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通过新生儿筛查诊断的I型戊二酸血症患儿的神经发育概况:一项随访病例系列研究

Neurodevelopmental profiles of children with glutaric aciduria type I diagnosed by newborn screening: a follow-up case series.

作者信息

Brown Amy, Crowe Louise, Beauchamp Miriam H, Anderson Vicki, Boneh Avihu

机构信息

Department of Child Neuropsychology, Murdoch Childrens Research Institute, Australian Centre for Child Neuropsychological Studies, Royal Children's Hospital, Flemington Road, Parkville, Melbourne, VIC, 3052, Australia,

出版信息

JIMD Rep. 2015;18:125-34. doi: 10.1007/8904_2014_360. Epub 2014 Dec 11.

Abstract

Glutaric aciduria type I (GA-I) is an inherited metabolic disorder that may lead to severe motor disorder and cognitive impairment. GA-I is now included in the newborn screening programme in many countries as early detection allows for prompt treatment and effectively reduces the risk of poor developmental outcome. Information regarding the long-term neurodevelopmental outcome of children with GA-I treated early is sparse.We recruited children with a confirmed diagnosis of GA-I diagnosed via newborn screening, treated in our centre and >3 years of age (n = 6). Children were assessed at two time points using a comprehensive neuropsychological test battery. Four of these had been the subject of a previous report. All participants were male, 3-6 years at the initial assessment and 6-12 years of age at the follow-up assessment.Fine motor skills were below average in all patients. Speech, which was affected in all four patients reported previously, improved following speech therapy. IQ scores remained generally stable within the normal range. Executive functioning was average to high average in four patients. Behaviour, as assessed through parental questionnaires, was problematic in two patients. Compounding factors included child neglect, family history of autism and multiple admissions to hospital (n = 1 in each).GA-I affects fine motor skills and speech, regardless of early treatment, but not IQ scores. Patients with GA-I should be referred for assessment and appropriate early intervention. Further research is needed to correlate specific neuropsychological deficits with neuroimaging.

摘要

I型戊二酸血症(GA-I)是一种遗传性代谢紊乱疾病,可能导致严重的运动障碍和认知障碍。目前,许多国家已将GA-I纳入新生儿筛查项目,因为早期检测能够实现及时治疗,并有效降低发育不良的风险。关于早期接受治疗的GA-I患儿长期神经发育结果的信息较为匮乏。我们招募了通过新生儿筛查确诊为GA-I、在我们中心接受治疗且年龄大于3岁的儿童(n = 6)。使用一套全面的神经心理测试组合在两个时间点对儿童进行评估。其中4名儿童曾是先前一份报告的研究对象。所有参与者均为男性,初次评估时年龄为3至6岁,随访评估时年龄为6至12岁。所有患者的精细运动技能均低于平均水平。先前报告的所有4名患者的言语能力均受到影响,在接受言语治疗后有所改善。智商分数总体上在正常范围内保持稳定。4名患者的执行功能处于平均水平至高平均水平。通过家长问卷评估的行为方面,有2名患者存在问题。复合因素包括儿童忽视、自闭症家族史以及多次住院(各1例)。无论早期治疗与否,GA-I都会影响精细运动技能和言语能力,但不影响智商分数。GA-I患者应接受评估并进行适当的早期干预。需要进一步研究以将特定的神经心理缺陷与神经影像学联系起来。

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