扩展 KAT6B 疾病的神经心理学表型:与 KAT6A 综合征的重叠特征。

IF 3.2 2区 心理学 Q1 PSYCHOLOGY, DEVELOPMENTAL
Rowena Ng, Allison Kalinousky, Jacqueline Harris
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引用次数: 0

摘要

KAT6B 和 KAT6A 属于赖氨酸乙酰转移酶 MYST 家族,通过组蛋白修饰调节基因表达。虽然这两种蛋白具有相似的结构和表观遗传调控功能,但目前仍不清楚KAT6A/6B突变疾病(这两种疾病都非常罕见)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。本研究对13名KAT6B障碍患者(平均年龄=9.01岁,SD=5.46)的神经心理功能进行了初步概述,并与最近发表的15名KAT6A综合征患者(平均年龄=10.32岁,SD=4.12)的神经心理功能进行了比较。参与者完成了一系列神经心理测试以评估非语言认知能力,护理人员完成了一系列标准化评分清单以评估日常行为功能。研究结果显示,KAT6B 患儿存在严重的适应障碍(92.3%)和自闭症相关行为(83.3%),而外化行为(7.6%)相对较少,这与 KAT6A 组的情况相同。那些患有 KAT6B 障碍的人表现出较高程度的自闭症特征,包括附属兴趣降低,而 KAT6A 组中社交动机受到的影响较小。总体而言,KAT6B 患者的非言语认知和接受性语言障碍程度相当,这一趋势在 KAT6A 组中也有所体现。简而言之,KAT6B 和 KAT6A 疾病产生了类似的神经心理学特征。研究结果表明,这些表观遗传性疾病具有共同的分子病理生理机制,因此类似的疗法可能对不同疾病具有共同的疗效。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

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

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

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.

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来源期刊
CiteScore
8.00
自引率
10.30%
发文量
433
期刊介绍: The Journal of Autism and Developmental Disorders seeks to advance theoretical and applied research as well as examine and evaluate clinical diagnoses and treatments for autism and related disabilities. JADD encourages research submissions on the causes of ASDs and related disorders, including genetic, immunological, and environmental factors; diagnosis and assessment tools (e.g., for early detection as well as behavioral and communications characteristics); and prevention and treatment options. Sample topics include: Social responsiveness in young children with autism Advances in diagnosing and reporting autism Omega-3 fatty acids to treat autism symptoms Parental and child adherence to behavioral and medical treatments for autism Increasing independent task completion by students with autism spectrum disorder Does laughter differ in children with autism? Predicting ASD diagnosis and social impairment in younger siblings of children with autism The effects of psychotropic and nonpsychotropic medication with adolescents and adults with ASD Increasing independence for individuals with ASDs Group interventions to promote social skills in school-aged children with ASDs Standard diagnostic measures for ASDs Substance abuse in adults with autism Differentiating between ADHD and autism symptoms Social competence and social skills training and interventions for children with ASDs Therapeutic horseback riding and social functioning in children with autism Authors and readers of the Journal of Autism and Developmental Disorders include sch olars, researchers, professionals, policy makers, and graduate students from a broad range of cross-disciplines, including developmental, clinical child, and school psychology; pediatrics; psychiatry; education; social work and counseling; speech, communication, and physical therapy; medicine and neuroscience; and public health.
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