IF 4.1 2区 医学 Q1 CLINICAL NEUROLOGY
Claire Mayor, Julie Husmann, Selma Benaghmouch, Stephan Eliez, Clémence Feller, Maude Schneider
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引用次数: 0

摘要

背景:最近有报告称,22q11.2 携带者的外显自传性未来思维能力较差。然而,这些障碍是由于语言能力差造成的,还是表明存在真正的外显自传体记忆缺陷,目前仍不清楚。语言障碍是 22q11DS 患儿神经心理学特征的标志,但青春期和青年期的语言结果,尤其是高级语言技能(如叙事),在很大程度上仍未得到研究。本研究的目的首先是与对照组相比,精确描述一组患有 22q11DS 且言语智力功能正常的青少年的叙事能力。其次,评估他们(过去的)自传体外显记忆和未来的外显思维能力。第三,研究语言记忆能力与自传体记忆能力之间的关系:将 15 名患有 22q11DS 的青少年与 15 名年龄和性别匹配的对照组进行比较。叙事能力通过讲故事任务进行评估,包括微观结构、宏观结构和语用测量。外显自传体记忆的评估采用的范式包括回忆过去的个人记忆和未来思考条件:结果发现:22q11DS 携带者的青少年和年轻成人仍然难以胜任讲故事等高级语言技能的任务,而且所有语言水平(即叙事的微观结构、宏观结构和语用成分)均受损。其次,与对照组相比,22q11DS 携带者对个人记忆的情节性自传回忆能力较差,对感官细节(视觉、听觉......)的获取能力较弱。他们的自传体外显记忆能力差与语言障碍无关,年龄或智力水平对他们的自传体(过去)记忆回忆也没有影响。另一方面,年龄和言语智力功能对他们在未来思维条件下进行外显叙述的能力有显著影响,这表明未来思维任务比单纯的外显记忆能力依赖于更复杂和更错综复杂的因素:言语叙事障碍并不能解释个人记忆回忆能力差的原因,这表明22q11DS患者海马和后部皮层区域之间的外显记忆网络功能失调,而这些区域的神经解剖学和神经功能改变确实已有报道。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Verbal narrative ability and episodic autobiographical memory in adolescents and young adults with 22q11.2 deletion syndrome.

Background: Poor episodic autobiographical future thinking has recently been reported in 22q11.2 carriers. However, whether these impairments are due to poor language skills or indicate a true episodic autobiographical memory deficit remains unclear. Language impairments are the hallmark of the neuropsychological profile of young children with 22q11DS, but language outcomes in adolescence and young adulthood, especially high-level linguistic skills such as narrative, remain largely unexplored. The aims of this study are first to precisely characterize the narrative abilities of a group of adolescents and young adults with a 22q11DS and normal verbal intellectual functioning, in comparison to a control group. Second, to assess their (past) autobiographical episodic memory and their future episodic thinking abilities. Third, to examine the relationship between linguistic and autobiographical memory skills.

Methods: Fifteen adolescents and young adults with 22q11DS were compared with 15 age- and sex-matched controls. Narrative ability was assessed with a storytelling task and included microstructural, macrostructural, and pragmatic linguistic measures. Episodic autobiographical memory was assessed using a paradigm involving recall of past personal memories and future thinking conditions.

Results: Adolescents and young adults with 22q11DS still struggled with high-level language skills such as storytelling tasks, and all linguistic levels were impaired, i.e., the microstructural, macrostructural, and pragmatic components of narrative. Second, 22q11DS carriers showed poor episodic autobiographical recall of their personal memories and reduced access to sensory details (visual, auditory…) compared to controls. Their poor autobiographical episodic memory skills were independent of language impairment, and there were no effects of age or intellectual level on their autobiographical (past) memories recollection. On the other hand, age and verbal intellectual functioning significantly contributed to their ability to produce episodic narratives in the future thinking condition, suggesting that the future thinking task relies on more complex and intricate factors than pure episodic memory ability.

Conclusions: Verbal narrative impairments did not account for poor recall of personal memories, suggesting dysfunctional episodic memory networks between hippocampi and posterior cortical areas in 22q11DS, where neuroanatomical and neurofunctional alterations have indeed been reported.

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来源期刊
CiteScore
7.60
自引率
4.10%
发文量
58
审稿时长
>12 weeks
期刊介绍: Journal of Neurodevelopmental Disorders is an open access journal that integrates current, cutting-edge research across a number of disciplines, including neurobiology, genetics, cognitive neuroscience, psychiatry and psychology. The journal’s primary focus is on the pathogenesis of neurodevelopmental disorders including autism, fragile X syndrome, tuberous sclerosis, Turner Syndrome, 22q Deletion Syndrome, Prader-Willi and Angelman Syndrome, Williams syndrome, lysosomal storage diseases, dyslexia, specific language impairment and fetal alcohol syndrome. With the discovery of specific genes underlying neurodevelopmental syndromes, the emergence of powerful tools for studying neural circuitry, and the development of new approaches for exploring molecular mechanisms, interdisciplinary research on the pathogenesis of neurodevelopmental disorders is now increasingly common. Journal of Neurodevelopmental Disorders provides a unique venue for researchers interested in comparing and contrasting mechanisms and characteristics related to the pathogenesis of the full range of neurodevelopmental disorders, sharpening our understanding of the etiology and relevant phenotypes of each condition.
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