Greater Gestational Vitamin D Status is Associated with Reduced Childhood Behavioral Problems in the Environmental Influences on Child Health Outcomes Program.

BACKGROUND: Vitamin D deficiency is common in pregnancy. Vitamin D plays an important role in the developing brain, and deficiency may impair childhood behavioral development. OBJECTIVES: This study examined the relationship between gestational 25(OH)D concentrations and childhood behavior in the Environmental influences on Child Health Outcomes (ECHO) Program. METHODS: Mother-child dyads from ECHO cohorts with data available on prenatal (first trimester through delivery) or cord blood 25(OH)D and childhood behavioral outcomes were included. Behavior was assessed using the Strengths and Difficulties Questionnaire or the Child Behavior Checklist, and data were harmonized using a crosswalk conversion. Linear mixed-effects models examined associations of 25(OH)D with total, internalizing, and externalizing problem scores while adjusting for important confounders, including age, sex, and socioeconomic and lifestyle factors. The effect modification by maternal race was also assessed. RESULTS: Early (1.5-5 y) and middle childhood (6-13 y) outcomes were examined in 1688 and 1480 dyads, respectively. Approximately 45% were vitamin D deficient [25(OH)D < 20 ng/mL], with Black women overrepresented in this group. In fully adjusted models, 25(OH)D concentrations in prenatal or cord blood were negatively associated with externalizing behavior T-scores in middle childhood [-0.73 (95% CI: -1.36, -0.10) per 10 ng/mL increase in gestational 25(OH)D]. We found no evidence of effect modification by race. In a sensitivity analysis restricted to those with 25(OH)D assessed in prenatal maternal samples, 25(OH)D was negatively associated with externalizing and total behavioral problems in early childhood. CONCLUSIONS: This study confirmed a high prevalence of vitamin D deficiency in pregnancy, particularly among Black women, and revealed evidence of an association between lower gestational 25(OH)D and childhood behavioral problems. Associations were more apparent in analyses restricted to prenatal rather than cord blood samples. Interventions to correct vitamin D deficiency during pregnancy should be explored as a strategy to improve childhood behavioral outcomes.

Behavioral and Social Functioning of Children With and Without Positional Plagiocephaly: Late Infancy to School Age.

Positional plagiocephaly and/or brachycephaly (PPB) is associated with cognition, motor, and other developmental outcomes, but little is known about the social-behavioral adjustment of children with PPB. The primary aim of this study was to compare the social-behavioral development of preschool and school-age children with and without PPB and to examine the potential moderating effects of PPB severity on group differences.Two hundred twenty children with a history of PPB and 164 controls participated in at least one behavioral assessment at 4-11 months, 18 months, 36 months, and 7 years. The frequencies of observed problem behaviors and social competence were estimated using the Child Behavior Checklist Ages 1.5-5 (CBCL/1.5-5), Caregiver-Teacher Report Form(C-TRF), CBCL/6-18, and Teacher Report Form.Children with PPB were similar to controls on the internalizing, externalizing, or total problems composites. At 7 years, CBCL/6-18 total competence scores were significantly lower in children with histories of PPB than controls. In analyses stratified by PPB severity, we found that children with moderate/severe PPB had slightly higher scores on the C-TRF internalizing scale at 36 months and lower total competence scores at age 7 years. Children who had a history of mild PPB were similar to controls on all outcomes.This study is the first to examine social and behavioral outcomes in a large cohort of children with and without a history of PPB. We found limited evidence of an association between PPB and parent and teacher-reported social-emotional and behavioral adjustment through early school-age.

Home Language Environment of Children With Orofacial Clefts as a Target for Intervention.

