Spinal lesion level in spina bifida: a source of neural and cognitive heterogeneity

Fletcher, Jack Μ.; Copeland, Kim; Frederick, Jon A.; Blaser, Susan; Kramer, Larry A.; Northrup, Hope; Hannay, H. Julia; Brandt, Michael E.; Francis, D.; Villarreal, Grace; Drake, James M.; Laurent, John P.; Townsend, Irene; Inwood, Susan; Boudousquie, Amy; Dennis, Maureen

doi:10.3171/ped.2005.102.3.0268

Cited by 153 publications

(276 citation statements)

References 19 publications

(18 reference statements)

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“…No significant group differences in total cerebral brain volume were observed [Mean, SD: SB =1,077cm 3 , ±120.2 cm 3 ; PC=1,144cm 3 , ±99.9cm 3 , t (29.03) = −1.71, p>0.09]. These results are consistent with published reports of global volumetric analyses in much larger samples of SB (Fletcher et al, 1996;Fletcher et al, 2005).…”

Section: Global Volumetricssupporting

confidence: 82%

Neocortical reorganization in spina bifida

et al. 2008

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Normal brain development throughout childhood and adolescence is usually characterized by decreased cortical thickness in the frontal regions as well as region-specific patterns of increased white matter myelination and volume. We investigated total cerebral volumes, neocortical surface area, and neocortical thickness in 16 children with a neural tube defect, spina bifida myelomeningocele (SB), and 16 age-matched typically developing controls using a semi-automated, quantitative approach to MRI-based brain morphometry. The results revealed no significant group differences in total cerebral volume. However, group differences were observed in the global distribution of distinct tissue classes within the cerebrum: the SB group demonstrated a significant 15% reduction in total white matter and a 69% increase in cerebrospinal fluid, with no differences in total gray matter. Group comparisons of neocortical surface area assessments were significantly smaller in the occipital regions for SB, with no significant group differences in the frontal regions. Group comparisons of cortical thickness measurements demonstrated reduced cortical thickness in all regions except the frontal regions, where the SB group exhibited an increase relative to the PC group. Although regional patterns of thinning may be associated with the mechanical effects of hydrocephalus, the overall reduction in white matter and increased neocortical thickness in the frontal regions suggest that SB reflects a long term disruption of brain development that extends far beyond the neural tube defect in the first weeks of gestation.

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Section: Global Volumetricssupporting

confidence: 82%

Neocortical reorganization in spina bifida

et al. 2008

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“…Musculoskeletal abnormalities associated with SA include a shortened intergluteal cleft, flattened buttock, hip dysplasia, hip and knee flexion contractures, distal leg atrophy, talipes and other foot deformities. The patient in this case did demonstrate, along with sacral agenesis, multiple congenital fusion abnormalities of the thoracic and lumbar vertebral bodies leading to a congenital scoliosis, uterus didelphy, renal abnormalities including renal cysts and bilateral hydronephrosis, and previously had required urostomy and colostomy procedures as a child related to lack of bladder and bowel function [15]. The neurologic deficit associated with SA correlates with the level of the vertebral defect and neuropathologic studies have demonstrated distal cord dysplasia in patients with lumbar or high SA [1,9].…”

Section: Discussionmentioning

confidence: 78%

A tail of sacral agenesis: delayed presentation of meningocele in sacral agenesis

2012

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Introduction Sacral agenesis is a congenital condition associated with multiple orthopedic, spinal, abdominal and thoracic organ deformities. Meningocele is commonly found among patients with sacral agenesis. Description We present the first case in the literature describing a delayed presentation of terminal (posterior) meningocele in an adult patient born with sacral agenesis. Conclusion Surgical repair was performed and is the best treatment option for significantly large lesions, with postoperative CSF leak being the main complication.

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“…Indeed, researchers have found that performance on neuropsychological measures varies among children with spina bifida (Barf et al, 2003;Fletcher et al, 2005;Snow et al, 1994;Wills, 1993). Significant within group differences could be indicative of variations of severity within the same profile (i.e., quantitative differences across profiles) or different patterns of performance that are indicative of multiple profiles (i.e., qualitative differences across profiles).…”

Section: Introductionmentioning

confidence: 99%

Profiles of Neuropsychological Functioning in Children and Adolescents with Spina Bifida: Associations with Biopsychosocial Predictors and Functional Outcomes

Wasserman

Holmbeck

2016

J Int Neuropsychol Soc

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Objectives: The current study examined neuropsychological performance among children with spina bifida (SB) to determine biological and functional correlates of distinct "profiles" of cognitive functioning. Methods: A total of 95 children with SB myelomeningocele (ages, 8-15 years) completed a neuropsychological assessment battery. Hierarchical and non-hierarchical cluster analyses were used to identify and confirm a cluster solution. Hypothesized predictors of cluster membership included lesion level, number of shunt surgeries, history of seizures, age, ethnicity, socio-economic status, and family stress. Outcomes included independence, academic success, expectations for the future, and quality of life. Results: Ward's cluster method indicated a three-cluster solution, and was replicated with two other cluster analytic methods. The following labels were applied to the clusters: "average to low average" (n = 39), "extremely low to borderline" (n = 27), and "broadly average with verbal strength" (n = 29). Socio-econimc status, lesion level, and seizure history significantly predicted group membership. Cluster membership significantly predicted independence, academic success, parent expectations for the future, and child reported physical quality of life. Conclusions: Findings from this study suggest qualitatively different cognitive profiles exist among children with SB, and the relevance of neuropsychological functioning for day-to-day adaptive functioning and quality of life. Clinical implications and future research are discussed. (JINS, 2016, 22, 804-815)

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Spinal lesion level in spina bifida: a source of neural and cognitive heterogeneity

Abstract: A higher level of spinal lesion in SBM-H is a marker for more severe anomalous brain development, which is in turn associated with poorer neurobehavioral outcomes in a wide variety of domains that determine levels of independent functioning for these children at home and school.

Cited by 153 publications

References 19 publications

Neocortical reorganization in spina bifida

Neocortical reorganization in spina bifida

A tail of sacral agenesis: delayed presentation of meningocele in sacral agenesis

Profiles of Neuropsychological Functioning in Children and Adolescents with Spina Bifida: Associations with Biopsychosocial Predictors and Functional Outcomes

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