The Combined Contributions of Newborn Stress and Parenting Stress on Toddler Language Development.

OBJECTIVE: To examine the longitudinal associations between newborn neurobehavioral stress signs, maternal parenting stress, and several indices of toddler language development. STUDY DESIGN: Participants include 202 mother-infant dyads (104 girls). We measured stress signs in neonates in the hospital at least 24 hours after birth using the Neonatal Intensive Care Unit Network Neurobehavioral Scale. At 7 months, parenting stress (competence, attachment, and role restriction) was assessed using the Parenting Stress Index. At 18 months, mothers completed the Communicative Development Inventories, which measured toddler gesturing, expressive vocabulary, and receptive vocabulary. Longitudinal path modeling was used to estimate associations between neonatal stress signs, parenting stress, and toddler language, and a model was generated for each language outcome. Child sex, birth weight, and family income were included as covariates. RESULTS: Infants who exhibited greater neurobehavioral stress signs at birth produced significantly fewer social-communicative gestures at 18 months of age. Among infants whose mothers reported low (but not high) levels of parenting stress during the first postnatal year, newborn stress signs were negatively associated with 18-month-olds' receptive vocabulary size. Neither newborn stress signs nor parenting stress were significantly related to toddler expressive vocabulary size. CONCLUSIONS: Our findings uncover a negative association between newborn stress signs and toddler gesturing. Furthermore, our results suggest that caregiver stress and neonatal stress signs interact to predict toddler receptive vocabulary. Taken together, these results demonstrate that some neonates who exhibit increased neurobehavioral stress signs may be at heightened risk for experiencing language difficulties. These children may benefit from additional support in infancy.

Internal state language factor structure and development in toddlerhood: Insights from WordBank.

Internal state language (ISL) research contains knowledge gaps, including dimensionality and predictors of growth, addressed here in a two-aim study. Parent-reported expressive language from N = 6,373 monolingual, English-speaking toddlers (Mage = 23.5mos, 46% male, 57% white) was collected using cross-sectional and longitudinal data in WordBank. Exploratory and confirmatory factor analyses suggested a best-fitting one-factor model of ISL. The single-factor model of ISL was then submitted to hierarchical linear modeling to evaluate predictors of ISL development. Age 2 ISL production was predicted by child sex, wherein females outperform males, and maternal education, wherein higher education contributes to higher ISL. Only maternal education emerged as a significant predictor of ISL growth. These results provide support to theory suggesting a unitary construct of ISL, as opposed to considering ISL as categorical, and further illustrate linear growth through the second postnatal year that varies as a function of child sex and maternal education.

True Incidence of Marginal Mandibular Nerve Palsy Following Neonatal Mandibular Distraction Osteogenesis.

INTRODUCTION: In children with PRS, MDO is routinely performed to alleviate airway obstruction; however, it involves risk of injury to the MMN. We hypothesize that MMN palsy incidence following MDO, reported at 1-15%, is underestimated. This study investigates the true incidence of MMN palsy after MDO to better guide follow-up care and improve treatment of this complication. METHODS: A retrospective review of PRS patients who underwent MDO at a single, tertiary pediatric hospital between September 2007 and March 2021 was conducted. Patients who underwent MDO under one year of age and had postoperative clinical evaluations detailing MMN function were included. Logistic regression analysis was performed to investigate predictors of MMN injury. RESULTS: Of 93 patients who underwent MDO, 59.1% met inclusion criteria. 56.4% were female, 43.6% were syndromic, and average age at MDO was 1.52 ± 2.04 months. The average length of mandibular distraction was 17.3 ± 4.36mm, average duration of intubation was 6.57 ± 2.37 days, and average time until hardware removal was 111.1 ± 23.6 days. Sixteen patients (29.1%) presented with permanent MMN dysfunction, comprised of 8 patients with bilateral weakness and 8 with unilateral weakness. An additional five patients (9.1%) presented with transient MMN weakness that resolved within a year. Average length of follow-up postoperatively was 6.02 years, and no significant predictors of nerve injury were found. CONCLUSION: In this 14-year review of patients with PRS who underwent MDO, 38.2% demonstrated evidence of MMN palsy (29.1% permanent, 9.1% transient), which is much greater than previously described.

