Literature DB >> 12023313

Neuro-epileptic determinants of autism spectrum disorders in tuberous sclerosis complex.

Patrick F Bolton1, Rebecca J Park, J Nicholas P Higgins, Paul D Griffiths, Andrew Pickles.   

Abstract

Tuberous sclerosis is one of the few established medical causes of autism spectrum disorder and is a unique neurogenetic model for testing theories about the brain basis of the syndrome. We conducted a retrospective case study of the neuro-epileptic risk factors predisposing to autism spectrum disorder in individuals with tuberous sclerosis to test current neurobiological theories of autism spectrum disorder. We found that an autism spectrum disorder diagnosis was associated with the presence of cortical tubers in the temporal but not other lobes of the brain. Indeed, the presence of tubers in the temporal lobes appeared to be a necessary but not sufficient risk factor for the development of an autism spectrum disorder. However, contrary to the predictions of some theories, the location of tubers in specific regions of the temporal lobe, such as the superior temporal gyrus or the right temporal lobe, did not determine which individuals with temporal lobe tubers developed an autism spectrum disorder. Instead, outcome was associated with various indices of epileptic activity including evidence of temporal lobe epileptiform discharges on EEG, the age to onset of seizures in the first 3 years of life and a history of infantile spasms. The results indicated that individuals with tuberous sclerosis are at very high risk of developing an autism spectrum disorder when temporal lobe tubers are present and associated with temporal lobe epileptiform discharges and early-onset, persistent spasm-like seizures. These risk markers constitute useful clinical indicators of prognosis, but further research is required to identify the neurobiological mechanisms responsible for their association with outcome. Most especially, it will be important to test whether, as the findings suggest, there is a critical early stage of brain maturation during which temporal lobe epilepsy perturbs the development of brain systems that underpin 'social intelligence' and possibly other cognitive skills, thereby inducing an autism spectrum disorder.

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Year:  2002        PMID: 12023313     DOI: 10.1093/brain/awf124

Source DB:  PubMed          Journal:  Brain        ISSN: 0006-8950            Impact factor:   13.501


  90 in total

1.  Impaired language pathways in tuberous sclerosis complex patients with autism spectrum disorders.

Authors:  William W Lewis; Mustafa Sahin; Benoit Scherrer; Jurriaan M Peters; Ralph O Suarez; Vanessa K Vogel-Farley; Shafali S Jeste; Matthew C Gregas; Sanjay P Prabhu; Charles A Nelson; Simon K Warfield
Journal:  Cereb Cortex       Date:  2012-06-01       Impact factor: 5.357

Review 2.  Targeted treatments for cognitive and neurodevelopmental disorders in tuberous sclerosis complex.

Authors:  Petrus J de Vries
Journal:  Neurotherapeutics       Date:  2010-07       Impact factor: 7.620

3.  Autism and Epilepsy: Exploring the Relationship Using Experimental Models.

Authors:  Carl E Stafstrom; Tim A Benke
Journal:  Epilepsy Curr       Date:  2015 Jul-Aug       Impact factor: 7.500

Review 4.  Consensus clinical guidelines for the assessment of cognitive and behavioural problems in Tuberous Sclerosis.

Authors:  Petrus de Vries; Ayla Humphrey; Deborah McCartney; Penny Prather; Patrick Bolton; Ann Hunt
Journal:  Eur Child Adolesc Psychiatry       Date:  2005-07       Impact factor: 4.785

5.  Etiologies of autism in a case-series from Tanzania.

Authors:  Raymond E Mankoski; Martha Collins; Noah K Ndosi; Ella H Mgalla; Veronica V Sarwatt; Susan E Folstein
Journal:  J Autism Dev Disord       Date:  2006-11

6.  Resting and task-modulated high-frequency brain rhythms measured by scalp encephalography in infants with tuberous sclerosis complex.

Authors:  Catherine Stamoulis; Vanessa Vogel-Farley; Geneva Degregorio; Shafali S Jeste; Charles A Nelson
Journal:  J Autism Dev Disord       Date:  2015-02

7.  Alterations in sociability and functional brain connectivity caused by early-life seizures are prevented by bumetanide.

Authors:  Gregory L Holmes; Chengju Tian; Amanda E Hernan; Sean Flynn; Devon Camp; Jeremy Barry
Journal:  Neurobiol Dis       Date:  2015-03-10       Impact factor: 5.996

8.  Effects of rapamycin on gene expression, morphology, and electrophysiological properties of rat hippocampal neurons.

Authors:  Stephan Rüegg; Marianna Baybis; Hal Juul; Marc Dichter; Peter B Crino
Journal:  Epilepsy Res       Date:  2007-11-05       Impact factor: 3.045

9.  Heterogeneous dysregulation of microRNAs across the autism spectrum.

Authors:  Kawther Abu-Elneel; Tsunglin Liu; Francesca S Gazzaniga; Yuhei Nishimura; Dennis P Wall; Daniel H Geschwind; Kaiqin Lao; Kenneth S Kosik
Journal:  Neurogenetics       Date:  2008-06-19       Impact factor: 2.660

Review 10.  Differentiating the mTOR inhibitors everolimus and sirolimus in the treatment of tuberous sclerosis complex.

Authors:  Jeffrey P MacKeigan; Darcy A Krueger
Journal:  Neuro Oncol       Date:  2015-08-19       Impact factor: 12.300

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