Purpose:Feminization of the forehead is a standard component of gender-affirming surgery. The purpose of this study was to quantify the preoperative to postoperative changes in forehead morphology associated with facial feminization surgery (FFS) using computerized tomography (CT) imaging and a novel segmentation technique. Methods:All patients underwent FFS at an academic medical center. Only patients who underwent upper third procedures and had preoperative and postoperative skull craniofacial CT imaging available were included. Measurements of forehead volume, surface area, frontonasal angle, and bossing angle were made with Mimics (Materialise, Leuven, Belgium). Three reviewers measured the scans independently, and intraclass coefficients were obtained to assess inter-rater reliability. Patient satisfaction was assessed preoperatively and postoperatively using the FACE-Q, a patient-reported outcome measure. Results:Twenty-two patients met the inclusion criteria. Average age at the time of surgery was 36 ± 11 years. All patients underwent frontal sinus setback, forehead contouring, and suprabrow contouring. On average, forehead volume decreased by 2,558 mm3 (p < 0.001), surface area decreased by 505 mm2 (p < 0.001), frontonasal angle decreased by 9.6° (p < 0.001), and bossing angle decreased by 9.9° (p < 0.001). Intraclass coefficients ranged from 0.62 to 0.87. Patients reported significant improvement in satisfaction postsurgery across a majority of FACE-Q modules. Conclusion:A measurable reduction of forehead dimensions, frontonasal angle, and bossing angle successfully aligns facial features with more feminine ideals, drawing attention to the critical role of FFS in facilitating gender transition for a patient and improving their overall quality of life.

While previous studies have investigated behavioral development after non-syndromic sagittal craniosynostosis (NSSC) repair, there is limited information available about the influence of surgical technique. This study examined behavioral differences among patients with NSSC following cranial vault remodeling (CVR) versus strip craniectomy (SC). Parents of patients ages 6–18 years old with NSSC repaired before 6 months via either CVR or SC completed four behavioral surveys: Conners Short 3rd Edition (ADHD), Social Responsiveness Scale 2nd edition (ASD), Behavior Rating Inventory of Executive Function 2nd Edition (executive functioning), and Child Behavior Checklist (emotional/behavioral regulation). Survey results were converted to t-scores based on a standardized population. Individual scale t-scores were compared between surgical techniques, as well as the number of patients with elevated scores (t-score ≥ 60). The distribution of t-score categories for each scale over the entire cohort was compared against the expected population distribution. Forty patients were enrolled, with 21 CVR and 19 SC. Age at surgery (4.78 ± 0.94 months for CVR vs. 2.81 ± 0.84 months for SC, p < 0.001) was the only significant demographic difference. There were no significant differences between individual scale t-scores or the number of patients with elevated scores. However, the following scales had t-score distributions that were significantly elevated compared to the expected population distribution: inattention, hyperactivity, executive functioning, and peer relations. When controlling for prompt surgical timing, CVR and SC demonstrated comparable behavioral risk profiles. However, providers should still regularly screen NSSC patients for behavioral problems.

OBJECTIVE Deformational Plagiocephaly (DP) is commonly treated with cranial orthosis, or helmet therapy. A large, national study on the impact of insurance status on helmet outcomes is lacking. We assessed treatment outcomes for helmet therapy based on insurance status. DESIGN This was a retrospective data analysis of patients referred to Cranial Technologies, Inc for helmet therapy between 2014-2020 across 21 states. PATIENTS, PARTICIPANTS There were a total of 211,417 patients referred for helmeting, of whom 141,513 received helmet therapy. MAIN OUTCOMES MEASURES Multivariate regression was used to assess the relationship of insurance status with post-treatment residual flattening, measured by cephalic index (CI) and cranial vault asymmetry index (CVAI), and treating provider rating of success. RESULTS Patients with Medicaid were more likely to complete treatment with residual flattening measured by CI and CVAI when compared to patients with private insurance (OR: 1.58, CI: 1.51-1.65, p < 0.001 and OR: 1.21, CI: 1.15-1.28, p < 0.001, respectively). Providers of patients with Medicaid were more likely to give a low rating of success following treatment (OR: 3.25, CI: 2.70-3.92, p < 0.001). CONCLUSIONS Our study investigating the impact of insurance status on helmet therapy across 21 states found that patients with Medicaid were more likely to experience residual flattening and have lower provider-rated outcomes compared to those with commercial insurance. Given significant caregiver burden posed by helmet therapy, which requires frequent visits and consistent helmet use, caregivers of patients with Medicaid may require greater support to reduce outcome disparities observed here.

