| Autor: |
Adams, P.E., Adams, J.S., Nguyen, D.V., Hessl, D., Brunberg, J.A., Tassone, F., Zhang, W., Koldewyn, K., Rivera, S.M., Grigsby, J., Zhang, L., DeCarli, C., Hagerman, P.J., Hagerman, R.J. |
| Zdroj: |
American Journal of Medical Genetics. Part B, Neuropsychiatric Genetics: The Official Publication of the International Society of Psychiatric Genetics; April 2010, Vol. 153 Issue: 3 p775-785, 11p |
| Abstrakt: |
Fragile Xassociated tremorataxia syndrome FXTAS is a neurodegenerative disorder occurring in male and occasional female carriers of a premutation expansion 55–200 CGG repeats of the fragile X mental retardation 1 gene FMR1. This study assessed the relationship between hippocampal volume and psychological symptoms in carriers, both with and without FXTAS, and controls. Volumetric MRI measures, clinical staging, cognitive testing, molecular analysis, and measures of psychological symptoms were performed for female premutation carriers both with FXTAS n 16, age: 57.50 ± 12.46 and without FXTAS n 17, age: 44.94 ± 11.23, in genetically normal female controls n 8, age: 50.63 ± 11.43, male carriers with FXTAS n 34, age: 66.44 ± 6.77 and without FXTAS n 21, age: 52.38 ± 12.11, and genetically normal male controls n 30, age: 57.20 ± 14.12. We examined the relationship between psychological symptom severity and hippocampal volume, as well as correlations with molecular data. We found a significant negative correlation between total hippocampal volume and anxiety in female carriers, with and without FXTAS. This finding was mainly driven by the significant negative correlation between right hippocampal volume and anxiety. Other anxietyrelated subscales also correlated with the right hippocampus in females. In male carriers with and without FXTAS, only paranoid ideation negatively correlated with hippocampal volume. Female premutation carriers demonstrated a negative association between hippocampal volume and the severity of anxietyrelated psychological symptoms. Though the presentation of FXTAS symptoms is less common in females, anxietyrelated problems are common both prior to and after the onset of FXTAS, and may be related to hippocampal changes. © 2009 WileyLiss, Inc. |
| Databáze: |
Supplemental Index |
| Externí odkaz: |
|
Plný text