OBJECTIVE: To evaluate the home language environment (HLE) in children with orofacial clefts as a potential modifiable target for language and literacy intervention. DESIGN: Feasibility study examining longitudinal trends in HLE and responses to parent-focused literacy intervention. SETTING: Tertiary care children's hospital. PARTICIPANTS: HLE data were collected for 38 children with orofacial clefts between ages 7 and 23 months. Twenty-seven participants received parent-focused literacy intervention. INTERVENTIONS: Reach Out and Read, a literacy intervention, was introduced during a clinic visit. To assess response, participants were randomized to age at intervention (9, 18, or 24 months). MAIN OUTCOME MEASURES: Primary outcome measures included measurements from recordings in the home language environment of adult word count, child vocalizations, and conversational turns. RESULTS: Baseline (preintervention) results showed lower adult word count and conversational turns for caregivers and children with cleft lip and palate, as well as for those from lower socioeconomic groups. After the literacy intervention was introduced, this cohort showed increasing measures of child and caregiver vocalizations, particularly when introduced at 18 months. CONCLUSIONS: Although these results are preliminary, findings suggest that HLE characteristics vary as a function of children's cleft type as well as family socioeconomic status. Further, our caregiver-focused literacy intervention was feasible and resulted in short-term improvements in HLE. This is the first study to document HLE as a target for intervention in children with oral clefts. These findings support further research on HLE and caregiver-focused intervention to improve language/literacy outcomes for children with oral clefts.

Cognitive, Motor, and Language Development of Preschool Children With Craniofacial Microsomia.

OBJECTIVE: To examine neurodevelopment in preschool-aged children with craniofacial microsomia (CFM) relative to unaffected peers. DESIGN: Multisite, longitudinal cohort study. SETTING: Tertiary care centers in the United States. PARTICIPANTS: We included 92 children with CFM ("cases") through craniofacial centers and clinics. Seventy-six children without CFM (controls) were included from pediatric practices and community advertisements. This study reports on outcomes assessed when participants were an average age of 38.4 months (SD = 1.9). MAIN OUTCOME MEASURES: We assessed cognitive and motor skills using the Bayley Scales of Infant and Toddler Development, third edition (Bayley-III), and language function using subtests from the Clinical Evaluation of Language Fundamentals-Preschool, second edition (CELF-P2). RESULTS: Case-control differences were negligible for Bayley-III cognitive (effect sizes [ES] = -0.06, P = .72) and motor outcomes (ES = -0.19, P = .25). Cases scored lower than controls on most scales of the CELF-P2 (ES = -0.58 to -0.20, P = .01 to .26). Frequency counts for "developmental delay" (ie, one or more scores > 1 SD below the normative mean) were higher for cases (39%) than controls (15%); however, the adjusted odds ratio = 1.73 (P = 0.21) was not significant. Case-control differences were most evident in children with microtia or other combinations of CFM-related facial features. CONCLUSIONS: Cognitive and motor scores were similar for preschool-aged children with and without CFM. However, children with CFM scored lower than controls on language measures. We recommend early monitoring of language to identify preschoolers with CFM who could benefit from intervention.

Behavioral Adjustment of Preschool Children With and Without Craniofacial Microsomia.

OBJECTIVE: The study aim was to assess behavioral adjustment in preschool children with and without craniofacial microsomia (CFM). DESIGN: Multisite cohort study of preschoolers with CFM ("cases") or without CFM ("controls"). PARTICIPANTS: Mothers (89%), fathers (9%), and other caregivers (2%) of 161 preschoolers. OUTCOME MEASURE: Child Behavior Check List (CBCL 1.5-5); linear regressions with standardized effect sizes (ES) adjusted for sociodemographic confounds. RESULTS: Child Behavior Check Lists for 89 cases and 72 controls (average age 38.3 ± 1.9 months). Children were male (54%), white (69%), and of Latino ethnicity (47%). Cases had microtia with mandibular hypoplasia (52%), microtia only (30%), or other CFM-associated features (18%). Nearly 20% of cases had extracranial anomalies. Composite CBCL scores were in the average range compared to test norms and similar for cases and controls. On the subscales, cases' parents reported higher Anxious/Depressed scores (ES = 0.35, P = .04), Stress Problems (ES = 0.40, P = .04), Anxiety Problems (ES = 0.34, P = .04), and Autism Spectrum Problems (ES = 0.41, P = .02); however, the autism subscale primarily reflected speech concerns. Among cases, more problems were reported for children with extracranial anomalies and certain phenotypic categories with small ES. CONCLUSIONS: Behavioral adjustment of preschoolers with CFM was comparable to peers. However, parental reports reflected greater concern for internalizing behaviors; thus, anxiety screening and interventions may benefit children with CFM. Among cases, more problems were reported for those with more complex presentations of CFM. Craniofacial microsomia-related speech problems should be distinguished from associated psychosocial symptoms during developmental evaluations.