Vocabulary size predicts behavioral problems in emotionally reactive children.

Internalizing and externalizing behavioral problems place children at an increased risk for low academic achievement and socioemotional maladjustment. Children's language skills and level of emotional reactivity have been shown to predict behavioral problems later in development. Yet, there is an absence of research investigating vocabulary by negative emotionality interactions with respect to the development of behavioral problems during early childhood. Our study sought to fill this gap by examining whether the relation between preschool (age 3) vocabulary size and internalizing/externalizing problems during the early school years (age 6) is moderated by preschool negative emotional reactivity, even after controlling for preschool behavioral problems as well as children's socioeconomic background. Longitudinal data was collected from 256 typically developing children (129 girls, 75% White) and their mothers (64% held a college degree). Linear regression analyses revealed moderate rank-order stability in children's internalizing and externalizing problems across this period of development. Evidence of an interaction effect emerged in both the internalizing and externalizing problems regression models. That is, age 3 vocabulary was negatively related to age 6 behavioral problems, but only among children exhibiting higher levels of negative emotional reactivity. Our results indicate that early vocabulary acquisition may serve as a buffer against adverse behavioral outcomes in children with a natural propensity toward expressing negative emotions. These findings point to a more nuanced picture of the relations between language, emotional reactivity, and behavioral problems in childhood, which are discussed in greater detail to inform future intervention and educational research.

A Road Map to Creating a High-Quality Clinical Database in Plastic Surgery.

BACKGROUND: Detailed in-house databases are a staple of surgical research and a crucial source of data for many studies from which clinical guidelines are built. Despite the importance of generating a clear and thorough developmental design, the literature on database creation and management is limited. In this article, the authors present their stepwise single-institution process of developing a clinical facial fracture database. METHODS: The authors outline the process of development of a large single-institution clinical pediatric facial fracture database. The authors highlight critical steps from conception, regulatory approval, data safety/integrity, human resource allocation, data collection, quality assurance, and error remediation. The authors recorded patient characteristics, comorbidities, details of the sustained fracture, associated injuries, hospitalization information, treatments, outcomes, and follow-up information on Research Electronic Data Capture. Protocols were created to ensure data quality assurance and control. Error identification analysis was subsequently performed on the database to evaluate the completeness and accuracy of the data. RESULTS: A total of 4451 records from 3334 patients between 2006 and 2021 were identified and evaluated to generate a clinical database. Overall, there were 259 incorrect entries of 120,177 total entries, yielding a 99.8% completion rate and a 0.216% error rate. CONCLUSIONS: The quality of clinical research is intrinsically linked to the quality and accuracy of the data collection. Close attention must be paid to quality control at every stage of a database setup. More studies outlining the process of database design are needed to promote transparent, accurate, and replicable research practices.

CranioRate: An Image-Based, Deep-Phenotyping Analysis Toolset and Online Clinician Interface for Metopic Craniosynostosis.

BACKGROUND: The diagnosis and management of metopic craniosynostosis involve subjective decision-making at the point of care. The purpose of this work was to describe a quantitative severity metric and point-of-care user interface to aid clinicians in the management of metopic craniosynostosis and to provide a platform for future research through deep phenotyping. METHODS: Two machine-learning algorithms were developed that quantify the severity of craniosynostosis-a supervised model specific to metopic craniosynostosis [Metopic Severity Score (MSS)] and an unsupervised model used for cranial morphology in general [Cranial Morphology Deviation (CMD)]. Computed tomographic (CT) images from multiple institutions were compiled to establish the spectrum of severity, and a point-of-care tool was developed and validated. RESULTS: Over the study period (2019 to 2021), 254 patients with metopic craniosynostosis and 92 control patients who underwent CT scanning between the ages of 6 and 18 months were included. CT scans were processed using an unsupervised machine-learning based dysmorphology quantification tool, CranioRate. The average MSS was 0.0 ± 1.0 for normal controls and 4.9 ± 2.3 ( P < 0.001) for those with metopic synostosis. The average CMD was 85.2 ± 19.2 for normal controls and 189.9 ± 43.4 ( P < 0.001) for those with metopic synostosis. A point-of-care user interface (craniorate.org) has processed 46 CT images from 10 institutions. CONCLUSIONS: The resulting quantification of severity using MSS and CMD has shown an improved capacity, relative to conventional measures, to automatically classify normal controls versus patients with metopic synostosis. The authors have mathematically described, in an objective and quantifiable manner, the distribution of phenotypes in metopic craniosynostosis.