Virtual surgical planning (VSP) has benefits in craniofacial surgery with growing popularity. However, while specific use cases are highlighted in the literature, no studies exist providing an overview of VSP use among craniofacial surgeons, and little is known about the extent of exposure to VSP during plastic surgery training. This study surveyed members of The American Society of Maxillofacial Surgeons (ASMS) to better characterize both the landscape of VSP use among practicing craniofacial surgeons and the extent of exposure to VSP throughout surgical training. An electronic survey was administered in the fall of 2023. Response data included surgeon demographics, VSP usage, including the use in residency/fellowship, procedures for which VSP is used, and assessment of VSP's impact on the surgeon's practice. Demographics and VSP use were analyzed using descriptive statistics, while categorical and continuous variables were analyzed using χ2 tests and t-tests, respectively. Of the 44 respondents, 40 (90.9%) completed a craniofacial surgery fellowship, and 18 (40.9%) utilized VSP in either residency or fellowship. In respondents' current practice, VSP is utilized most commonly for orthognathic surgery (n=32, 91.4%), postablative reconstruction (n=23, 82.1%), and facial feminization (n=11, 73.3%). Shorter operative time and improved esthetic outcomes were frequently reported as benefits derived from VSP use. Finally, surgeons in practice for less than 10 years were significantly more likely to have used VSP in both residency (OR=20.3, P<0.01) and in fellowship (OR=40.6, P<0.01) than those practicing for more than 10 years. These findings suggest that craniofacial surgeons apply VSP more commonly for certain procedure types. Our results additionally suggest that incorporation of VSP into residency and fellowship training has become significantly more common over time, with a pivot towards integration in the last decade.

Purpose Nonsyndromic craniosynostosis (NSC) is associated with neurocognitive deficits, and intervention at infancy is standard of care to limit the negative effects of NSC on brain development. In this study, diffusion tensor imaging (DTI) was implemented to investigate white matter microstructure in infants with NSC undergoing cranial vault remodeling, and a comparison was made with white matter development in neurotypical controls. Methods Infants presenting with NSC ( n = 12) underwent DTI scans before and after cranial vault remodeling. Neurotypical infants ( n = 5), age matched to NSC patients at preoperative scans, were compared to preoperative DTI scans. Pre- and postoperative NSC scans were compared in aggregate, and the sagittal synostosis ( n = 8) patients were evaluated separately. Finally, neurotypical infants from the University of North Carolina/University of New Mexico Baby Connectome Project (BCP), who underwent DTI scans at timepoints matching the NSC pre- and postoperative DTI scans, were analyzed ( n = 9). Trends over the same time period were compared between NSC and BCP scans. Results No significant differences were found between preoperative NSC scans and controls. White matter development was more limited in NSC patients than in BCP patients, with microstructural parameters of the corpus body and genu and inferior and superior longitudinal fasciculi consistently lagging behind developmental changes observed in healthy patients. Conclusion Infant white matter development appears more limited in NSC patients undergoing cranial vault remodeling relative to that in neurotypical controls. Further investigation is needed to explore these differences and the specific effects of early surgical intervention.

