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An Investigation of Brain Functional Connectivity by Form of Craniosynostosis

Sun, Alexander H. MD, MHS*; Eilbott, Jeffrey; Chuang, Carolyn MD, MHS*; Yang, Jenny F. MD, MHS*; Brooks, Eric D. MD, MHS*; Beckett, Joel MD, MHS*; Steinbacher, Derek M. MD, FACS*; Pelphrey, Kevin PhD; Persing, John A. MD*

doi: 10.1097/SCS.0000000000005537
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Purpose: Long-term neurocognitive sequelae of nonsyndromic craniosynostosis (NSC) patients are just beginning to be clarified. This study uses functional MRI (fMRI) to determine if there is evidence of altered brain functional connectivity in NSC, and whether these aberrations vary by form of synostosis.

Methods: Twenty adolescent participants with surgically treated NSC (10 sagittal synostosis, 5 right unilateral coronal synostosis [UCS], 5 metopic synostosis [MSO]) were individually matched to controls by age, gender, and handedness. A subgroup of MSO was classified as severe metopic synostosis (SMS) based on the endocranial bifrontal angle. Resting state fMRI was acquired in a 3T Siemens TIM Trio scanner (Erlangen, Germany), and data were motion corrected and then analyzed with BioImage Suite (Yale School of Medicine). Resulting group-level t-maps were cluster corrected with nonparametric permutation tests. A region of interest analysis was performed based on the left Brodmann's Areas 7, 39, and 40.

Results: Sagittal synostosis had decreased whole-brain intrinsic connectivity compared to controls in the superior parietal lobules and the angular gyrus (P = 0.071). Unilateral coronal synostosis had decreased intrinsic connectivity throughout the prefrontal cortex (P = 0.031). The MSO cohort did not have significant findings on intrinsic connectivity, but the SMS subgroup had significantly decreased connectivity among multiple subcortical structures.

Conclusion: Sagittal synostosis had decreased connectivity in regions associated with visuomotor integration and attention, while UCS had decreased connectivity in circuits crucial in executive function and cognition. Finally, severity of metopic synostosis may influence the degree of neurocognitive aberration. This study provides data suggestive of long-term sequelae of NSC that varies by suture type, which may underlie different phenotypes of neurocognitive impairment.

*Section of Plastic and Reconstructive Surgery, Yale School of Medicine

Yale Child Study Center, Yale School of Medicine, New Haven, CT.

Address correspondence and reprint requests to John A. Persing, MD, Yale Plastic Surgery, PO Box 208062, New Haven, CT 06510; E-mail: john.persing@yale.edu

Received 24 June, 2018

Accepted 3 March, 2019

Parts of this research were supported by grants from the American Society of Maxillofacial Surgeons and Komedyplast awarded to John Persing. Alexander Sun, Carolyn Chuang, and Eric Brooks received research support from the NIH Clinical Translational Science Award Multidisciplinary Pre-Doctoral Training Program in Translational Research (NIH CTSA TL1).

The authors report no conflicts of interest.

© 2019 by Mutaz B. Habal, MD.