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Thinning of the Motor–Cingulate–Insular Cortices in Siblings Concordant for Tourette Syndrome

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Abstract

Fraternal twin studies on normal subjects have demonstrated low heritability (intra-class correlation coefficient) estimates for frontal brain regions (r = 0.43). Here we aimed to investigate the relatedness/similarity estimates of the frontal brain regions in fraternal subjects concordant for Tourette syndrome (TS). We sought to identify regional brain similarities between siblings concordant for TS as an exploratory step towards the identification of potential brain structures involved in the TS phenotype. The identified brain structures may then serve in subsequent molecular genetic and linkage studies. In addition, we regressed cortical thickness and TS clinical severity scores to assess the relation between TS clinical symptoms and cortical structures. Sixteen sibling pairs concordant for TS were scanned using a 1.5 T magnetic resonance imaging scanner (age range 10–25, mean 17.19 ± 4.1). Brain morphology was assessed using the fully automated Civet pipeline at the Montreal Neurological Institute. TS was assessed using the Children’s Yale-Brown Obsessive Compulsive Scale (CY-BOCS), Yale Global Tic Severity Scale (YGTSS) and the Goetz Tic Scale. We report high relatedness/similarity estimates for fraternal siblings concordant for TS (r = 0.86–0.60) in the middle frontal-motor/cingulate/insular cortices. Regression analysis revealed significant negative correlations in the right insula with the YGTSS (r = −0.41, F = 6.09, P < 0.02) and the left cingulated cortex with the (CY-BOCS) (r = −0.35, F = 4.30, P < 0.05). Since previous findings have concluded that normal fraternal siblings are less alike in frontal cortices, the present findings may be attributed to TS. We speculate that the high ICC between siblings and the negative correlation between TS symptoms severity and cortical thickness measurements are related to the disturbances in the maturation of the motor–cingulate–insular cortical neural system that mediate self-regulatory processes. Such delayed maturation may consequently contribute to the development of TS by releasing motor and vocal tics from regulatory control. These findings may have important genetic implications.

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Acknowledgments

We thank the subjects who participated in this study. C·F. is a post-doctoral fellow of the Canadian Institutes of Health Research (CIHR), of the CIHR Genes, Mind, Behavior Training Program (M. Meany, PI) and the Jeanne Timmins Costello at the Montreal Neurological Institute.

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Authors and Affiliations

  1. Department of Neurology and Neurosurgery, McGill University, Montreal, Canada

    Cherine Fahim, Uicheul Yoon & Alan C. Evans

  2. Laboratory for the Experimental Research in Behavior (LERB), Faculty of SSP, Institute of Psychology, University of Lausanne, Lausanne, Switzerland

    Cherine Fahim

  3. Department of Psychiatry, University of Montreal, Montreal, Canada

    Cherine Fahim

  4. McConnell Brain Imaging Center, Montreal Neurological Institute, McGill University, 3801 University Street, Montreal, QC, H3A 2B4, Canada

    Cherine Fahim, Uicheul Yoon & Alan C. Evans

  5. Sainte-Justine Hospital Research Centre, Montreal, Canada

    Cherine Fahim

  6. Department of Biomedical Engineering, McGill University, Montreal, Canada

    Alan C. Evans

  7. Division of Brain Imaging & Behaviour Systems—Neuroscience Toronto Western Research Institute, University of Toronto, Toronto, Canada

    Paul Sandor

  8. Departments of Radiology and of Neurology, Mental Health Research Institute, University of Michigan, Ann Arbor, USA

    Kirk Frey

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  1. Cherine Fahim
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  2. Uicheul Yoon
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  3. Paul Sandor
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  4. Kirk Frey
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  5. Alan C. Evans
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Correspondence to Cherine Fahim.

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Fahim, C., Yoon, U., Sandor, P. et al. Thinning of the Motor–Cingulate–Insular Cortices in Siblings Concordant for Tourette Syndrome. Brain Topogr 22, 176–184 (2009). https://doi.org/10.1007/s10548-009-0105-6

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  • DOI: https://doi.org/10.1007/s10548-009-0105-6

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