OBJECTIVE:Previous work identified an association between genetics and neurodevelopmental delays in patients with nonsyndromic craniosynostosis. The authors investigated the role of genetic mutations on behavioral outcomes of patients with treated sagittal synostosis. METHODS:Parents of children aged 6-18 years with surgically corrected sagittal synostosis were recruited to complete the Child Behavioral Checklist (overall behavioral problems), Conners 3rd Edition-Parent (attention-deficit/hyperactivity disorder), Social Responsiveness Scale 2nd Edition (autism spectrum disorder [ASD]), and Behavior Rating Inventory of Executive Function 2nd Edition (executive function). Genomic analysis was completed, and patients were identified if they had mutations in high probability of loss of function intolerant (pLI) genes (high pLI vs nonhigh pLI). Genetic burden was assessed relative to controls. Multivariate linear regression determined the association of mutations in high pLI genes with behavioral scores, while controlling for sociodemographic factors, age at surgery, surgery type, and IQ. RESULTS:Sixteen of 45 patients were in the high pLI group. There were no differences between the groups in terms of sociodemographic factors. A greater proportion of children in the high pLI group scored at or above borderline clinical levels for aggression (18.8% vs 0.0%, p = 0.05) and externalizing problems (31.3% vs 3.7%, p = 0.02). Among children in the nonhigh pLI group, older age at surgery was associated with worse scores on the rule-breaking, aggression, and externalizing problems domains and four out of five ASD domains. CONCLUSIONS:Children with treated nonsyndromic sagittal synostosis and mutations in high pLI genes had worse behavioral problems in externalizing behaviors and aggression, whereas older age at surgery was a significant predictor of worse behavioral outcomes in patients without mutations in high pLI genes.

Children with metopic synostosis have been found to have more neurocognitive and behavioral difficulties. The variables that may affect future neurodevelopmental outcomes, including presenting morphologic severity, have not been fully studied. In the largest study to date, we aimed to assess what portends worse neurocognitive and behavioral outcomes at school age. Children 6–18 years old with surgically corrected metopic nonsyndromic craniosynostosis underwent neurocognitive testing. Parents completed behavior rating surveys about their child: Conners-3 (ADHD), Social Responsiveness Scale-2 (autism spectrum disorder), Behavior Rating Inventory of Executive Function-2 (BRIEF-2: executive function), and Child’s Behavior Checklist (overall behavior). The endocranial bifrontal angle (EBA), adjusted EBA (aEBA), frontal angle (FA), and AI-derived metopic severity score (MSS) were determined on pre-operative CT images. Multivariate linear regressions were used to evaluate the association of age at surgery and severity. There were 87 children who underwent neurocognitive testing (average age 10.9 ± 3.3 years) of whom 67 also completed behavioral assessments. Greater phenotypical severity of metopic synostosis (lower FA, aEBA, and EBA) was associated with worse scores on the subscales of the BRIEF-2 (executive function) and executive subscale of the Conners-3. Increasing age at surgery was associated with worse executive function subscale scores of the Conners-3 when controlling for each severity measurement and sociodemographic risk. Children with greater phenotypic severity of metopic synostosis have worse executive function at school age. The majority of children with metopic synostosis have signs of ADHD. Later surgeries (greater than 12 months) may impact executive functioning, regardless of the degree of severity. Future research should aim at identifying the direct structural changes to the brain.

Children with surgically corrected nonsyndromic craniosynostosis have been previously found to have neurocognitive and behavioral difficulties. Children with metopic synostosis have been described to have more difficulties than children with sagittal synostosis. This study aims to characterize the behavioral differences between children with metopic and sagittal synostosis. Children with metopic and sagittal synostosis were recruited at school age. Parents completed four separated behavioral assessments: Conners-3 (evaluation of ADHD), Social Responsiveness Scale-2 (SRS-2: evaluation of autism), Behavior Rating Inventory of Executive Function-2 (BRIEF-2: evaluation of executive function), and Child Behavior Checklist (CBCL: evaluation of overall behavioral problems). Children underwent intelligence quotient (IQ) testing using the Wechsler Abbreviated Scale of Intelligence (WASI-II). There were 91 children (45 with metopic and 46 with sagittal synostosis). More children with metopic synostosis reported requiring supportive services (57.7

