Literature DB >> 21940691

Pallidal deep-brain stimulation associated with complete remission of self-injurious behaviors in a patient with Lesch-Nyhan syndrome: a case report.

Laura L Deon1, Miriam A Kalichman, Cynthia L Booth, Konstantin V Slavin, Deborah J Gaebler-Spira.   

Abstract

The purpose of this case report is to review the management of a boy with Lesch-Nyhan syndrome with deep-brain stimulation who had remission of self-injurious behaviors as a result. This patient was treated with intrathecal baclofen and, later, with deep-brain stimulation to reduce hypertonia. Goals were to improve wheelchair positioning for school attendance and to reduce the use of restraints for comfort. Intrathecal baclofen was implanted twice and decreased the hypertonia, but both were explanted because of infection. Deep-brain stimulation was initiated 2.5 years ago, and since that time, comfort and function have improved and caregiver burden has decreased. Improvements in dystonia with deep-brain stimulation have also occurred, and self-injurious behaviors have resolved.

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Mesh:

Year:  2011        PMID: 21940691     DOI: 10.1177/0883073811415853

Source DB:  PubMed          Journal:  J Child Neurol        ISSN: 0883-0738            Impact factor:   1.987


  7 in total

Review 1.  Lesch-Nyhan Syndrome: Models, Theories, and Therapies.

Authors:  Scott Bell; Ilaria Kolobova; Liam Crapper; Carl Ernst
Journal:  Mol Syndromol       Date:  2016-09-24

2.  Childhood dystonias.

Authors:  Samer D Tabbal
Journal:  Curr Treat Options Neurol       Date:  2015-03       Impact factor: 3.598

Review 3.  New innovations: therapeutic opportunities for intellectual disabilities.

Authors:  Jonathan D Picker; Christopher A Walsh
Journal:  Ann Neurol       Date:  2013-09       Impact factor: 10.422

4.  Forniceal deep brain stimulation rescues hippocampal memory in Rett syndrome mice.

Authors:  Shuang Hao; Bin Tang; Zhenyu Wu; Kerstin Ure; Yaling Sun; Huifang Tao; Yan Gao; Akash J Patel; Daniel J Curry; Rodney C Samaco; Huda Y Zoghbi; Jianrong Tang
Journal:  Nature       Date:  2015-10-15       Impact factor: 49.962

Review 5.  A Proposed Diagnostic Algorithm for Inborn Errors of Metabolism Presenting With Movements Disorders.

Authors:  Juan Darío Ortigoza-Escobar
Journal:  Front Neurol       Date:  2020-11-13       Impact factor: 4.003

6.  Treatment of motor and behavioural symptoms in three Lesch-Nyhan patients with intrathecal baclofen.

Authors:  Marco Pozzi; Luigi Piccinini; Maurizio Gallo; Francesco Motta; Sonia Radice; Emilio Clementi
Journal:  Orphanet J Rare Dis       Date:  2014-12-12       Impact factor: 4.123

7.  Characterization of the Basal Ganglia Using Diffusion Tensor Imaging in Children with Self-Injurious Behavior and Tuberous Sclerosis Complex.

Authors:  Tanjala T Gipson; Andrea Poretti; Sarah A Kelley; Kathryn A Carson; Michael V Johnston; Thierry A G M Huisman
Journal:  J Neuroimaging       Date:  2019-05-06       Impact factor: 2.486

  7 in total

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