Behavioral features in child and adolescent huntingtin gene‐mutation carriers
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...
| Main Authors: | , , , , , , , , |
|---|---|
| Format: | Article |
| Language: | English |
| Published: |
Wiley
2022-07-01
|
| Series: | Brain and Behavior |
| Subjects: | |
| Online Access: | https://doi.org/10.1002/brb3.2630 |
| _version_ | 1811288652885524480 |
|---|---|
| author | Erin E. Reasoner Ellen van derPlas Hend M. Al‐Kaylani Douglas R. Langbehn Amy L. Conrad Jordan L. Schultz Eric A. Epping Vincent A. Magnotta Peggy C. Nopoulos |
| author_facet | Erin E. Reasoner Ellen van derPlas Hend M. Al‐Kaylani Douglas R. Langbehn Amy L. Conrad Jordan L. Schultz Eric A. Epping Vincent A. Magnotta Peggy C. Nopoulos |
| author_sort | Erin E. Reasoner |
| collection | DOAJ |
| description | 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. |
| first_indexed | 2024-04-13T03:41:02Z |
| format | Article |
| id | doaj.art-15f1ae227a0948beb8cdd957c2fcc1c1 |
| institution | Directory Open Access Journal |
| issn | 2162-3279 |
| language | English |
| last_indexed | 2024-04-13T03:41:02Z |
| publishDate | 2022-07-01 |
| publisher | Wiley |
| record_format | Article |
| series | Brain and Behavior |
| spelling | doaj.art-15f1ae227a0948beb8cdd957c2fcc1c12022-12-22T03:04:09ZengWileyBrain and Behavior2162-32792022-07-01127n/an/a10.1002/brb3.2630Behavioral features in child and adolescent huntingtin gene‐mutation carriersErin E. Reasoner0Ellen van derPlas1Hend M. Al‐Kaylani2Douglas R. Langbehn3Amy L. Conrad4Jordan L. Schultz5Eric A. Epping6Vincent A. Magnotta7Peggy C. Nopoulos8Department of Psychiatry University of Iowa Hospital and Clinics Iowa City Iowa USADepartment of Psychiatry University of Iowa Hospital and Clinics Iowa City Iowa USADepartment of Psychiatry University of Iowa Hospital and Clinics Iowa City Iowa USADepartment of Psychiatry University of Iowa Hospital and Clinics Iowa City Iowa USAStead Family Children's Hospital at the University of Iowa Iowa City Iowa USADepartment of Psychiatry University of Iowa Hospital and Clinics Iowa City Iowa USADepartment of Psychiatry University of Iowa Hospital and Clinics Iowa City Iowa USADepartment of Radiology University of Iowa Hospital and Clinics Iowa City Iowa USADepartment of Psychiatry University of Iowa Hospital and Clinics Iowa City Iowa USAAbstract 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.https://doi.org/10.1002/brb3.2630anxietydepressionHuntington diseaseneostriatumneuropsychological testspediatric psychology |
| spellingShingle | Erin E. Reasoner Ellen van derPlas Hend M. Al‐Kaylani Douglas R. Langbehn Amy L. Conrad Jordan L. Schultz Eric A. Epping Vincent A. Magnotta Peggy C. Nopoulos Behavioral features in child and adolescent huntingtin gene‐mutation carriers Brain and Behavior anxiety depression Huntington disease neostriatum neuropsychological tests pediatric psychology |
| title | Behavioral features in child and adolescent huntingtin gene‐mutation carriers |
| title_full | Behavioral features in child and adolescent huntingtin gene‐mutation carriers |
| title_fullStr | Behavioral features in child and adolescent huntingtin gene‐mutation carriers |
| title_full_unstemmed | Behavioral features in child and adolescent huntingtin gene‐mutation carriers |
| title_short | Behavioral features in child and adolescent huntingtin gene‐mutation carriers |
| title_sort | behavioral features in child and adolescent huntingtin gene mutation carriers |
| topic | anxiety depression Huntington disease neostriatum neuropsychological tests pediatric psychology |
| url | https://doi.org/10.1002/brb3.2630 |
| work_keys_str_mv | AT erinereasoner behavioralfeaturesinchildandadolescenthuntingtingenemutationcarriers AT ellenvanderplas behavioralfeaturesinchildandadolescenthuntingtingenemutationcarriers AT hendmalkaylani behavioralfeaturesinchildandadolescenthuntingtingenemutationcarriers AT douglasrlangbehn behavioralfeaturesinchildandadolescenthuntingtingenemutationcarriers AT amylconrad behavioralfeaturesinchildandadolescenthuntingtingenemutationcarriers AT jordanlschultz behavioralfeaturesinchildandadolescenthuntingtingenemutationcarriers AT ericaepping behavioralfeaturesinchildandadolescenthuntingtingenemutationcarriers AT vincentamagnotta behavioralfeaturesinchildandadolescenthuntingtingenemutationcarriers AT peggycnopoulos behavioralfeaturesinchildandadolescenthuntingtingenemutationcarriers |