Plastic Surgery Research Council

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Frustration and Emotional Regulation in Nonsyndromic Craniosynostosis: an fMRI Study
Robin T. Wu, BS, Jenny F. Yang, MD, William B. Zucconi, DO, Cheryl Lacadie, BS, Matthew Swallow, BS, Andrew Timberlake, PhD, Alexander H. Sun, BS, Raysa Cabrejo, BS, Jeffrey Eilbott, BS, Carolyn Chuang, MD, Eric Brooks, MD, Derek M. Steinbacher, MD, DMD, Kevin Pelphrey, PhD, John A. Persing, MD.
Yale University School of Medicine, New Haven, CT, USA.

PURPOSE: Nonsyndromic craniosynostosis (NSC) may manifest with complex behavioral, attentional, and emotional sequelae. We seek to characterize higher level brain connectivity with the first-reported use of task-based fMRI analysis in adolescent NSC patients.
METHODS: Surgically corrected patients age >9 with NSC and age/gender/handedness matched controls were recruited. fMRI data was gathered with a 3T Siemens TIM Trio scanner. Participants were presented with a standard GoNoGo task. Accumulated points were displayed every 20 frames with a target goal of 1000 to win a prize. NoGo error rate was maintained at 50+10% by adjusting stimulus duration to control difficulty. Unbeknownst to subjects, trials were structured as “win/lose/recovery” paradigms to induce frustration and index emotional regulation. “Win” blocks allowed point increases, “lose” blocks led to net negative points, and “recovery” blocks allowed re-accumulation to ultimately win the prize. BioImage Suite was used to analyze whole-brain intrinsic connectivity between tasks with cluster-corrected group-level T-maps. P<0.05 was significant.
RESULTS: Seven right unilateral-coronal (ULC; average age 12.2, 3 females), six metopic (MCS; average age 11.5 years, 2 females), and respective matched controls were included. During “win”, ULC displayed significantly increased activity in the right inferior temporal gyrus(p<0.05), calcaneus(p<0.03), and cerebellar vermis(p<0.01), and reductions in the right parietal operculum of the supramarginal gyrus(SMG; p<0.03). “Lose” induced decreased left superior/middle frontal(p<0.05), left precuneus(p<0.01) and left pre/post central gyri activity(p<0.03). In “recovery”, ULC cohort experienced increases in the left pre/post central gyri (p<0.05), left inferior temporal gyri(p<0.02), left paracentral lobule(p<0.01), bilateral precuneus/calcaneus (p<0.01), right anterior subinsula(p<0.05), and cerebellar vermis(p<0.01). MCS participants during “win” demonstrated significantly decreased activity in the left supramarginal/angular gyrus(p<0.03) and bilateral posterior cingulate(p<0.01). “Lose” induced decreased activity in similar regions with additional increases at the anterior paracentral lobule/supplementary motor area(p<0.03). “Recovery” prompted similar suppression in the posterior cingulate gyrus(p<0.05) and activation in the anterior paracentral lobule/supplementary motor area(p<0.05). Sagittal synostosis studies are ongoing.
CONCLUSION: NSC adolescents respond variably to reward, frustration, and emotional regulation. During “win” and “recovery” R-ULC patients evidenced increased activity in regions associated with visual recognition/processing and motor coordination. “Lose” prompted broad suppression ipsilateral to the calvarial defect, particularly in areas responsible for inhibition, suggesting poor self-control when frustrated. ULC patients experienced a surge of activity during “recovery” in reward regulation and visual processing, suggesting possible difficulties in down-regulating emotional based sensory input. Importantly, while ULC morphological defects are isolated to the anterior cranium, posterior brain connectivity were influenced. MSO adolescents had comparable activity suppression across all conditions, located in regions producing emotional regulation and recognition. This suggests global deficits but less pronounced dynamic processing differences. This study provides insight into higher level neural networks in patients with NSC, providing evidence for targeted neuropsychiatric therapy depending on the suture phenotype.


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