Perceptual Barriers to Becoming a Plastic Surgeon among Underrepresented Medical Students.

Background: The field of plastic surgery has experienced difficulty increasing diversity among trainees, despite significant efforts. Barriers to recruitment of underrepresented in medicine (URM) students are poorly understood. This study assesses URM students' exposure to plastic surgery, access to mentors and research opportunities, and the importance of diversity in the field. Methods: A survey was designed and distributed to members of the Student National Medical Association over 3 months. Survey data were collected using Qualtrics and descriptive statistics, and logistical regressions were performed using SAS. Results: Of the 136 respondents, 75.0% identified as Black (n = 102/136), and 57.4% (n = 66/115) reported a plastic surgery program at their home institution. Of the total respondents, 97.7% (n = 127/130) were concerned about racial representation in plastic surgery, and 44.9% (n = 53/114) would be more likely to apply if there were better URM representation. Most respondents disagreed that there was local (73.4%, n = 58/79) or national (79.2%, n = 57/72) interest in URM recruitment. Students whose plastic surgery programs had outreach initiatives were more likely to have attending (OR 11.7, P < 0.05) or resident mentors (OR 3.0 P < 0.05) and access to research opportunities (OR 4.3, P < 0.05). Conclusions: URM students feel there is an evident lack of interest in recruiting URM applicants in plastic surgery. Programs with outreach initiatives are more likely to provide URM students access to mentorship and research opportunities, allowing students to make informed decisions about pursuing plastic surgery.

Pediatric Nasoorbitoethmoid Fractures: A Single Institution's 15-Year Experience.

BACKGROUND: Nasoorbitoethmoid (NOE) fractures impact growth of the craniofacial skeleton in children, which may necessitate differentiated management from adult injuries. This study describes characteristics, management, and outcomes of NOE fractures in children seen at a single institution. METHODS: A retrospective review of patients under 18 years who presented to our institution from 2006 to 2021 with facial fractures was conducted; patients with NOE fractures were included. Data collected included demographics, mechanism of injury, fracture type, management, and outcomes. RESULTS: Fifty-eight patients met inclusion criteria; 77.6% presented with Manson-Marcowitz Type I fractures, 17.2% with Type II, and 5.2% with Type III. The most common cause of injury was motor vehicle accidents (MVAs, 39.7%) and sports (31%). Glasgow Coma Scale and injury mechanism were not predictive of injury severity in the pediatric population ( P =0.353, P =0.493). Orbital fractures were the most common associated fractures (n=55, 94.8%); parietal bone fractures were more likely in Type III fractures ( P =0.047). LeFort III fractures were more likely in type II fractures ( P =0.011). Soft tissue and neurological injuries were the most common associated injuries regardless of NOE fracture type (81% and 58.6%, respectively). There was no significant difference in type of operative management or in the rates of adverse outcomes between types of NOE fractures. CONCLUSIONS: These findings suggest that pediatric NOE fractures, although rare, present differently from adult NOE fractures and that revisiting predictive heuristics and treatment strategies is warranted in this population.

The longitudinal contributions of child language, negative emotionality, and maternal positive affect on toddler executive functioning development.

