Literature DB >> 30832924

Regression in Rett syndrome: Developmental pathways to its onset.

Christa Einspieler1, Peter B Marschik2.   

Abstract

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.
Copyright © 2019 The Authors. Published by Elsevier Ltd.. All rights reserved.

Entities:  

Keywords:  Early; General movements; Home video analysis; Regression; Rett syndrome; Speech-language; Stereotypies; Vocalization

Mesh:

Year:  2019        PMID: 30832924     DOI: 10.1016/j.neubiorev.2019.01.028

Source DB:  PubMed          Journal:  Neurosci Biobehav Rev        ISSN: 0149-7634            Impact factor:   8.989


  9 in total

1.  Repeated Motor Training on Attention Reaching Skills and Stereotypies in Rett Syndrome.

Authors:  Rosa Angela Fabio; Samantha Giannatiempo; Tindara Caprì; Martina Semino
Journal:  Mov Disord Clin Pract       Date:  2022-05-19

2.  A normative chart for cognitive development in a genetically selected population.

Authors:  Jacob Vorstman; Elemi J Breetvelt; Ania M Fiksinski; Carrie E Bearden; Anne S Bassett; René S Kahn; Janneke R Zinkstok; Stephen R Hooper; Wanda Tempelaar; Donna McDonald-McGinn; Ann Swillen; Beverly Emanuel; Bernice Morrow; Raquel Gur; Eva Chow; Marianne van den Bree; Joris Vermeesch; Stephen Warren; Michael Owen; Therese van Amelsvoort; Stephan Eliez; Doron Gothelf; Celso Arango; Wendy Kates; Tony Simon; Kieran Murphy; Gabriela Repetto; Damian Heine Suner; Stefano Vicari; Joseph Cubells; Marco Armando; Nicole Philip; Linda Campbell; Sixto Garcia-Minaur; Maude Schneider; Vandana Shashi
Journal:  Neuropsychopharmacology       Date:  2021-03-29       Impact factor: 8.294

3.  Psychiatric illness and regression in individuals with Phelan-McDermid syndrome.

Authors:  Teresa M Kohlenberg; M Pilar Trelles; Brittany McLarney; Catalina Betancur; Audrey Thurm; Alexander Kolevzon
Journal:  J Neurodev Disord       Date:  2020-02-12       Impact factor: 4.025

Review 4.  Canonical Babbling: A Marker for Earlier Identification of Late Detected Developmental Disorders?

Authors:  Sigrun Lang; Katrin D Bartl-Pokorny; Florian B Pokorny; Dunia Garrido; Nivedita Mani; Annette V Fox-Boyer; Dajie Zhang; Peter B Marschik
Journal:  Curr Dev Disord Rep       Date:  2019-05-30

Review 5.  MeCP2: The Genetic Driver of Rett Syndrome Epigenetics.

Authors:  Katrina V Good; John B Vincent; Juan Ausió
Journal:  Front Genet       Date:  2021-01-21       Impact factor: 4.599

Review 6.  The future of General Movement Assessment: The role of computer vision and machine learning - A scoping review.

Authors:  Nelson Silva; Dajie Zhang; Tomas Kulvicius; Alexander Gail; Carla Barreiros; Stefanie Lindstaedt; Marc Kraft; Sven Bölte; Luise Poustka; Karin Nielsen-Saines; Florentin Wörgötter; Christa Einspieler; Peter B Marschik
Journal:  Res Dev Disabil       Date:  2021-02-08

7.  Automatic vocalisation-based detection of fragile X syndrome and Rett syndrome.

Authors:  Florian B Pokorny; Maximilian Schmitt; Mathias Egger; Katrin D Bartl-Pokorny; Dajie Zhang; Björn W Schuller; Peter B Marschik
Journal:  Sci Rep       Date:  2022-08-03       Impact factor: 4.996

8.  Systematic analysis of goal-related movement sequences during maternal behaviour in a female mouse model for Rett syndrome.

Authors:  Parker K Stevenson; Devin M Casenhiser; Billy Y B Lau; Keerthi Krishnan
Journal:  Eur J Neurosci       Date:  2021-06-22       Impact factor: 3.698

9.  Enhancing early detection of neurological and developmental disorders and provision of intervention in low-resource settings in Uttar Pradesh, India: study protocol of the G.A.N.E.S.H. programme.

Authors:  Moreno Toldo; Swami Varishthananda; Christa Einspieler; Neeraj Tripathi; Anshu Singh; Surendra K Verma; Kanchan Vishwakarma; Dajie Zhang; Agyeya Dwivedi; Ritika Gupta; Sanjay Karn; Nirmal Kerketta; Ram Narayan; Karuna Nikam Singh; Sumitra Rani; Akanksha Singh; Divyanshu Singh; Krishna Pratap Singh; Navin Singh; Neeraj Singh; Rishi Singh; Shyam P Singh; Rakesh Srivastava; Sandeep Srivastava; Sanjeev Srivastava; Gopal Yadav; Preeti Yadav; Sheshnath Yadav; Sujata Yadav; Peter B Marschik
Journal:  BMJ Open       Date:  2020-11-03       Impact factor: 2.692

  9 in total

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