ObjectiveWhile previous literature has investigated the psychosocial impact and aesthetic satisfaction associated with post-operative scarring for certain pediatric craniofacial conditions, the impact of the scar burden resulting from craniosynostosis surgery has not been adequately studied.ParticipantsSCAR-Q was shared with patients ages 8 and older. Thirty-two complete patient responses were recorded.InterventionsSCAR-Q is a PROM that consists of three independent scales - appearance, symptoms, and psychosocial impact - associated with a scar.Main Outcome MeasuresMann-Whitney U, linear regression, and Pearson correlation tests were used to evaluate associations between the scales, in addition to patient characteristics such as sex and suture involvement.ResultsMean ages at time of surgery and survey completion were 9.65 ± 10.10 months and 12.10 ± 3.92 years, respectively. Mean scale scores were 81.5 ± 17.9 for appearance, 86.8 ± 12.4 for symptoms, and 79.3 ± 25.7 for psychosocial impact. Higher patient dissatisfaction with scar appearance correlated with more scar-related symptoms (r = 0.389; p = 0.028) and a greater psychosocial impact (r = 0.725; p < 0.001). SCAR-Q scales did not significantly correlate with age at surgery, age at survey completion, type of synostosis, or type of surgery; however, female patients reported lower mean appearance (65.4 vs. 86.0; p = 0.012) and psychosocial impact (57.3 vs. 85.5; p = 0.010) scores when compared to their male counterparts.ConclusionsIt is vital that surgeons discuss patients’ aesthetic satisfaction following craniosynostosis surgery in order to appropriately address and limit deleterious, long-term physical and psychosocial outcomes.

OBJECTIVE Occurring once in every 2000 live births, craniosynostosis (CS) is the most frequent cranial birth defect. Although the genetic etiologies of syndromic CS cases are well defined, the genetic cause of most nonsyndromic cases remains unknown. METHODS The authors analyzed exome or RNA sequencing data from 876 children with nonsyndromic CS, including 291 case-parent trios and 585 additional probands. The authors also utilized the GeneMatcher platform and the Gabriella Miller Kids First genome sequencing project to identify additional CS patients with AXIN1 mutations. RESULTS The authors describe 11 patients with nonsyndromic CS harboring rare, damaging mutations in AXIN1, an inhibitor of Wnt signaling. AXIN1 regulates signaling upstream of key mediators of osteoblast differentiation. Three of the 6 mutations identified in trios occurred de novo in the proband, while 3 were transmitted from unaffected parents. Patients with nonsyndromic CS were highly enriched for mutations in AXIN1 compared to both expectation (p = 0.0008) and exome sequencing data from > 76,000 healthy controls (p = 2.3 × 10−6), surpassing the thresholds for genome-wide significance. CONCLUSIONS These findings describe the first phenotype associated with mutations in AXIN1, with mutations identified in approximately 1% of nonsyndromic CS cases. The results strengthen the existing link between Wnt signaling and maintenance of cranial suture patency and have implications for genetic testing in families with CS.

Facial feminization surgery (FFS) is characterized by a series of relatively diverse procedures aimed at aligning skeletal and soft tissue facial appearance with one's experienced feminine gender. Although there are several well-described outcomes from surgical techniques, there is no standardized methodology to provide reliable analyses of postoperative FFS outcomes. This paper describes the first reliable and reproducible technique to accurately and consistently measure post-FFS changes to guide surgical planning to optimize patient outcomes.

