Spatial working memory in neurofibromatosis 1: Altered neural activity and functional connectivity
Background: Neurofibromatosis Type 1 (NF1) is a genetic disorder that disrupts central nervous system development and neuronal function. Cognitively, NF1 is characterized by difficulties with executive control and visuospatial abilities. Little is known about the neural substrates underlying these d...
| Main Authors: | , , , , , , , , , |
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| Format: | Article |
| Language: | English |
| Published: |
Elsevier
2017-01-01
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| Series: | NeuroImage: Clinical |
| Online Access: | http://www.sciencedirect.com/science/article/pii/S2213158217301638 |
| _version_ | 1819266198544580608 |
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| author | Amira F.A. Ibrahim Caroline A. Montojo Kristen M. Haut Katherine H. Karlsgodt Laura Hansen Eliza Congdon Tena Rosser Robert M. Bilder Alcino J. Silva Carrie E. Bearden |
| author_facet | Amira F.A. Ibrahim Caroline A. Montojo Kristen M. Haut Katherine H. Karlsgodt Laura Hansen Eliza Congdon Tena Rosser Robert M. Bilder Alcino J. Silva Carrie E. Bearden |
| author_sort | Amira F.A. Ibrahim |
| collection | DOAJ |
| description | Background: Neurofibromatosis Type 1 (NF1) is a genetic disorder that disrupts central nervous system development and neuronal function. Cognitively, NF1 is characterized by difficulties with executive control and visuospatial abilities. Little is known about the neural substrates underlying these deficits. The current study utilized Blood-Oxygen-Level-Dependent (BOLD) functional MRI (fMRI) to explore the neural correlates of spatial working memory (WM) deficits in patients with NF1. Methods: BOLD images were acquired from 23 adults with NF1 (age M=32.69; 61% male) and 25 matched healthy controls (age M=33.08; 64% male) during an in-scanner visuo-spatial WM task. Whole brain functional and psycho-physiological interaction analyses were utilized to investigate neural activity and functional connectivity, respectively, during visuo-spatial WM performance. Participants also completed behavioral measures of spatial reasoning and verbal WM. Results: Relative to healthy controls, participants with NF1 showed reduced recruitment of key components of WM circuitry, the left dorsolateral prefrontal cortex and right parietal cortex. In addition, healthy controls exhibited greater simultaneous deactivation between the posterior cingulate cortex (PCC) and temporal regions than NF1 patients. In contrast, NF1 patients showed greater PCC and bilateral parietal connectivity with visual cortices as well as between the PCC and the cerebellum. In NF1 participants, increased functional coupling of the PCC with frontal and parietal regions was associated with better spatial reasoning and WM performance, respectively; these relationships were not observed in controls. Conclusions: Dysfunctional engagement of WM circuitry, and aberrant functional connectivity of ‘task-negative’ regions in NF1 patients may underlie spatial WM difficulties characteristic of the disorder. Keywords: fMRI, NF1, Working memory, Spatial ability, Psychophysiological interaction |
| first_indexed | 2024-12-23T20:57:28Z |
| format | Article |
| id | doaj.art-f0b27a9934a54c7eab40ef2c613c779b |
| institution | Directory Open Access Journal |
| issn | 2213-1582 |
| language | English |
| last_indexed | 2024-12-23T20:57:28Z |
| publishDate | 2017-01-01 |
| publisher | Elsevier |
| record_format | Article |
| series | NeuroImage: Clinical |
| spelling | doaj.art-f0b27a9934a54c7eab40ef2c613c779b2022-12-21T17:31:29ZengElsevierNeuroImage: Clinical2213-15822017-01-0115801811Spatial working memory in neurofibromatosis 1: Altered neural activity and functional connectivityAmira F.A. Ibrahim0Caroline A. Montojo1Kristen M. Haut2Katherine H. Karlsgodt3Laura Hansen4Eliza Congdon5Tena Rosser6Robert M. Bilder7Alcino J. Silva8Carrie E. Bearden9Department of Psychology, University of Michigan, Ann Arbor, United StatesKavli Foundation, Oxnard, United StatesDepartment of Psychiatry, Rush University Medical Center, United StatesDepartment of Psychology, University of California, Los Angeles, United States; Department of Psychiatry & Biobehavioral Sciences, University of California, Los Angeles, United StatesDepartment of Psychiatry & Biobehavioral