Caregiver Perspectives on School Participation Among Students With Craniofacial Microsomia.

IMPORTANCE: Knowledge of unmet school participation needs for students with craniofacial microsomia (CFM) can inform decisions regarding intervention support. OBJECTIVE: To compare students with and without CFM on school participation (i.e., frequency, involvement, desire for participation to change) and caregivers' perceptions of environmental support for participation in occupations. DESIGN: Cross-sectional design using secondary analyses of a subset of data. SETTING: Multisite cohort study. PARTICIPANTS: Caregivers of students with CFM (n = 120) and of students without CFM (n = 315), stratified by history of education- and health-related service use. OUTCOMES AND MEASURES: School participation and environmental support, obtained with the Participation and Environment Measure-Children and Youth. RESULTS: Significant group differences were found in frequency of school participation (effect size [ES] = -0.38, 95% confidence interval [-0.64, -0.12], p = .005), level of involvement (ES = -0.14, p = .029), and desired change (p = .001), with students with CFM exhibiting greater participation restriction than students without CFM and no history of service use. No statistically significant group differences were found in environmental support for participation in the school setting. Item-level findings showed statistically significant higher desire for participation to change in three of five school occupations (odds ratio = 1.77-2.39, p = .003-.045) for students with CFM compared with students without CFM and no history of service use. CONCLUSIONS AND RELEVANCE: The results suggest that students with CFM experience restriction in participation at school. WHAT THIS ARTICLE ADDS: Students with CFM may benefit from targeted school-based interventions to optimize their inclusion.

Prediction of school-age IQ, academic achievement, and motor skills in children with positional plagiocephaly.

INTRODUCTION: Children with positional plagiocephaly and/or brachycephaly (PPB) are at risk of early developmental delay, but little is known about early life factors associated with school-age neurodevelopment. This study examined associations of demographic characteristics, prenatal risk factors and early neurodevelopment assessment with school-age IQ, academic performance, and motor development in children with PPB. METHODS: The study sample consisted of 235 school-age children with PPB followed since infancy. Outcome measures included IQ using the Differential Ability Scales-Second Edition, academic achievement as measured by the Wechsler Individualized Achievement Tests-Third Edition), and motor function using the Bruininks-Oseretsky Test of Motor Proficiency, Second Edition. Linear regression was used to examine the incremental improvement of model fit of demographics, prenatal and early life characteristics, severity of PPB, and neurodevelopment at ages 7, 18, and 36 months as measured by the Bayley-3 on school-age scores. RESULTS: Mean age at school-age assessment was 9.0 years. Adjusted r2 for demographic, prenatal, and early life risk factors ranged from 0.10 to 0.22. Addition of PPB severity and Bayley-3 measures at ages 7 and 18 months did not meaningfully change model fit. Adjusted r2 after inclusion of Bayley-3 at 36 months ranged from 0.35 to 0.41. CONCLUSION: This study suggests that PPB severity and very early life neurodevelopment have little association with school-age neurodevelopment above and beyond demographic and early life risk factors. However, preschool-age neurodevelopmental assessment may still be useful in identifying children with PPB at risk for delay and who may benefit from early intervention.