OBJECTIVE:While studies have shown that access to facial feminization surgery can be restricted by financial and geographic limitations, there is a lack of information on the impact of surgery on the most vulnerable patients. Therefore, this study assessed the impact of social vulnerability and neighborhood socioeconomic disadvantage on patient-reported outcomes after facial feminization surgery. METHODS:Patients were surveyed pre and postoperatively using the FACE-Q Aesthetics Questionnaire and geo-coded using home addresses to obtain social vulnerability index (SVI) and Area Deprivation Index scores. Two sets of Pearson correlation values were calculated: (1) between SVI scores and each of the pre and postoperative FACE-Q modules and (2) between SVI scores and differences between pre and postoperative FACE-Q modules. Univariate linear regression analyses were performed for the latter. All analyses were repeated for Area Deprivation Index scores. RESULTS:Twenty patients participated in this study. Postoperative facial appearance satisfaction positively correlated with total SVI ( r = 0.48, P = 0.031), socioeconomic status theme ( r =0.47, P =0.037), and racial and ethnic minority theme ( r = 0.48, P = 0.031) scores. The difference between pre and postoperative facial appearance satisfaction positively correlated with total SVI (coefficient = 37.40, r = 0.47, P = 0.035), racial and ethnic minority theme (coefficient = 44.00, r = 0.46, P = 0.040), and housing type and transportation theme (coefficient = 46.97, r = 0.46, P = 0.042) scores. CONCLUSION:Patients impacted by greater social vulnerability disproportionally experience the greatest benefit from gender-affirming facial surgery.

Despite previous research supporting patient safety in sports after craniosynostosis surgery, parental anxiety remains high. This study sought to evaluate the role of healthcare providers in guiding patients and families through the decision-making process. Parents of children with repaired craniosynostosis were asked to assess sports involvement and parental decision-making in children ages 6 and older. Questions were framed primarily on 5-point Likert scales. Sport categorizations were made in accordance with the American Academy of Pediatrics. Chi-squared, linear regression, and Pearson correlation tests were used to analyze associations between the questions. Forty-three complete parental responses were recorded. Mean ages at surgery and time of sports entry were 7.93 ± 4.73 months and 4.76 ± 2.14 years, respectively. Eighty-two percent of patients participated in a contact sport. Discussions with the primary surgeon were more impactful on parental decisions about sports participation than those with other healthcare providers (4.04 ± 1.20 vs. 2.69 ± 1.32). Furthermore, children whose parents consulted with the primary surgeon began participating in sports at a younger age (4.0 ± 1.0 vs. 5.8 ± 2.7 years, p = 0.034). The mean comfort level with contact sports (2.8 ± 1.4) was lower than that with limited-contact (3.8 ± 1.1, p = 0.0001) or non-contact (4.4 ± 1.3, p < 0.0001) sports. This study underscores the critical role that healthcare professionals, primarily surgeons, have in guiding families through the decision-making process regarding their children’s participation in contact sports.

BACKGROUND:Facial feminization surgery (FFS) consists of multiple, complex procedures. Well-informed patients have been shown to have better outcomes and expectations. However, there is limited data evaluating FFS patient-oriented material online. This study aims to evaluate the quality and readability of FFS literature online. METHODS:Facial feminization surgery-related terms were queried in Google with location, cookies, and user account information disabled. Websites were analyzed for readability using Flesch Reading Ease and Flesch-Kincaid Reading Grade Level, suitability using the suitability assessment of materials (SAM), and quality using the DISCERN scale. Unpaired t tests and χ2 tests were used to compare the websites of community-based and academic or public institutions. RESULTS:One hundred twenty websites met inclusion criteria (71 community-based and 49 academic). The average reading grade level was 11.68 ± 1.71. The average SAM score was 53.11 ± 11.75, denoting adequate readability. Only 16.67% of websites contained visual aids to assist in explaining procedures and benefits. Overall, 68.33% of websites' DISCERN score was rated very poor or poor, whereas only 8.33% were rated as good. The following DISCERN elements had the lowest scores across all graded websites: "clear source and date of information," "details of additional source of support," "refers to areas of uncertainty," and "describes risks of each treatment." Websites published by community-based institutions were significantly better in describing how FFS works and the benefits of each procedure. Academic sites overall were less biased (4.84 vs 4.62, P = 0.03) and provided additional sources of support (2.35 vs 1.32, P = <0.001). CONCLUSIONS:Online FFS patient resources should be written at a more inclusive reading level and should increase the use of pictorial aids to improve patient comprehension. Facial feminization surgery patient resources were significantly above the recommended sixth grade reading level for health literature. Academic and community-based institutions should include more information about procedural risks and limitations in surgical outcomes to ensure a broader scope of understanding.

