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Abnormal Auditory Mismatch Fields in Children and Adolescents with 47,XYY Syndrome.
Developmental Neuroscience ( IF 2.3 ) Pub Date : 2019-07-05 , DOI: 10.1159/000500799
Junko Matsuzaki 1 , Luke Bloy 1 , Lisa Blaskey 1, 2 , Judith Miller 2 , Emily S Kuschner 1, 2 , Matthew Ku 1 , Marissa Dipiero 1 , Megan Airey 1 , J Christopher Edgar 1 , David Embick 3 , Judith L Ross 4, 5 , Timothy P L Roberts 6, 7
Affiliation  

47,XYY syndrome (XYY) is one of the common forms of sex chromosome aneuploidy in males. XYY males tend to have tall stature, early speech, motor delays, social and behavioral challenges, and a high rate of language impairment. Recent studies indicate that 20-40% of males with XYY meet diagnostic criteria for autism spectrum disorder (ASD; the rate in the general population is 1-2%). Although many studies have examined the neural correlates of language impairment in ASD, few similar studies have been conducted on individuals with XYY. Studies using magnetoencephalography (MEG) in idiopathic ASD (ASD-I) have demonstrated delayed neurophysiological responses to changes in the auditory stream, revealed in the mismatch negativity or its magnetic counterpart, the mismatch field (MMF). This study investigated whether similar findings are observed in XYY-associated ASD and whether delayed processing is also present in individuals with XYY without ASD. MEG measured MMFs arising from the left and the right superior temporal gyrus during an auditory oddball paradigm with vowel stimuli (/a/ and /u/) in children/adolescents with XYY both with and without a diagnosis of ASD, as well as in those with ASD-I and in typically developing controls (TD). Ninety male participants (6-17 years old) were included in the final analyses (TD, n = 38, 11.50 ± 2.88 years; ASD-I, n = 21, 13.83 ± 3.25 years; XYY without ASD, n = 15, 12.65 ± 3.91 years; XYY with ASD, n = 16, 12.62 ± 3.19 years). The groups did not differ significantly in age (p > 0.05). There was a main effect of group on MMF latency (p < 0.001). Delayed MMF latencies were found in participants with XYY both with and without an ASD diagnosis, as well as in the ASD-I group compared to the TD group (ps < 0.001). Furthermore, participants with XYY (with and without ASD) showed a longer MMF latency than the ASD-I group (ps < 0.001). There was, however, no significant difference in MMF latency between individuals with XYY with ASD and those with XYY without ASD. Delayed MMF latencies were associated with severity of language impairment. Our findings suggest that auditory MMF latency delays are pronounced in this specific Y chromosome aneuploidy disorder, both with and without an ASD diagnosis, and thus may implicate the genes of the Y chromosome in mediating atypical MMF activity.

中文翻译:

患有47,XYY综合征的儿童和青少年的异常听觉不匹配场。

47,XYY综合征(XYY)是男性性染色体非整倍性的常见形式之一。XYY男性往往身材高大,说话早,运动迟缓,社交和行为挑战以及语言障碍率高。最近的研究表明,患有XYY的男性中有20-40%符合自闭症谱系障碍(ASD;一般人群中的比率为1-2%)。尽管许多研究已经检查了ASD中语言障碍的神经相关性,但很少有人对XYY患者进行过类似的研究。在特发性ASD(ASD-1)中使用脑磁图(MEG)进行的研究表明,对听觉流变化的神经生理反应延迟,这在失配负性或其磁性对应物失配场(MMF)中显示出来。这项研究调查了在与XYY相关的ASD中是否观察到相似的发现,以及在没有ASD的XYY患者中是否也存在延迟处理。MEG测量了在患有和不患有ASD的XYY儿童/青少年中,在元音刺激(/ a /和/ u /)的听觉奇数球范例中,左右颞上回产生的MMF。与ASD-I一起使用,并且通常用于开发控件(TD)。90名男性参与者(6-17岁)被纳入最终分析(TD,n = 38,11.50±2.88岁; ASD-I,n = 21,13.83±3.25岁;没有ASD的XYY,n = 15,12,65 ±3.91年;具有ASD的XYY,n = 16,12.62±3.19年)。各组的年龄没有显着差异(p> 0.05)。小组对MMF潜伏期有主要影响(p <0.001)。与TD组相比,在有和没有ASD诊断的XYY参与者中以及在ASD-1组中都发现了延迟的MMF潜伏期。此外,患有XYY(有和没有ASD)的参与者表现出比ASD-1组更长的MMF潜伏期(ps <0.001)。但是,患有ASD的XYY患者与没有ASD的XYY患者之间的MMF潜伏期没有显着差异。MMF延迟与语言障碍的严重程度有关。我们的发现表明,在有或没有ASD诊断的情况下,在这种特定的Y染色体非整倍性疾病中明显存在听觉MMF潜伏期延迟,因此可能暗示Y染色体的基因介导了非典型MMF活性。以及TD组与ASD-1组相比(ps <0.001)。此外,患有XYY(有和没有ASD)的参与者表现出比ASD-1组更长的MMF潜伏期(ps <0.001)。但是,患有ASD的XYY患者和没有ASD的XYY患者之间的MMF潜伏期没有显着差异。MMF延迟与语言障碍的严重程度有关。我们的发现表明,在有或没有ASD诊断的情况下,在这种特定的Y染色体非整倍性疾病中明显存在听觉MMF潜伏期延迟,因此可能暗示Y染色体的基因介导了非典型MMF活性。以及TD组与ASD-1组相比(ps <0.001)。此外,患有XYY(有和没有ASD)的参与者表现出比ASD-1组更长的MMF潜伏期(ps <0.001)。但是,患有ASD的XYY患者与没有ASD的XYY患者之间的MMF潜伏期没有显着差异。MMF延迟与语言障碍的严重程度有关。我们的发现表明,在有或没有ASD诊断的情况下,在这种特定的Y染色体非整倍性疾病中明显存在听觉MMF潜伏期延迟,因此可能暗示Y染色体的基因介导了非典型MMF活性。患有ASD的XYY个体与没有ASD的XYY个体之间的MMF潜伏期没有显着差异。MMF延迟与语言障碍的严重程度有关。我们的发现表明,在有或没有ASD诊断的情况下,在这种特定的Y染色体非整倍性疾病中明显存在听觉MMF潜伏期延迟,因此可能暗示Y染色体的基因介导了非典型MMF活性。患有ASD的XYY个体与没有ASD的XYY个体之间的MMF潜伏期没有显着差异。MMF延迟与语言障碍的严重程度有关。我们的发现表明,在有或没有ASD诊断的情况下,在这种特定的Y染色体非整倍性疾病中明显存在听觉MMF潜伏期延迟,因此可能暗示Y染色体的基因介导了非典型MMF活性。
更新日期:2019-11-01
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