Executive functions (EF) develop rapidly across early childhood and play a prominent role in promoting adaptive outcomes later in development. Although the existing literature suggests that the development of early EF is sensitive to the influence of intrinsic and extrinsic factors, limited research has examined the joint contributions of multiple child and contextual factors in infancy/toddlerhood. The purpose of our longitudinal study was therefore to identify early environmental, behavioral, biologically-based factors that influence children's EF outcomes in late toddlerhood. Participants included 409 mother-child dyads (209 girls) and the data was collected across children's first three postnatal years. Parent-report measures were used to assess infant negative affectivity (5-months; IBQ-R) and toddler language (age 2; MCDI), and both maternal positive affect (5-months) and toddler frustration (age 2) were coded during mother-child interaction tasks. A battery of behavioral tasks was used to measure child EF in late toddlerhood (age 3). After controlling for maternal education (a proxy for children's socio-economic environment), path analysis indicated that both infant and maternal affect at 5-months directly predicted toddlers' language skills and frustration expression at age 2. Toddler language (but not frustration) also predicted child performance on multiple EF tasks at age 3. Finally, 5-month infant and maternal affect indirectly predicted age 3 EF via age 2 language. Our results identify language as a mechanism through which children's early caregiving environment influences their EF development. Taken together, these findings illustrate the importance of applying a biopsychosocial perspective to the examination of early childhood EF development.

Characterization of the Saddle Nose Deformity Following Endoscopic Endonasal Skull Base Surgery.

Objective The endoscopic endonasal approach (EEA) is commonly employed in skull base surgery for neoplasm resection. While nasal deformity following EEA is described, this study aimed to perform a detailed qualitative and quantitative assessment of the associated saddle nose deformity (SND) in particular. Setting/Participants This is a retrospective review of 20 adult patients with SND after EEA for resection of skull base tumors over a 5-year period at the University of Pittsburgh Medical Center. Main Outcome Measures Fifteen measurements related to SND were obtained on pre- and postoperative imaging. Statistical analyses were performed to evaluate differences between pre- and postoperative anatomies. Results The most common EEA was transsellar. Reconstruction techniques included nine free mucosal grafts alone, eight vascularized nasoseptal flaps (NSFs), one combined free mucosal graft/abdominal fat graft, and one combined NSF/fascia lata graft. Imaging analysis showed a trend toward loss of mean nasal height, nasal tip projection, and nasolabial angle postoperatively. Subgroup analysis showed that patients with NSF reconstruction had a significantly decreased nasal tip projection (1.2 mm, p = 0.039) and increased alar base width (1.2 mm, p = 0.046) postoperatively. Patients without functional pituitary microadenomas demonstrated significantly increased nasofrontal angle and decreased nasal tip projection on postoperative imaging, in contrast to those with functional adenomas who had no measurable significant changes. Conclusion Clinically evident SND does not always lead to significant radiographic changes. This analysis suggests that patients who undergo surgery for indications other than functional pituitary microadenomas or who receive NSF reconstruction develop more marked SND on standard imaging tests.

From terrible twos to sassy sixes: The development of vocabulary and executive functioning across early childhood.

Across the early childhood period of development, young children exhibit considerable growth in their executive functioning (EF) and vocabulary abilities. Understanding the developmental trajectory of these seemingly interrelated processes is important as both early vocabulary and EF have been shown to predict critical academic and socio-emotional outcomes later in childhood. Although previous research suggests that EF and vocabulary are correlated in early childhood, much of the existing longitudinal research has focused on unidirectional relations among preschool child samples. The current large-scale study, therefore, sought to examine whether children's vocabulary and EF abilities are bidirectionally related over time across four measurement waves in early childhood (i.e., at ages 2, 3, 4, and 6). At each timepoint, children's vocabulary skills were positively correlated with their concurrent EF abilities. After controlling for child sex and maternal education status, the best-fitting, cross-lagged panel model was a unidirectional model whereby children's early vocabulary scores predicted their later EF performance at each timepoint. Although age 2 EF significantly predicted age 3 vocabulary size, this association was no longer significant after accounting for maternal education status. Our results illustrate that vocabulary size plays an important role in predicting children's later EF performance across various timepoints in early childhood, even after controlling for children's initial EF scores. These findings have important implications for intervention research as fostering early vocabulary acquisition may serve as a possible avenue for improving EF outcomes in young children. RESEARCH HIGHLIGHTS: Children's vocabulary size is positively correlated with their concurrent executive functioning skill at ages 2, 3, 4, and 6 Young children's early vocabulary scores predict their later EF performance across measurement waves, even after controlling for initial EF skill There is stability in children's relative vocabulary size and executive functioning performance over time in early childhood.