Sciences, University of California, Los Angeles, United StatesDepartment of Psychiatry & Biobehavioral Sciences, University of California, Los Angeles, United StatesDepartment of Neurology, Children's Hospital of Los Angeles, University of Southern California Keck School of Medicine, Los Angeles, United StatesDepartment of Psychology, University of California, Los Angeles, United States; Department of Psychiatry & Biobehavioral Sciences, University of California, Los Angeles, United StatesDepartment of Psychology, University of California, Los Angeles, United States; Department of Psychiatry & Biobehavioral Sciences, University of California, Los Angeles, United States; Department of Neurobiology, University of California, Los Angeles, United States; Integrative Center for Learning and Memory, University of California, Los Angeles, United StatesDepartment of Psychology, University of California, Los Angeles, United States; Department of Psychiatry & Biobehavioral Sciences, University of California, Los Angeles, United States; Integrative Center for Learning and Memory, University of California, Los Angeles, United States; Corresponding author at: Department of Psychology, University of California, Los Angeles, United States.Background: Neurofibromatosis Type 1 (NF1) is a genetic disorder that disrupts central nervous system development and neuronal function. Cognitively, NF1 is characterized by difficulties with executive control and visuospatial abilities. Little is known about the neural substrates underlying these deficits. The current study utilized Blood-Oxygen-Level-Dependent (BOLD) functional MRI (fMRI) to explore the neural correlates of spatial working memory (WM) deficits in patients with NF1. Methods: BOLD images were acquired from 23 adults with NF1 (age M=32.69; 61% male) and 25 matched healthy controls (age M=33.08; 64% male) during an in-scanner visuo-spatial WM task. Whole brain functional and psycho-physiological interaction analyses were utilized to investigate neural activity and functional connectivity, respectively, during visuo-spatial WM performance. Participants also completed behavioral measures of spatial reasoning and verbal WM. Results: Relative to healthy controls, participants with NF1 showed reduced recruitment of key components of WM circuitry, the left dorsolateral prefrontal cortex and right parietal cortex. In addition, healthy controls exhibited greater simultaneous deactivation between the posterior cingulate cortex (PCC) and temporal regions than NF1 patients. In contrast, NF1 patients showed greater PCC and bilateral parietal connectivity with visual cortices as well as between the PCC and the cerebellum. In NF1 participants, increased functional coupling of the PCC with frontal and parietal regions was associated with better spatial reasoning and WM performance, respectively; these relationships were not observed in controls. Conclusions: Dysfunctional engagement of WM circuitry, and aberrant functional connectivity of ‘task-negative’ regions in NF1 patients may underlie spatial WM difficulties characteristic of the disorder. Keywords: fMRI, NF1, Working memory, Spatial ability, Psychophysiological interactionhttp://www.sciencedirect.com/science/article/pii/S2213158217301638 |
| spellingShingle | Amira F.A. Ibrahim Caroline A. Montojo Kristen M. Haut Katherine H. Karlsgodt Laura Hansen Eliza Congdon Tena Rosser Robert M. Bilder Alcino J. Silva Carrie E. Bearden Spatial working memory in neurofibromatosis 1: Altered neural activity and functional connectivity NeuroImage: Clinical |
| title | Spatial working memory in neurofibromatosis 1: Altered neural activity and functional connectivity |
| title_full | Spatial working memory in neurofibromatosis 1: Altered neural activity and functional connectivity |
| title_fullStr | Spatial working memory in neurofibromatosis 1: Altered neural activity and functional connectivity |
| title_full_unstemmed | Spatial working memory in neurofibromatosis 1: Altered neural activity and functional connectivity |
| title_short | Spatial working memory in neurofibromatosis 1: Altered neural activity and functional connectivity |
| title_sort | spatial working memory in neurofibromatosis 1 altered neural activity and functional connectivity |
| url | http://www.sciencedirect.com/science/article/pii/S2213158217301638 |
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