interdisciplinary Developmental Neuroscience, Division of Phoniatrics, Medical University of Graz, Graz, Austria.
interdisciplinary Developmental Neuroscience, Division of Phoniatrics, Medical University of Graz, Graz, Austria; Child and Adolescent Psychiatry and Psychotherapy, interdisciplinary Developmental Neuroscience, University Medical Center Göttingen, Göttingen, Germany.
Neurosci Biobehav Rev. 2019 Mar;98:320-332. doi: 10.1016/j.neubiorev.2019.01.028.
Rett syndrome (RTT) is an X-linked genetic disorder that occurs predominantly in females. The clinical picture associated with RTT is defined by core and supportive consensus criteria, with a period of behavioural regression being a conditio sine qua non. This review sheds light on atypical neurofunctions and potential behavioural biomarkers before the onset of regression. The main focus lies on (a) motor development, especially on purposeful hand movements and the occurrence of stereotypies; and (b) speech-language and socio-communicative development. We outline potentially specific atypical behavioural patterns in these domains (e.g., vocalisations on inspiratory airstream) and different developmental traits of regression: (i) non-achievement of certain milestones: 'regression', here, might point to the fact that the lack of respective behavioural patterns appeared more and more worrisome with increasing age; and (ii) developmental milestones were achieved and functions deteriorate or even get lost during regression. To conclude, we are not quite there yet, but seem to be on the right track towards defining new and reliable neurofunctional markers for early detection of RTT.
雷特综合征(RTT)是一种主要发生在女性身上的 X 连锁遗传性疾病。与 RTT 相关的临床特征由核心和支持性共识标准定义,行为倒退期是必要条件。这篇综述探讨了行为倒退前的非典型神经功能和潜在的行为生物标志物。主要关注(a)运动发育,特别是有目的的手部运动和刻板行为的发生;以及(b)言语-语言和社会交流发展。我们概述了这些领域中潜在的特定非典型行为模式(例如,吸气气流中的发声)和不同的倒退发育特征:(i)未达到某些里程碑:“倒退”可能表明,随着年龄的增长,缺乏相应的行为模式变得越来越令人担忧;以及(ii)达到了发育里程碑,但在倒退期间功能恶化甚至丧失。总之,我们还没有完全做到这一点,但似乎正在正确的轨道上,为早期发现 RTT 定义新的、可靠的神经功能标志物。