View in EDS HTML Full Text PDF Full Text

Accumulated seizure burden predicts neurodevelopmental outcome at 36 months of age in patients with tuberous sclerosis complex.

Saved in:
Bibliographic Details
Title: Accumulated seizure burden predicts neurodevelopmental outcome at 36 months of age in patients with tuberous sclerosis complex.
Authors: Ihnen, S. Katie Z. (AUTHOR), Alperin, Samuel (AUTHOR), Capal, Jamie K. (AUTHOR), Cohen, Alexander L. (AUTHOR), Peters, Jurriaan M. (AUTHOR), Bebin, E. Martina (AUTHOR), Northrup, Hope A. (AUTHOR), Sahin, Mustafa (AUTHOR), Krueger, Darcy A. (AUTHOR)
Source: Epilepsia (Series 4). Jan2025, Vol. 66 Issue 1, p117-133. 17p.
Subjects: Seizures (Medicine), Intellectual disabilities, Cognitive ability, Machine learning, Epilepsy, Biomarkers, Tuberous sclerosis, Neural development
Abstract: Objective: Epilepsy and intellectual disability are common in tuberous sclerosis complex (TSC). Although early life seizures and intellectual disability are known to be correlated in TSC, the differential effects of age at seizure onset and accumulated seizure burden on development remain unclear. Methods: Daily seizure diaries, serial neurodevelopmental testing, and brain magnetic resonance imaging were analyzed for 129 TSC patients followed from 0 to 36 months. We used machine learning to identify subgroups of patients based on neurodevelopmental test scores at 36 months of age and assessed the stability of those subgroups at 12 months. We tested the ability of candidate biomarkers to predict 36‐month neurodevelopmental subgroup using univariable and multivariable logistic regression. Candidate biomarkers included age at seizure onset, accumulated seizure burden, tuber volume, sex, and earlier neurodevelopmental test scores. Results: Patients clustered into two neurodevelopmental subgroups at 36 months of age, higher and lower scoring. Subgroup was mostly (75%) the same at 12 months. Significant univariable effects on subgroup were seen only for accumulated seizure burden (largest effect), earlier test scores, and tuber volume. Neither age at seizure onset nor sex significantly distinguished 36‐month subgroups, although for girls but not boys there was a significant effect of age at seizure onset. In the multivariable model, accumulated seizure burden and earlier test scores together predicted 36‐month neurodevelopmental group with 82% accuracy and an area under the curve of.86. Significance: These results untangle the contributions of age at seizure onset and accumulated seizure burden to neurodevelopmental outcomes in young children with TSC. Accumulated seizure burden, rather than the age at seizure onset, most accurately predicts neurodevelopmental outcome at 36 months of age. These results emphasize the need to manage seizures aggressively during the first 3 years of life for patients with TSC, not only to promote seizure control but to optimize cognitive function. [ABSTRACT FROM AUTHOR]
Copyright of Epilepsia (Series 4) is the property of Wiley-Blackwell and its content may not be copied or emailed to multiple sites without the copyright holder's express written permission. Additionally, content may not be used with any artificial intelligence tools or machine learning technologies. However, users may print, download, or email articles for individual use. This abstract may be abridged. No warranty is given about the accuracy of the copy. Users should refer to the original published version of the material for the full abstract. (Copyright applies to all Abstracts.)
Database: Psychology and Behavioral Sciences Collection
Full text is not displayed to guests.
FullText Links:
  – Type: pdflink
Text:
  Availability: 1
Header DbId: pbh
DbLabel: Psychology and Behavioral Sciences Collection
An: 184044451
AccessLevel: 6
PubType: Academic Journal
PubTypeId: academicJournal
PreciseRelevancyScore: 0
IllustrationInfo
Items – Name: Title
  Label: Title
  Group: Ti
  Data: Accumulated seizure burden predicts neurodevelopmental outcome at 36 months of age in patients with tuberous sclerosis complex.
– Name: Author
  Label: Authors
  Group: Au
  Data: <searchLink fieldCode="AR" term="%22Ihnen%2C+S%2E+Katie+Z%2E%22">Ihnen, S. Katie Z.</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Alperin%2C+Samuel%22">Alperin, Samuel</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Capal%2C+Jamie+K%2E%22">Capal, Jamie K.</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Cohen%2C+Alexander+L%2E%22">Cohen, Alexander L.</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Peters%2C+Jurriaan+M%2E%22">Peters, Jurriaan M.</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Bebin%2C+E%2E+Martina%22">Bebin, E. Martina</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Northrup%2C+Hope+A%2E%22">Northrup, Hope A.</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Sahin%2C+Mustafa%22">Sahin, Mustafa</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Krueger%2C+Darcy+A%2E%22">Krueger, Darcy A.</searchLink> (AUTHOR)
– Name: TitleSource
  Label: Source
  Group: Src
  Data: <searchLink fieldCode="JN" term="%22Epilepsia+%28Series+4%29%22">Epilepsia (Series 4)</searchLink>. Jan2025, Vol. 66 Issue 1, p117-133. 17p.
