title: Assessment of white matter microstructural integrity in children with syndromic craniosynostosis: a diffusion-tensor imaging study.
abstract: OBJECTIVE
  To assess whether architectural alterations exist in the white matter of patients with syndromic and complex craniosynostosis.
  
  
  METHODS
  The medical ethics committee approved this study. Written informed consent was obtained from parents or guardians before imaging. A prospective study was performed in children with syndromic and complex craniosynostosis aged 6-14 years. Forty-five patients were included: four had Apert syndrome, 14 had Crouzon-Pfeiffer syndrome, eight had Muenke syndrome, 11 had Saethre-Chotzen syndrome, and eight had complex craniosynostosis. In addition, seven control subjects were evaluated. For diffusion-tensor imaging, an echo-planar sequence was used with a diffusion gradient (b = 1000 sec/mm(2)) applied in 25 noncollinear directions. Regions of interest (ROIs) were placed in the following white matter structures: pontine crossing tract, corticospinal tracts, medial cerebral peduncles, uncinate fasciculus (measured bilaterally), anterior commissure, frontal and occipital white matter (measured bilaterally), fornix, corpus callosum (measured in the genu and splenium), and corpus cingulum (measured bilaterally). Eigenvalues were measured in all ROIs and fractional anisotropy (FA) was calculated.
  
  
  RESULTS
  Across all measured ROIs, FA values were generally lower in all patients combined than in the control subjects (P < .001). There were no significant differences among subgroups of patients.
  
  
  CONCLUSIONS
  Diffusion-tensor imaging measurements of white matter tracts reveal significant white matter integrity differences between children with craniosynostosis and healthy control subjects. This could imply that the developmental delays seen in these patients could be caused by the presence of a primary disorder of the white matter microarchitecture.
authors.affiliation: Department of Plastic, Reconstructive, and Hand Surgery, Erasmus Medical Center, Sophia Children's Hospital, PO Box 2040, Room Ee 15.91, 3000 CA Rotterdam, The Netherlands. j.florisson@erasmusmc.nl
authors.forename: Joyce M G
authors.initials: JM
authors.lastname: Florisson
authors.forename: Jeroen
authors.initials: J
authors.lastname: Dudink
authors.forename: Irene V
authors.initials: IV
authors.lastname: Koning
authors.forename: Wim C J
authors.initials: WC
authors.lastname: Hop
authors.forename: Marie-Lise C
authors.initials: ML
authors.lastname: van Veelen
authors.forename: Irene M J
authors.initials: IM
authors.lastname: Mathijssen
authors.forename: Maarten H
authors.initials: MH
authors.lastname: Lequin
mesh_terms: D000293:Adolescent; D000704:Analysis of Variance; D016022:Case-Control Studies; D002648:Child; D003398:Craniosynostoses / Q000473:pathology*; D038524:Diffusion Magnetic Resonance Imaging / Q000379:methods*; D005260:Female; D006801:Humans; D008297:Male; D009413:Nerve Fibers, Myelinated / Q000473:pathology*; D011446:Prospective Studies; D015203:Reproducibility of Results; D012680:Sensitivity and Specificity; D018709:Statistics, Nonparametric