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© 2022. This work is published under http://creativecommons.org/licenses/by/4.0/ (the “License”). Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License.

Abstract

Introduction

We compared neuropsychiatric symptoms between child and adolescent huntingtin gene‐mutation carriers and noncarriers. Given previous evidence of atypical striatal development in carriers, we also assessed the relationship between neuropsychiatric traits and striatal development.

Methods

Participants between 6 and 18 years old were recruited from families affected by Huntington's disease and tested for the huntingtin gene expansion. Neuropsychiatric traits were assessed using the Pediatric Behavior Scale and the Behavior Rating Inventory of Executive Function. Striatal volumes were extracted from 3T neuro‐anatomical images. Multivariable linear regression models were conducted to evaluate the impact of group (i.e., gene nonexpanded [GNE] or gene expanded [GE]), age, and trajectory of striatal growth on neuropsychiatric symptoms.

Results

There were no group differences in any behavioral measure with the exception of depression/anxiety score, which was higher in the GNE group compared to the GE group (estimate = 4.58, t(129) = 2.52, FDR = 0.051). The growth trajectory of striatal volume predicted depression scores (estimate = 0.429, 95% CI 0.15:0.71, p = .0029), where a negative slope of striatal volume over time was associated with lower depression/anxiety.

Conclusions

The current findings show that GE children may have lower depression/anxiety compared to their peers. Previously, we observed a unique pattern of early striatal hypertrophy and continued decrement in volume over time among GE children and adolescents. In contrast, GNE individuals largely show striatal volume growth. These findings suggest that the lower scores of depression and anxiety seen in GE children and adolescents may be associated with differential growth of the striatum.

Details

Title
Behavioral features in child and adolescent huntingtin gene‐mutation carriers
Author
Reasoner, Erin E 1   VIAFID ORCID Logo  ; van der Plas, Ellen 1 ; Hend M. Al‐Kaylani 1   VIAFID ORCID Logo  ; Langbehn, Douglas R 1 ; Conrad, Amy L 2 ; Schultz, Jordan L 1 ; Epping, Eric A 1 ; Magnotta, Vincent A 3 ; Nopoulos, Peggy C 4   VIAFID ORCID Logo 

 Department of Psychiatry, University of Iowa Hospital and Clinics, Iowa City, Iowa, USA 
 Stead Family Children's Hospital at the University of Iowa, Iowa City, Iowa, USA 
 Department of Radiology, University of Iowa Hospital and Clinics, Iowa City, Iowa, USA 
 Department of Psychiatry, University of Iowa Hospital and Clinics, Iowa City, Iowa, USA; Stead Family Children's Hospital at the University of Iowa, Iowa City, Iowa, USA; Department of Neurology, University of Iowa Hospital and Clinics, Iowa City, Iowa, USA 
Section
ORIGINAL ARTICLES
Publication year
2022
Publication date
Jul 2022
Publisher
John Wiley & Sons, Inc.
e-ISSN
21623279
Source type
Scholarly Journal
Language of publication
English
ProQuest document ID
2692529581
Copyright
© 2022. This work is published under http://creativecommons.org/licenses/by/4.0/ (the “License”). Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License.