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扩展KAT6B障碍的神经心理学表型:与KAT6A综合征的重叠特征。

Expanding the Neuropsychological Phenotype of KAT6B Disorders: Overlapping Features with KAT6A Syndrome.

作者信息

Ng Rowena, Kalinousky Allison, Harris Jacqueline

机构信息

Dept of Neuropsychology, Kennedy Krieger Institute, 1750 E. Fairmount Ave, Baltimore, USA.

Department of Psychiatry and Behavioral Sciences, Johns Hopkins University School of Medicine, Baltimore, USA.

出版信息

J Autism Dev Disord. 2024 Aug 17. doi: 10.1007/s10803-024-06500-5.

Abstract

KAT6B and KAT6A belong to the MYST family of lysine acetyltransferases, and regulate gene expression via histone modification. Although both proteins share similar structure and epigenetic regulatory functions, it remains unclear if KAT6A/6B mutation disorders, both very rare conditions, yield the same neurocognitive presentation and thus benefit from similar treatment approaches. This study provides a preliminary overview of neuropsychological functioning of 13 individuals with KAT6B disorder (Mean age = 9.01 years, SD = 5.46), which was compared to that of a recently published sample of 15 individuals with KAT6A syndrome (Mean age = 10.32 years, SD = 4.12). Participants completed a neuropsychological test battery to assess non-verbal cognition, and caregivers completed a series of standardized rating inventories to assess daily behavioral functioning. Results reveal those with KAT6B disorders present with severe adaptive deficits (92.3%) and autism-related behaviors (83.3%), juxtaposed with relatively low concerns with externalizing behaviors (7.6%), a pattern shared by the KAT6A group. Those with KAT6B disorders present with high levels of autistic features, including reduced affiliative interest, whereas social motivation is less affected within the KAT6A group. Overall, the levels of impairment in nonverbal cognition and receptive language were comparable among those with KAT6B disorders, a trend also seen in the KAT6A group. In brief, KAT6B and KAT6A disorders yield analogous neuropsychological profiles. Findings implicate common molecular pathophysiological mechanisms for these epigenetic disorders, such that similar therapies may have shared effect across diseases.

摘要

KAT6B和KAT6A属于赖氨酸乙酰转移酶的MYST家族,通过组蛋白修饰调节基因表达。尽管这两种蛋白质具有相似的结构和表观遗传调控功能,但目前尚不清楚KAT6A/6B突变障碍(这两种都是非常罕见的病症)是否会产生相同的神经认知表现,从而受益于相似的治疗方法。本研究初步概述了13名患有KAT6B障碍的个体(平均年龄 = 9.01岁,标准差 = 5.46)的神经心理功能,并与最近发表的15名患有KAT6A综合征的个体样本(平均年龄 = 10.32岁,标准差 = 4.12)进行了比较。参与者完成了一套神经心理测试,以评估非语言认知,照顾者完成了一系列标准化的评定量表,以评估日常行为功能。结果显示,患有KAT6B障碍的个体存在严重的适应性缺陷(92.3%)和自闭症相关行为(83.3%),同时对外显行为的关注相对较低(7.6%),KAT6A组也有类似模式。患有KAT6B障碍的个体表现出高水平的自闭症特征,包括社交兴趣降低,而KAT6A组的社交动机受影响较小。总体而言,KAT6B障碍患者的非语言认知和接受性语言障碍水平相当,KAT6A组也有类似趋势。简而言之,KAT6B和KAT6A障碍产生类似的神经心理特征。研究结果暗示了这些表观遗传障碍存在共同的分子病理生理机制,因此相似的疗法可能对这两种疾病都有共同的效果。

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