BACKGROUND:Previous work has identified an association between de novo and transmitted loss-of-function mutations in genes under high evolutionary constraint with neurodevelopmental delays in nonsyndromic craniosynostosis (NSC). The authors sought to quantify the neurocognitive effect of these genetic lesions.METHODS:In a prospective, double-blinded cohort study, demographic surveys and neurocognitive tests were administered to patients recruited from a national sample of children with sagittal NSC. Scores for academic achievement, Full-Scale Intelligence Quotient (FSIQ), and visuomotor skills were directly compared between patients with and without damaging mutations in genes with a high probability of loss of function intolerance using two-tailed t tests. Analysis of covariance was also used to compare test scores while controlling for surgery type, age at surgery, and sociodemographic risk.RESULTS:Fifty-six patients completed neurocognitive testing, 18 of whom had a mutation in a highly constrained gene. There was no significant difference between groups in any sociodemographic factors. After controlling for patient factors, patients with high-risk mutations had poorer performance compared with patients without high-risk mutations in every testing category, with significant differences in FSIQ (102.9 ± 11.4 versus 110.1 ± 11.3; P = 0.033) and visuomotor integration (100.0 ± 11.9 versus 105.2 ± 9.5; P = 0.003). There were no significant differences in neurocognitive outcome when stratifying groups based on type of surgery or age at time of surgery.CONCLUSIONS:Even after controlling for exogenous factors, the presence of mutations in high-risk genes led to poorer neurocognitive outcomes. High-risk genotypes may predispose individuals with NSC to deficits, particularly in FSIQ and visuomotor integration.CLINICAL QUESTION/LEVEL OF EVIDENCE:Risk, II.

PURPOSE: Nonsyndromic craniosynostosis is associated with neurocognitive and behavioral deficits. We examined the association of age at surgery and long-term behavioral functioning at school age in children with surgically-corrected metopic synostosis. METHODS: Parents of children ages 6-18 years old with metopic synostosis completed the Child’s Behavioral Checklist (CBCL), Conners 3rd Edition (Conners-3), Social Responsiveness Scale 2nd Edition (SRS-2), and Behavior Rating Inventory of Executive Function 2nd Edition (BRIEF-2). The Conners-3 assesses features of ADHD, the SRS-2 assesses features of autism spectrum disorder, and the CBCL and BRIEF-2 broadly assess behavioral and emotional functioning. Multivariate linear regression was used to evaluate the association of behavioral scores with age at surgery while controlling for sociodemographic factors. RESULTS: The parents of 41 patients completed surveys. Average age at surgery was 8.46±5.34 months, and average age at assessment was 10.42±3.62 years. Average scores were greatest for the peer relations and inattention subscales of the Conners-3 with 22 (53.7%) and 20 (48.8%) patients scoring at or above borderline clinical levels, respectively. Greater age at surgery was associated with significantly higher scores on the inattention (B=1.40, p=0.02) and executive function (B=1.37, p=0.01) subscales of the Conners-3 and cognitive regulation index (B=1.01, p=0.03) and global executive composite (B=1.05, p=0.03) of the BRIEF-2. CONCLUSIONS: Nearly half of patients with surgically-corrected metopic synostosis reached borderline clinical scores for attention and peer relations. Greater age at surgery was associated with features of ADHD and worse executive function. Prompt surgical correction of metopic synostosis may benefit long-term emotional and behavioral function.

