Normalization of brain morphology after surgery in sagittal craniosynostosis

Abstract

<jats:sec> <jats:title>OBJECT</jats:title> <jats:p>Nonsyndromic craniosynostosis (NSC) is associated with significant learning disability later in life. Surgical reconstruction is typically performed before 1 year of age to correct the cranial vault morphology and to allow for normalized brain growth with the goal of improving cognitive function. Yet, no studies have assessed to what extent normalized brain growth is actually achieved. Recent advances in MRI have allowed for automated methods of objectively assessing subtle and pronounced brain morphological differences. The authors used one such technique, deformation-based morphometry (DBM) Jacobian mapping, to determine how previously treated adolescents with sagittal NSC (sNSC) significantly differ in brain anatomy compared with healthy matched controls up to 11.5 years after surgery.</jats:p></jats:sec> <jats:sec> <jats:title>METHODS</jats:title> <jats:p>Eight adolescent patients with sNSC, previously treated via whole-vault cranioplasty at a mean age of 7 months, and 8 age- and IQ-matched control subjects without craniosynostosis (mean age for both groups = 12.3 years), underwent functional 3-T MRI. Statistically significant group tissue-volume differences were assessed using DBM, a whole-brain technique that estimates morphological differences between 2 groups at each voxel (p &lt; 0.01). Group-wise Jacobian volume maps were generated using a spacing of 1.5 mm and a resolution of 1.05 × 1.05 × 1.05 mm<jats:sup>3</jats:sup>.</jats:p></jats:sec> <jats:sec> <jats:title>RESULTS</jats:title> <jats:p>There were no significant areas of volume reduction or expansion in any brain areas in adolescents with sNSC compared with controls at a significance level of p &lt; 0.01. At the more liberal threshold of p &lt; 0.05, two areas of brain expansion extending anteroposteriorly in the right temporooccipital and left frontoparietal regions appeared in patients with sNSC compared with controls.</jats:p></jats:sec> <jats:sec> <jats:title>CONCLUSIONS</jats:title> <jats:p>Compared with previous reports on untreated infants with sNSC, adolescents with sNSC in this cohort had few areas of brain dysmorphology many years after surgery. This result suggests that comprehensive cranioplasty performed at an early age offers substantial brain normalization by adolescence, but also that some effects of vault constriction may still persist after treatment. Specifically, few areas of expansion in frontoparietal and temporooccipital regions may persist. Overall, data from this small cohort support the primary goal of surgery in allowing for more normalized brain growth. Larger samples, and correlating degree of normalization with cognitive performance in NSC, are warranted.</jats:p></jats:sec>

Examining the Role of Early Diagnostic Imaging for Craniosynostosis in the Era of Endoscopic Suturectomy: A Single Institution Experience

“…6,7 Although the data regarding the cognitive impact of synostosis is evolving, recent neuropsychological studies suggest that earlier surgery in craniosynostosis may improve subsequent neurodeve-lopmental outcomes. [8][9][10][11][12][13] As endoscopic suturectomy has become increasingly accepted as standard of care, intervention is occurring in a younger cohort of children. Although our institution tends to rely almost uniformly on physical examination for the diagnosis of synostosis, there was a concern that this practice may be altered in unclear cases with limited time for observation given the need to make an expedient diagnosis and identify endoscopic surgical candidates at a younger age.…”

Long-term Follow-up of Preoperative Infant Event-related Potentials in School-age Children with Craniosynostosis

“…13,14 In particular, results from deformation-based morphometry Jacobian mapping, which uses automated methods of objectively quantifying volumetric changes, showed no differences in surgically-treated adolescents with sagittal NSC compared with controls. 15 Another study examining distances between 32 identifiable cortical and subcortical brain structures found that after surgery, the relative positions of structures in the brain still deviated from normal, although the overall brain shape reorganized to a morphology somewhere closer to normal, and where it was preoperatively. 16 These findings are encouraging, as timely intervention may have permitted significant recovery even in those with more severe presurgical neural attenuation.…”

Predicting calvarial morphology in sagittal craniosynostosis

“…In the case of sagittal synostosis this method can be used to compare the existing techniques for the management of this condition and their potential impacts on the brain development. The contact pressure maps that FE models provide us, together with the functional brain imaging data, can advance our understand of the interplay between calvarial reconstruction and brain development 31,32 .…”