and physical agents on the developing, adult or aging nervous system. In this context, the fields of neurotoxicology and teratology include studies of agent-induced alterations of nervous system function, with a focus on behavioral outcomes and their underlying physiological and neurochemical mechanisms. The Journal publishes original, peer-reviewed Research Reports of experimental, clinical, and epidemiological studies that address the neurotoxicity and/or functional teratology of pesticides, solvents, heavy metals, nanomaterials, organometals, industrial compounds, mixtures, drugs of abuse, pharmaceuticals, animal and plant toxins, atmospheric reaction products, and physical agents such as radiation and noise. These reports include traditional mammalian neurotoxicology experiments, human studies, studies using non-mammalian animal models, and mechanistic studies in vivo or in vitro. Special Issues, Reviews, Commentaries, Meeting Reports, and Symposium Papers provide timely updates on areas that have reached a critical point of synthesis, on aspects of a scientific field undergoing rapid change, or on areas that present special methodological or interpretive problems. Theoretical Articles address concepts and potential mechanisms underlying actions of agents of interest in the nervous system. The Journal also publishes Brief Communications that concisely describe a new method, technique, apparatus, or experimental result.
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