Setting the stage: Biopsychosocial predictors of early childhood externalizing behaviors.

Research has indicated that biological (self-regulation), psychological (temperament), and social (maternal parenting behaviors) factors predict childhood externalizing behaviors. Few studies, however, have evaluated psychological, biological, and social factors in conjunction as predictors of childhood externalizing behaviors. Further, limited research has examined whether these biopsychosocial predictors during infancy and toddlerhood predict the onset of externalizing behaviors in early childhood. The present study aimed to examine the longitudinal relations between biopsychosocial predictors of child externalizing behaviors. Children and their mothers (n = 410) participated when children were 5, 24, and 36 months old. Child self-regulation was assessed via baseline respiratory sinus arrhythmia (RSA) at age 5 months, and child psychology was measured via maternal report of effortful control at age 24 months. Additionally, maternal intrusiveness was assessed during a mother-child interaction at age 5 months. At 36 months, mothers reported on child externalizing behaviors. Longitudinal path modeling was used to examine the direct and indirect effects of maternal intrusiveness and child effortful control on child externalizing behavior, as well as whether these effects were conditional upon child baseline RSA. Results showed a significant indirect effect of maternal intrusiveness on externalizing behavior through effortful control, and this pathway was moderated by baseline RSA after controlling for orienting regulation at age 5 months. These results suggest that early childhood externalizing behaviors are jointly affected by biological, psychological, and social factors during toddlerhood.

Late-Presenting Sagittal Craniosynostosis: An Update to a Standardized Treatment Protocol.

BACKGROUND: The authors previously published their protocol to treat patients who present with sagittal craniosynostosis after the age of 1 year. The purpose of this article is to present a follow-up and update of this cohort to evaluate outcomes of their treatment protocol. METHODS: Patients with isolated sagittal craniosynostosis who presented after the age of 1 year between July of 2013 and April of 2021 were included. RESULTS: A total of 108 patients met inclusion criteria. The average age at presentation was 5.2 ± 3.4 years, and 79 patients (73.1%) were male. The indications for imaging were head shape (54.6%), headache (14.8%), trauma (9.3%), seizure (4.6%), papilledema (2.8%), and other (13.9%). Of the 108 patients, 12 (11.1%) underwent surgery following their initial consultation: five for papilledema, four for elevated intracranial pressure, two for severely scaphocephalic head shapes, and one for abnormal funduscopic findings. Two of these patients underwent additional reconstructive surgery, one for the recurrence of papilledema and headache and the other for progressive scaphocephaly. The average length of time between operations was 4.9 years. Of the 96 patients who were managed conservatively, four (4.2%) underwent surgery at an average of 1.2 ± 0.5 years later (average age, 4.4 ± 1.5 years) for brain growth restriction ( n = 2), aesthetic concerns ( n = 1), and refractory headaches ( n = 1). The average follow-up of all patients with craniofacial surgery was 2.7 ± 2.3 years (median, 2.1 years; interquartile range, 3.7 years). CONCLUSIONS: Patients with late-presenting sagittal craniosynostosis require surgical correction less often than younger patients, likely because of milder phenotype. Few patients in the conservative treatment arm ultimately required surgery (4%). CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, IV.

Quantifying the Severity of Metopic Craniosynostosis Using Unsupervised Machine Learning.

