Journal article
Behavioral features in child and adolescent huntingtin gene-mutation carriers
Brain and behavior, Vol.12(7), pp.e2630-n/a
05/23/2022
DOI: 10.1002/brb3.2630
PMCID: PMC9304841
PMID: 35604958
Abstract
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.
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.
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.
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: Subtitle
- Behavioral features in child and adolescent huntingtin gene-mutation carriers
- Creators
- Erin E Reasoner - University of IowaEllen van der Plas - University of IowaHend M Al-Kaylani - University of IowaDouglas R Langbehn - University of IowaAmy L Conrad - University of IowaJordan L Schultz - University of IowaEric A Epping - University of IowaVincent A Magnotta - University of IowaPeggy C Nopoulos - Department of Neurology, University of Iowa Hospital and Clinics, Iowa City, Iowa, USA
- Resource Type
- Journal article
- Publication Details
- Brain and behavior, Vol.12(7), pp.e2630-n/a
- DOI
- 10.1002/brb3.2630
- PMID
- 35604958
- PMCID
- PMC9304841
- NLM abbreviation
- Brain Behav
- ISSN
- 2162-3279
- eISSN
- 2162-3279
- Grant note
- 1S10OD025025-01 / NIH HHS
- Language
- English
- Date published
- 05/23/2022
- Academic Unit
- Roy J. Carver Department of Biomedical Engineering; Neurology; Radiology; Psychiatry; Pediatric Psychology; Stead Family Department of Pediatrics; Iowa Neuroscience Institute; Craniofacial Anomalies Research Center; Pharmacy Practice and Science
- Record Identifier
- 9984281755002771
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