Erin E Reasoner1, Ellen van der Plas1, Douglas R Langbehn1, Amy L Conrad2, Timothy R Koscik1, Eric A Epping1, Vincent A Magnotta1,3, Peggy C Nopoulos1,2,4. 1. Department of Psychiatry, University of Iowa Hospital and Clinics, Iowa City, IA, USA. 2. Stead Family Department of Pediatrics, University of Iowa Hospital and Clinics, Iowa City, IA, USA. 3. Department of Radiology, University of Iowa Hospital and Clinics, Iowa City, IA, USA. 4. Department of Neurology, University of Iowa Hospital and Clinics, Iowa City, IA, USA.
Abstract
BACKGROUND: Molecular studies provide evidence that mutant huntingtin (mHTT) affects early cortical development; however, cortical development has not been evaluated in child and adolescent carriers of mHTT. OBJECTIVE: To evaluate the impact of mHTT on the developmental trajectories of cortical thickness and surface area. METHODS: Children and adolescents (6-18 years) participated in the KidsHD study. mHTT carrier status was determined for research purposes only to classify participants as gene expanded (GE) and gene non-expanded (GNE). Cortical features were extracted from 3T neuroimaging using FreeSurfer. Nonlinear mixed effects models were conducted to determine if age, group, and CAG repeat were associated with cortical morphometry. RESULTS: Age-related changes in cortical morphometry were similar across groups. Expanded CAG repeat was not significantly associated with cortical features. CONCLUSION: While striatal development is markedly different in GE and GNE, developmental change of the cortex appears grossly normal among child and adolescent carrier of mHTT.
BACKGROUND: Molecular studies provide evidence that mutant huntingtin (mHTT) affects early cortical development; however, cortical development has not been evaluated in child and adolescent carriers of mHTT. OBJECTIVE: To evaluate the impact of mHTT on the developmental trajectories of cortical thickness and surface area. METHODS: Children and adolescents (6-18 years) participated in the KidsHD study. mHTT carrier status was determined for research purposes only to classify participants as gene expanded (GE) and gene non-expanded (GNE). Cortical features were extracted from 3T neuroimaging using FreeSurfer. Nonlinear mixed effects models were conducted to determine if age, group, and CAG repeat were associated with cortical morphometry. RESULTS: Age-related changes in cortical morphometry were similar across groups. Expanded CAG repeat was not significantly associated with cortical features. CONCLUSION: While striatal development is markedly different in GE and GNE, developmental change of the cortex appears grossly normal among child and adolescent carrier of mHTT.
Entities:
Keywords:
Huntington’s disease; children at risk for HD; cortical development; magnetic resonance imaging; trinucleotide repeat disorder
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