Maternal acetaminophen use during pregnancy and childhood behavioural problems: Discrepancies between mother- and teacher-reported outcomes.

BACKGROUND: Maternal acetaminophen use during pregnancy is common and has been associated with childhood behavioural problems among offspring, specifically hyperactivity and conduct problems. OBJECTIVE: Assessments of child behaviour in many previous studies have relied on maternal or parent report. Acknowledging that results of behavioural assessments vary between informants, we examined the association between maternal acetaminophen use during pregnancy and behaviour problems in childhood based on mother- and teacher-report. METHODS: A longitudinal study of 560 mother-child pairs with data on illnesses and medication use during pregnancy and neurodevelopmental assessments during childhood was conducted. Acetaminophen use during pregnancy was captured using a standardised maternal interview, completed 1 year after delivery on average. Measures of childhood (6-12 years of age) behaviour were obtained via mother- and teacher-report, using the Child Behaviour Checklist and Teacher Report Form. Linear and log-binomial models were used to calculate adjusted mean differences (MD) and risk ratios (RR), respectively and 95% confidence intervals (CI) for internalising, externalising, and total behaviour problems comparing acetaminophen users to non-users. Stabilized inverse probability weights were used to account for loss to follow-up, and adjustments for indication were made. RESULTS: Approximately 60% (n = 354) of women reported use of acetaminophen during pregnancy. Acetaminophen use during pregnancy was associated with an increase in total behaviour problem score and risk of clinical behaviour problems according to mother report (MD 2.2, 95% CI 0.3, 4.1 and RR 1.93, 95% CI 0.99, 3.76) but not according to teacher report. Weighting to account for participation did not alter results, while adjustment for indications of acetaminophen use greatly attenuated the associations with mother-reported total behaviour problem score and risk of clinical behaviour problems (MD 0.1, 95% CI -2.1, 2.3 and RR 1.31, 95% CI 0.67, 2.58). CONCLUSIONS: Acetaminophen use during pregnancy was weakly associated with mother-reported behaviour problems and not associated with teacher-reported problems.

Motor Function in School-Aged Children With Positional Plagiocephaly or Brachycephaly.

OBJECTIVE: To determine whether children with a history of positional plagiocephaly/brachycephaly (PPB) show persistent deficits in motor development. METHODS: In a longitudinal cohort study, we completed follow-up assessments with 187 school-aged children with PPB and 149 participants without PPB who were originally enrolled in infancy. Primary outcomes were the Bruininks-Oseretsky Test of Motor Proficiency-Second Edition (BOT-2) composite scores. RESULTS: Children with PPB scored lower than controls on the BOT-2. Stratified analyses indicated that differences were restricted to children who had moderate-severe PPB. No consistent differences were observed in children who had mild PPB. CONCLUSION: Children who had moderate-severe PPB in infancy show persistent differences in motor function. We suggest close developmental monitoring and early intervention to address motor deficits.

Infant/toddler motor skills as predictors of cognition and language in children with and without positional skull deformation.

PURPOSE: To estimate associations between early motor abilities (at two age points, 7 and 18 months on average) and cognitive/language outcomes at age 3. To determine whether these associations are similar for children with and without positional plagiocephaly and/or brachycephaly (PPB). METHODS: The Bayley Scales of Infant/Toddler Development 3 were given at all age points to 235 children with PPB and 167 without PPB. Linear regressions assessed longitudinal associations between fine and gross motor scales and cognition/language. Item analyses examined the contributions of specific motor skills. RESULTS: Associations between 7-month motor skills and cognition/language were modest overall (effect sizes [ES] = - 0.08 to 0.10, p = .13 to .95). At 18 months, both fine and gross motor skills were associated with outcomes for children with PPB (ES = 0.21 to 0.41, p < .001 to .01), but among those without PPB, only fine motor skills were associated with outcomes (ES = 0.21 to 0.27, p < .001 to .001). CONCLUSIONS: Toddlers' motor skills were associated with cognition and language at 3 years, particularly among children with PPB. Interventions targeting early motor development in infants and toddlers with PPB may have downstream benefits for other outcomes.