BACKGROUND: Quantifying the severity of head shape deformity and establishing a threshold for operative intervention remains challenging in patients with metopic craniosynostosis (MCS). This study combines three-dimensional skull shape analysis with an unsupervised machine-learning algorithm to generate a quantitative shape severity score (cranial morphology deviation) and provide an operative threshold score. METHODS: Head computed tomography scans from subjects with MCS and normal controls (5 to 15 months of age) were used for objective three-dimensional shape analysis using ShapeWorks software and in a survey for craniofacial surgeons to rate head-shape deformity and report whether they would offer surgical correction based on head shape alone. An unsupervised machine-learning algorithm was developed to quantify the degree of shape abnormality of MCS skulls compared to controls. RESULTS: One hundred twenty-four computed tomography scans were used to develop the model; 50 (24% MCS, 76% controls) were rated by 36 craniofacial surgeons, with an average of 20.8 ratings per skull. The interrater reliability was high (intraclass correlation coefficient, 0.988). The algorithm performed accurately and correlates closely with the surgeons assigned severity ratings (Spearman correlation coefficient, r = 0.817). The median cranial morphology deviation for affected skulls was 155.0 (interquartile range, 136.4 to 194.6; maximum, 231.3). Skulls with ratings of 150.2 or higher were very likely to be offered surgery by the experts in this study. CONCLUSIONS: This study describes a novel metric to quantify the head shape deformity associated with MCS and contextualizes the results using clinical assessments of head shapes by craniofacial experts. This metric may be useful in supporting clinical decision making around operative intervention and in describing outcomes and comparing patient population across centers.

"Validation of Artificial Intelligence Severity Assessment in Metopic Craniosynostosis".

OBJECTIVE: Several severity metrics have been developed for metopic craniosynostosis, including a recent machine learning-derived algorithm. This study assessed the diagnostic concordance between machine learning and previously published severity indices. DESIGN: Preoperative computed tomography (CT) scans of patients who underwent surgical correction of metopic craniosynostosis were quantitatively analyzed for severity. Each scan was manually measured to derive manual severity scores and also received a scaled metopic severity score (MSS) assigned by the machine learning algorithm. Regression analysis was used to correlate manually captured measurements to MSS. ROC analysis was performed for each severity metric and were compared to how accurately they distinguished cases of metopic synostosis from controls. RESULTS: In total, 194 CT scans were analyzed, 167 with metopic synostosis and 27 controls. The mean scaled MSS for the patients with metopic was 6.18 ± 2.53 compared to 0.60 ± 1.25 for controls. Multivariable regression analyses yielded an R-square of 0.66, with significant manual measurements of endocranial bifrontal angle (EBA) (P = 0.023), posterior angle of the anterior cranial fossa (p < 0.001), temporal depression angle (P = 0.042), age (P < 0.001), biparietal distance (P < 0.001), interdacryon distance (P = 0.033), and orbital width (P < 0.001). ROC analysis demonstrated a high diagnostic value of the MSS (AUC = 0.96, P < 0.001), which was comparable to other validated indices including the adjusted EBA (AUC = 0.98), EBA (AUC = 0.97), and biparietal/bitemporal ratio (AUC = 0.95). CONCLUSIONS: The machine learning algorithm offers an objective assessment of morphologic severity that provides a reliable composite impression of severity. The generated score is comparable to other severity indices in ability to distinguish cases of metopic synostosis from controls.

Mandibular Measurements at the 20-Week Anatomy Ultrasound as a Prenatal Diagnostic Predictor of Pierre Robin Sequence.