Craniosynostosis (CS) is the most common congenital cranial anomaly. Several Mendelian forms of syndromic CS are well described, but a genetic etiology remains elusive in a substantial fraction of probands. Analysis of exome sequence data from 526 proband-parent trios with syndromic CS identified a marked excess (observed 98, expected 33, p = 4.83 3 10-20) of damaging de novo variants (DNVs) in genes highly intolerant to loss-of-function variation (probability of LoF intolerance > 0.9). 30 probands harbored damaging DNVs in 21 genes that were not previously implicated in CS but are involved in chromatin modification and remodeling (4.7-fold enrichment, p = 1.1 3 10-11). 17 genes had multiple damaging DNVs, and 13 genes (CDK13, NFIX, ADNP, KMT5B, SON, ARID1B, CASK, CHD7, MED13L, PSMD12, POLR2A, CHD3, and SETBP1) surpassed thresholds for genome-wide significance. A recurrent gain-of-function DNV in the ret-inoic acid receptor alpha (RARA; c.865G>A [p.Gly289Arg]) was identified in two probands with similar CS phenotypes. CS risk genes overlap with those identified for autism and other neurodevelopmental disorders, are highly expressed in cranial neural crest cells, and converge in networks that regulate chromatin modification, gene transcription, and osteoblast differentiation. Our results identify several CS loci and have major implications for genetic testing and counseling.

BACKGROUND:Craniofacial surgery is the standard treatment for children with moderate to severe trigonocephaly. The added value of surgery to release restriction of the frontal lobes is unproven, however. In this study, the authors aim to address the hypothesis that the frontal lobe perfusion is not restricted in trigonocephaly patients by investigating cerebral blood flow.METHODS:Between 2018 and 2020, trigonocephaly patients for whom a surgical correction was considered underwent magnetic resonance imaging brain studies with arterial spin labeling to measure cerebral perfusion. The mean value of cerebral blood flow in the frontal lobe was calculated for each subject and compared to that of healthy controls.RESULTS:Magnetic resonance imaging scans of 36 trigonocephaly patients (median age, 0.5 years; interquartile range, 0.3; 11 female patients) were included and compared to those of 16 controls (median age, 0.83 years; interquartile range, 0.56; 10 female patients). The mean cerebral blood flow values in the frontal lobe of the trigonocephaly patients (73.0 ml/100 g/min; SE, 2.97 ml/100 g/min) were not significantly different in comparison to control values (70.5 ml/100 g/min; SE, 4.45 ml/100 g/min; p = 0.65). The superior, middle, and inferior gyri of the frontal lobe showed no significant differences either.CONCLUSIONS:The authors' findings suggest that the frontal lobes of trigonocephaly patients aged less than 18 months have a normal cerebral blood flow before surgery. In addition to the very low prevalence of papilledema or impaired skull growth previously reported, this finding further supports the authors' hypothesis that craniofacial surgery for trigonocephaly is rarely indicated for signs of raised intracranial pressure or restricted perfusion for patients younger than 18 months.CLINICAL QUESTION/LEVEL OF EVIDENCE:Risk, II.

Background: Radiographic severity of metopic synostosis has been suggested as a predictor of long-term neurocognitive outcomes, and artificial intelligence (AI) has recently been used to quantify severity. Age at surgery is predictive of long-term neurocognition in sagittal synostosis but has not been adequately explored in metopic synostosis. Methods: Children ages 6 to 18 years old with corrected metopic synostosis underwent testing of intelligence quotient (IQ), academic achievement, and visuomotor integration (VMI). Various manual measurements and AI-derived severity scores were determined. Scans were categorized as moderate or severe for head-to-head comparisons and multivariable linear regressions were used to assess the relationship of age at surgery and severity with neurocognitive outcomes. Results: 41 patients with average age at testing of 10.8 ± 3.4 years were included. 18 patients were in the severe group while 23 patients were in the moderate group with average ages at surgery 6.6 ± 2.7 and 10.6 ± 8.4 months, respectively (p = 0.062). Greater AI-derived severity was significantly associated with lower reading comprehension (p = 0.040 and 0.018) and reading composite scores (p = 0.024 and p = 0.008). Older age at surgery was significantly associated with lower VMI scores (p-values ranging from 0.017 to 0.045) and reading composite scores (p = 0.047 and 0.019). Conclusions: This study suggests an association between greater AI-derived radiographic severity and lower reading ability in corrected metopic synostosis. Older age at surgery was independently associated with lower reading ability and visuomotor integration. Surgical correction may mitigate neurodevelopmental differences based on severity that have been observed pre-operatively.