Methods and Challenges in a Cohort Study of Infants and Toddlers With Craniofacial Microsomia: The Clock Study.

OBJECTIVE: The Craniofacial microsomia: Longitudinal Outcomes in Children pre-Kindergarten (CLOCK) study is a longitudinal cohort study of neurobehavioral outcomes in infants and toddlers with craniofacial microsomia (CFM). In this article, we review the data collection and methods used to characterize this complex condition and describe the demographic and clinical characteristics of the cohort. SETTING: Craniofacial and otolaryngology clinics at 5 study sites. PARTICIPANTS: Infants with CFM and unaffected infants (controls) ages 12 to 24 months were recruited from the same geographical regions and followed to age 36 to 48 months. METHODS: Phenotypic, neurodevelopmental, and facial expression assessments were completed during the first and third waves of data collection (time 1 and time 3, respectively). Medical history data were taken at both of these time points and during an intermediate parent phone interview (time 2). RESULTS: Our cohort includes 108 cases and 84 controls. Most cases and controls identified as white and 55% of cases and 37% of controls identified as Hispanic. Nearly all cases had microtia (95%) and 59% had mandibular hypoplasia. Cases received extensive clinical care in infancy, with 59% receiving care in a craniofacial clinic and 28% experiencing at least one surgery. Study visits were completed at a study site (92%) or at the participant's home (8%). CONCLUSIONS: The CLOCK study represents an effort to overcome the challenges of characterizing the phenotypic and neurodevelopmental outcomes of CFM in a large, demographically and geographically diverse cohort.

Neurodevelopment of Infants with and without Craniofacial Microsomia.

OBJECTIVES: To determine whether infant cases with craniofacial microsomia (CFM) evidence poorer neurodevelopmental status than demographically similar infants without craniofacial diagnoses ("controls"), and to examine cases' neurodevelopmental outcomes by facial phenotype and hearing status. STUDY DESIGN: Multicenter, observational study of 108 cases and 84 controls aged 12-24 months. Participants were assessed by the Bayley Scales of Infant and Toddler Development-Third Edition and the Preschool Language Scales-Fifth Edition (PLS-5). Facial features were classified with the Phenotypic Assessment Tool for Craniofacial Microsomia. RESULTS: After adjustment for demographic variables, there was little difference in Bayley Scales of Infant and Toddler Development-Third Edition or Preschool Language Scales-Fifth Edition outcomes between cases and controls. Estimates of mean differences ranged from -0.23 to 1.79 corresponding to standardized effect sizes of -.02 to 0.12 (P values from .30 to .88). Outcomes were better among females and those with higher socioeconomic status. Among cases, facial phenotype and hearing status showed little to no association with outcomes. Analysis of individual test scores indicated that 21% of cases and 16% of controls were developmentally delayed (OR 0.68, 95% CI 0.29-1.61). CONCLUSIONS: Although learning problems have been observed in older children with CFM, we found no evidence of developmental or language delay among infants. Variation in outcomes across prior studies may reflect differences in ascertainment methods and CFM diagnostic criteria.

Associations between laterality of orofacial clefts and medical and academic outcomes.