BACKGROUND: Pierre Robin Sequence (PRS) is characterized by micrognathia, glossoptosis, and upper airway obstruction. Early recognition and appropriate perinatal management is crucial for optimizing outcomes. This study aimed to evaluate 20-week fetal ultrasounds to determine if specific mandibular measurements could predict PRS diagnosis and disease severity. METHODS: A retrospective case-control study of 48 patients with PRS and gender-matched controls was performed. Medical records were reviewed for respiratory and surgical interventions. Three parameters to assess micrognathia were measured on mid-sagittal profile ultrasound images: frontal nasal-mental angle (FNMA), facial-maxillary angle (FMA), and alveolar overjet. Student's t-test and univariate logistic regression was performed. P ≤ 0.05 was considered statistically significant. RESULTS: Patients with PRS demonstrated a significantly smaller mean FNMA compared to the control group, 129.3 ± 8.6° and 137.4 ± 3.2°, respectively (p < 0.0001), as well as significantly smaller mean FMA, 63.2 ± 9.2° and 74.8 ± 6.1°, respectively (p < 0.0001). The PRS group also demonstrated significantly larger mean alveolar overjet compared to the control group, 3.9 ± 1.4 mm and 2.1 ± 0.9 mm, respectively (p < 0.0001). The odds of respiratory intervention increased among cases when FMA was <68°. Additionally, there was a significant difference in median overjet between patients with PRS who did and did not require respiratory intervention. CONCLUSIONS: Mandibular features on the 20-week ultrasound can be measured to predict diagnosis and severity of PRS. This is an important first step to prepare for potential respiratory intervention at delivery to minimize perinatal hypoxia. Alveolar overjet, previously not described in prenatal ultrasound literature, is measurable and has utility in prenatal screening for PRS, as do FMA and FNMA.

3D Photography to Quantify the Severity of Metopic Craniosynostosis.

This study aims to determine the utility of 3D photography for evaluating the severity of metopic craniosynostosis (MCS) using a validated, supervised machine learning (ML) algorithm.This single-center retrospective cohort study included patients who were evaluated at our tertiary care center for MCS from 2016 to 2020 and underwent both head CT and 3D photography within a 2-month period.The analysis method builds on our previously established ML algorithm for evaluating MCS severity using skull shape from CT scans. In this study, we regress the model to analyze 3D photographs and correlate the severity scores from both imaging modalities.14 patients met inclusion criteria, 64.3% male (n = 9). The mean age in years at 3D photography and CT imaging was 0.97 and 0.94, respectively. Ten patient images were obtained preoperatively, and 4 patients did not require surgery. The severity prediction of the ML algorithm correlates closely when comparing the 3D photographs to CT bone data (Spearman correlation coefficient [SCC] r = 0.75; Pearson correlation coefficient [PCC] r = 0.82).The results of this study show that 3D photography is a valid alternative to CT for evaluation of head shape in MCS. Its use will provide an objective, quantifiable means of assessing outcomes in a rigorous manner while decreasing radiation exposure in this patient population.

The Clinical Significance of Clinocephaly in Late-Presentation Sagittal Craniosynostosis.

The diagnosis of late-presentation sagittal suture craniosynostosis (SCS) can be challenging, especially in the setting of subtle physical exam findings. The clinical significance of clinocephaly-a retro-coronal concavity along the midvault-in this context remains unknown. The aim of this study is to evaluate the predictive value of clinocephaly in identifying late-presentation SCS.A retrospective chart review of all patients >1 year old presenting to the craniofacial clinic with a concern for SCS was performed. The presence or absence of SCS in the setting of clinocephaly was recorded following diagnostic imaging. Student's t test, Chi Square test, and multivariate logistic regression analysis were performed to determine predictors for SCS.75 patients met inclusion criteria. 32 patients (42.7%, 6% female) were diagnosed with SCS. No difference in age between patients with and without SCS was detected. Stratification of patients by age (1-2, 2-4, and >4 years) revealed a higher rate of SCS in younger patients (P = 0.04). The cephalic index (C.I.) of those with sagittal synostosis was significantly smaller but within the normal range, indicating a more scaphocephalic shape (P = 0.003). Logistic regression analysis revealed that C.I. was a strong predictor for SCS (P = 0.003). Of those with SCS, a mix of complete and partial fusion of the sagittal suture was appreciated.This study found that 42.7% of patients with clinocephaly had SCS. C.I. was the only predictor for SCS and unique suture fusion patterns were identified in those with SCS. This study suggests that clinocephaly should be considered a core component of the exam and work-up for SCS. Future studies aimed at evaluating the positive predictive value of this exam finding and identifying risk factors associated with late-presentation SCS are underway.