– Name: Subject
  Label: Subjects
  Group: Su
  Data: <searchLink fieldCode="DE" term="%22Seizures+%28Medicine%29%22">Seizures (Medicine)</searchLink><br /><searchLink fieldCode="DE" term="%22Intellectual+disabilities%22">Intellectual disabilities</searchLink><br /><searchLink fieldCode="DE" term="%22Cognitive+ability%22">Cognitive ability</searchLink><br /><searchLink fieldCode="DE" term="%22Machine+learning%22">Machine learning</searchLink><br /><searchLink fieldCode="DE" term="%22Epilepsy%22">Epilepsy</searchLink><br /><searchLink fieldCode="DE" term="%22Biomarkers%22">Biomarkers</searchLink><br /><searchLink fieldCode="DE" term="%22Tuberous+sclerosis%22">Tuberous sclerosis</searchLink><br /><searchLink fieldCode="DE" term="%22Neural+development%22">Neural development</searchLink>
– Name: Abstract
  Label: Abstract
  Group: Ab
  Data: Objective: Epilepsy and intellectual disability are common in tuberous sclerosis complex (TSC). Although early life seizures and intellectual disability are known to be correlated in TSC, the differential effects of age at seizure onset and accumulated seizure burden on development remain unclear. Methods: Daily seizure diaries, serial neurodevelopmental testing, and brain magnetic resonance imaging were analyzed for 129 TSC patients followed from 0 to 36 months. We used machine learning to identify subgroups of patients based on neurodevelopmental test scores at 36 months of age and assessed the stability of those subgroups at 12 months. We tested the ability of candidate biomarkers to predict 36‐month neurodevelopmental subgroup using univariable and multivariable logistic regression. Candidate biomarkers included age at seizure onset, accumulated seizure burden, tuber volume, sex, and earlier neurodevelopmental test scores. Results: Patients clustered into two neurodevelopmental subgroups at 36 months of age, higher and lower scoring. Subgroup was mostly (75%) the same at 12 months. Significant univariable effects on subgroup were seen only for accumulated seizure burden (largest effect), earlier test scores, and tuber volume. Neither age at seizure onset nor sex significantly distinguished 36‐month subgroups, although for girls but not boys there was a significant effect of age at seizure onset. In the multivariable model, accumulated seizure burden and earlier test scores together predicted 36‐month neurodevelopmental group with 82% accuracy and an area under the curve of.86. Significance: These results untangle the contributions of age at seizure onset and accumulated seizure burden to neurodevelopmental outcomes in young children with TSC. Accumulated seizure burden, rather than the age at seizure onset, most accurately predicts neurodevelopmental outcome at 36 months of age. These results emphasize the need to manage seizures aggressively during the first 3 years of life for patients with TSC, not only to promote seizure control but to optimize cognitive function. [ABSTRACT FROM AUTHOR]
– Name: AbstractSuppliedCopyright
  Label:
  Group: Ab
  Data: <i>Copyright of Epilepsia (Series 4) is the property of Wiley-Blackwell and its content may not be copied or emailed to multiple sites without the copyright holder's express written permission. Additionally, content may not be used with any artificial intelligence tools or machine learning technologies. However, users may print, download, or email articles for individual use. This abstract may be abridged. No warranty is given about the accuracy of the copy. Users should refer to the original published version of the material for the full abstract.</i> (Copyright applies to all Abstracts.)
PLink https://search.ebscohost.com/login.aspx?direct=true&site=eds-live&db=pbh&AN=184044451
RecordInfo BibRecord:
  BibEntity:
    Identifiers:
      – Type: doi
        Value: 10.1111/epi.18172
    Languages:
      – Code: eng
        Text: English
    PhysicalDescription:
      Pagination:
        PageCount: 17
        StartPage: 117
    Subjects:
      – SubjectFull: Seizures (Medicine)
        Type: general
      – SubjectFull: Intellectual disabilities
        Type: general
      – SubjectFull: Cognitive ability
        Type: general
      – SubjectFull: Machine learning
        Type: general
      – SubjectFull: Epilepsy
        Type: general
      – SubjectFull: Biomarkers
        Type: general
      – SubjectFull: Tuberous sclerosis
        Type: general
      – SubjectFull: Neural development
        Type: general
    Titles:
      – TitleFull: Accumulated seizure burden predicts neurodevelopmental outcome at 36 months of age in patients with tuberous sclerosis complex.
        Type: main
  BibRelationships:
    HasContributorRelationships:
      – PersonEntity:
          Name:
            NameFull: Ihnen, S. Katie Z.
      – PersonEntity:
          Name:
            NameFull: Alperin, Samuel
      – PersonEntity:
          Name:
            NameFull: Capal, Jamie K.
      – PersonEntity:
          Name:
            NameFull: Cohen, Alexander L.
      – PersonEntity:
          Name:
            NameFull: Peters, Jurriaan M.
      – PersonEntity:
          Name:
            NameFull: Bebin, E. Martina
      – PersonEntity:
          Name:
            NameFull: Northrup, Hope A.
      – PersonEntity:
          Name:
            NameFull: Sahin, Mustafa
      – PersonEntity:
          Name:
            NameFull: Krueger, Darcy A.
    IsPartOfRelationships:
      – BibEntity:
          Dates:
            – D: 01
              M: 01
              Text: Jan2025
              Type: published
              Y: 2025
          Identifiers:
            – Type: issn-print
              Value: 00139580
          Numbering:
            – Type: volume
              Value: 66
            – Type: issue
              Value: 1
          Titles:
            – TitleFull: Epilepsia (Series 4)
              Type: main
ResultId 1