Neurodevelopmental outcomes in children and adults with Fetal Valproate Spectrum Disorder: A contribution from the ConcePTION project

IF 2.6 3区医学 Q3 NEUROSCIENCES Neurotoxicology and teratology Pub Date : 2023-09-04 DOI:10.1016/j.ntt.2023.107292

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

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Abstract

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.

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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

目的描述诊断为胎儿丙戊酸钠谱系障碍（FVSD）的年龄较大的儿童和成人的神经发育表型，包括正式诊断为FVSD的个体（n=99）、暴露于丙戊酸钠但未被诊断为FVSD的个体（n=24）和未暴露于丙戊酸钠的个体（n/23）。FVSD诊断个体的丙戊酸钠暴露平均剂量为1470 mg/天。结果FVSD诊断的个体表现出明显高于其他组的中度（43.4%）和重度（14.4%）认知障碍水平（p=0.003）、高水平的所需正规教育支持（77.6%）、，与未接触丙戊酸钠的个体相比，学习能力较差（p=0.001）。在诊断为FVSD的个体中，总体心理社会问题（p=0.02）、内在问题（p=0.05）和注意力问题（p=0.001）（而非外在问题）增加。神经发育障碍，特别是自闭症谱系障碍（62.9%）和感觉问题（80.6%）的发生率是FVSD表型的核心。没有证据表明存在统计学上的剂量依赖性效应，可能是由于高平均暴露剂量对整个样本产生了一致的负面影响。FVSD患者需要大量的健康和儿童发展服务。解释诊断为FVSD的儿童和年轻人发生一系列神经发育结果改变的风险增加，这突出了在整个生命周期内采用多学科临床管理方法的必要性。

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来源期刊

Neurotoxicology and teratology 医学-毒理学

CiteScore

5.60

自引率

10.30%

发文量

审稿时长

58 days

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.