Squamosal Suture Synostosis: An Under-Recognized Phenomenon.

INTRODUCTION: The squamosal suture (SQS) joins the temporal to the parietal bones bilaterally and is a poorly described site of craniosynostosis. SQS fusion is thought to occur as late as the fourth decade of life and beyond; however, we have incidentally noted its presence among our pediatric patients and hypothesize that it may occur earlier in life and more frequently than previously believed. METHODS: A retrospective review of imaging performed on pediatric patients was completed to identify patients with SQS synostosis. This included a review of clinical notes as well as computed tomography (CT) images obtained by our craniofacial clinic. Relevant patient data and imaging were reviewed. RESULTS: Forty-seven patients were identified with SQS synostosis, 21 were female (45%). Age at the time of radiographic diagnosis was 10.1 ± 8.4 years (range 17 days to 27 years). A majority of patients had bilateral SQS synostosis (57%), with a relatively even distribution of unilateral right (23%) versus left (19%). SQS was an isolated finding (no other suture involvement) in 15 patients (32%), all of whom were normocephalic and did not require surgical intervention. Thirty-two patients (68%) had concomitant craniosynostosis of other sutures, most commonly sagittal and coronal. Nine patients (19%) underwent surgery to correct cranial malformations-all these patients had multi-suture synostosis (P = 0.012). Twenty-seven patients (57%) had SQS synostosis diagnosed incidentally compared to 20 (43%) who were imaged with suspicion for synostosis. In those who were symptomatic, common findings included developmental delay, elevated intracranial pressure, hydrocephalus, seizures, and visual/hearing impairments. Ten patients (21%) were syndromic, the most frequent of which was Crouzon syndrome. No single pattern of calvarial malformation could be definitively described for SQS synostosis. CONCLUSION: Given that most isolated SQS synostosis cases were normocephalic, asymptomatic, and discovered incidentally, it is likely that there are many cases of unidentified SQS synostosis. The significance of SQS synostosis is currently unclear, and warrants further investigation into this phenomenon, its natural course, and its potential presence in the spectrum of normal development.

A Three-Dimensional-Based Morphometric Analysis of a Standardized Overcorrection Technique for Fronto-Orbital Advancement in Metopic Craniosynostosis.

INTRODUCTION: The concept of "overcorrection" for trigonocephaly has been reported to achieve both anterior cranial fossa expansion and normalization of craniofacial form. The purpose of this study is to describe in detail a standardized technique to fronto-orbital advancement utilizing the concept of "overcorrection" and objectively evaluate intermediate results. METHODS: This retrospective study included patients with isolated metopic synostosis who underwent surgery via the proposed surgical technique and age and sex-matched unaffected controls. Craniofacial morphometric analysis was performed on pre-, immediate post-, and intermediate postoperative (>2 years) three-dimensional (3D)-rendered computed tomographic (CT) scans and photographs. Key CT-based measurements included interzygomaticofrontal suture distance (IZFS), endocranial bifrontal angle (ECA), and temporal expansion. 3D photogrammetry was performed using established measurements and associated Z-scores converted. A Paired t-test and analysis of variance were performed when appropriate. RESULTS: Forty-one patients were included. A comparison of pre- and immediate postoperative CT scans demonstrated statistically significant increases in all measurements. Subset analysis of 12 patients with intermediate follow-up (age: 39.6 ± 3.6 months) demonstrated significant differences from preoperative values except for IZFS, which decreased from immediate postoperative values and was smaller than age- and sex-matched controls. 3D photogrammetry demonstrated a mean Z-score above the norm for frontal breath. 3D photogrammetry is also positively correlated with CT-based measurements. CONCLUSIONS: This standardized "overcorrection" approach for trigonocephaly can provide the appropriate changes to maintain a normal ECA despite a reduction in bifrontal width over time. 3D photogrammetry positively correlated with CT-based measurements and may provide useful information when following patients clinically. Long-term follow-up assessment to determine the necessary degree of overcorrection at skeletal mature is needed.