Altered brain connectivity in sagittal craniosynostosis

Abstract

<jats:sec> <jats:title>Object</jats:title> <jats:p>Sagittal nonsyndromic craniosynostosis (sNSC) is the most common form of NSC. The condition is associated with a high prevalence (&gt; 50%) of deficits in executive function. The authors employed diffusion tensor imaging (DTI) and functional MRI to evaluate whether hypothesized structural and functional connectivity differences underlie the observed neurocognitive morbidity of sNSC.</jats:p></jats:sec> <jats:sec> <jats:title>Methods</jats:title> <jats:p>Using a 3-T Siemens Trio MRI system, the authors collected DTI and resting-state functional connectivity MRI data in 8 adolescent patients (mean age 12.3 years) with sNSC that had been previously corrected via total vault cranioplasty and 8 control children (mean age 12.3 years) without craniosynostosis. Data were analyzed using the FMRIB Software Library and BioImageSuite.</jats:p></jats:sec> <jats:sec> <jats:title>Results</jats:title> <jats:p>Analyses of the DTI data revealed white matter alterations approaching statistical significance in all supratentorial lobes. Statistically significant group differences (sNSC &lt; control group) in mean diffusivity were localized to the right supramarginal gyrus. Analysis of the resting-state seed in relation to whole-brain data revealed significant increases in negative connectivity (anticorrelations) of Brodmann area 8 to the prefrontal cortex (Montreal Neurological Institute [MNI] center of mass coordinates [x, y, z]: −6, 53, 6) and anterior cingulate cortex (MNI coordinates 6, 43, 14) in the sNSC group relative to controls. Furthermore, in the sNSC patients versus controls, the Brodmann area 7, 39, and 40 seed had decreased connectivity to left angular gyrus (MNI coordinates −31, −61, 34), posterior cingulate cortex (MNI coordinates 13, −52, 18), precuneus (MNI coordinates 10, −55, 54), left and right parahippocampus (MNI coordinates −13, −52, 2 and MNI coordinates 11, −50, 2, respectively), lingual (MNI coordinates −11, −86, −10), and fusiform gyri (MNI coordinates −30, −79, −18). Intrinsic connectivity analysis also revealed altered connectivity between central nodes in the default mode network in sNSC relative to controls; the left and right posterior cingulate cortices (MNI coordinates −5, −35, 34 and MNI coordinates 6, −42, 39, respectively) were negatively correlated to right hemisphere precuneus (MNI coordinates 6, −71, 46), while the left ventromedial prefrontal cortex (MNI coordinates 6, 34, −8) was negatively correlated to right middle frontal gyrus (MNI coordinates 40, 4, 33). All group comparisons (sNSC vs controls) were conducted at a whole brain–corrected threshold of p &lt; 0.05.</jats:p></jats:sec> <jats:sec> <jats:title>Conclusions</jats:title> <jats:p>This study demonstrates altered neocortical structural and functional connectivity in sNSC that may, in part or substantially, underlie the neuropsychological deficits commonly reported in this population. Future studies combining analysis of multimodal MRI and clinical characterization data in larger samples of participants are warranted.</jats:p></jats:sec>

Language, learning, and memory in children with and without single-suture craniosynostosis

“…4,6,9,13,15,23,34,41,48 The etiology of the neurodevelopmental consequences and correlates of SSC remains unclear, with some hypotheses focusing on the effects of the fused suture on brain development [1][2][3]47,49 and others emphasizing preexisting neuropathology that may affect both brain and cranium. 5,19 These studies provide some evidence that changes in brain morphology present prior to surgery persist after surgery. 2 There is also evidence that changes in auditory functioning, potentially related to later language and literacy development, 29,37 are present prior to surgical intervention for infants with SSC.…”

“…19 Functional changes in brain circuitry known to be associated with language and number processing, memory retrieval, and reading have been identified in older children with sagittal SSC who underwent surgery as infants. 5 Further research examining brain morphology and function prior to and after cranioplasty is needed to clarify these issues.…”

Diffusion Tensor Imaging and Fiber Tractography in Children with Craniosynostosis Syndromes

“…We assume that the role of the central CSF spaces is at least as big as the genetic influence in causing WM alterations. As in patients with sagittal craniosynostosis in whom altered DTI parameters may underlie their neuropsychological deficits, 37 WM abnormalities of the cingulate gyrus and corpus callosum in syndromic patients with craniosynostosis syndromes may be responsible for existing attention and memory problems. 14 However, neurologic assessment of our patients with craniosynostosis syndromes cannot explain diffusivity abnormalities in the corticospinal tracts, and motor deficits might rather be caused by impair-ment of the frontal WM.…”

Una revisión de la literatura acerca de las características neuropsicológicas de niños con craneosinostosis simple en diferentes edades

“…Con el uso de doppler transcraneal, realizando una comparación pre y postquirúrgica en 10 pacientes con craneosinostosis tanto sindrómicas como simples, se reportó que después de la cirugía de corrección aumentó el flujo sanguíneo 16 . Por su parte, Beckett et al 17 reportaron que 8 adolescentes postoperados de escafocefalia presentaron alteraciones en las conexiones de la corteza prefrontal, el lóbulo parietal superior y los giros angular y supramarginal en una comparación de grupos.…”