Patients with oral clefts have an increased risk of other malformations, syndromes, and lower academic performance in school. Few studies have investigated if laterality of clefts is associated with medical and academic outcomes. Oral clefts have nonrandom laterality, with left-sided clefts occurring approximately twice as often as right-sided clefts. Using a retrospective study design, we examined potential associations of cleft attributes and outcomes in patients with cleft lip with or without cleft palate (CL/P) born in 2003-2010 who were treated at the Seattle Children's Craniofacial Center. The following variables were extracted from medical records: cleft type, medical history, maternal hyperglycemia, other malformations, and the need for academic support at school. We used logistic regression to examine risk of associations with outcomes of interest. Relative to patients with left-sided clefts, patients with bilateral CL/P were more likely to have a syndrome. Patients with nonsyndromic right-sided CL/P had a higher risk (OR and 95%CI: 3.5, 1.3-9.5, and 5.5, 1.9-16.0, respectively) of having other malformations and requiring academic support at school, when compared to patients with left-sided CL/P. Understanding the etiology of oral clefts is complicated, in part because both genetic and environmental factors contribute to the risk of developing a cleft. However, the different outcomes associated with cleft laterality suggest that right-sided clefts may have a distinct etiology. Using laterality to study cleft subgroups may advance our understanding of the etiology of this common birth defect.

Behavioral-Social Adjustment of Adolescents with Craniofacial Microsomia.

OBJECTIVE: The objective was to assess differences in psychosocial adjustment between adolescents with and without craniofacial microsomia (CFM). DESIGN: This is a case-control follow-up study in adolescents with and without CFM. SETTING: Participants were originally recruited as infants from 26 cities across the United States and Canada. PARTICIPANTS: Participants included 142 adolescents with CFM (cases) and 316 peers without CFM (controls), their caregivers, and their teachers. MAIN OUTCOME MEASURES: Social and behavior measures from the Achenbach System of Empirically Based Assessments (ASEBA), the PedsQL: Core Version, and the Children's Communication Checklist-2nd edition (CCC-2) were used. Linear regression was used to estimate case-control differences and corresponding standardized effect sizes (ES) and 95% confidence intervals after adjustment for sociodemographic confounds. We also examined case-control differences by facial phenotype and hearing status. RESULTS: The magnitude and direction of case-control differences varied across assessment and respondent, but were generally modest (ES = -0.4 to 0.02, P values ranged from .003 to .85). There was little evidence for variation in case-control differences across different facial phenotypes or as a function of hearing status. CONCLUSIONS: Our results suggest that in spite of multiple risk factors, adolescents with CFM exhibit behavior problems no more frequently than their peers without CFM. Future studies of individuals with CFM should focus on resilience and social coping mechanisms, in addition to maladjustment.

Sleep Outcomes in Children With Single-Suture Craniosynostosis Compared With Unaffected Controls.

OBJECTIVE: To compare risk for sleep-disordered breathing between children with and without single-suture craniosynostosis. PARTICIPANTS: A total of 184 children with single-suture craniosynostosis and 184 controls. MAIN OUTCOME MEASURES: Parent reported sleep-disordered breathing-related symptoms. RESULTS: Current sleep problems were reported in 19% of patients with single-suture craniosynostosis and 14% of controls (adjusted odds ratio = 1.6; 95% CI, 0.9 to 2.8). Ever having sleep problems was reported in 25% and 23% of cases and controls, respectively (adjusted odds ratio = 1.2; 95% CI, 0.7 to 1.9). Overall, snoring was statistically associated with single-suture craniosynostosis (P = .01) and was more often reported as 2+ nights per week (versus never) in patients with single-suture craniosynostosis (13%) than in controls (4%) (adjusted odds ratio = 3.5; 95% CI, 1.5 to 8.2). CONCLUSIONS: Though preliminary, increased presence of snoring during sleep in children with single-suture craniosynostosis compared with controls suggests that children with isolated single-suture craniosynostosis may be at increased risk for sleep-disordered breathing. Further study using standardized assessments of sleep-disordered breathing is needed.

Characterizing facial features in individuals with craniofacial microsomia: A systematic approach for clinical research.

BACKGROUND: Craniofacial microsomia (CFM) is a congenital condition with wide phenotypic variability, including hypoplasia of the mandible and external ear. We assembled a cohort of children with facial features within the CFM spectrum and children without known craniofacial anomalies. We sought to develop a standardized approach to assess and describe the facial characteristics of the study cohort, using multiple sources of information gathered over the course of this longitudinal study and to create case subgroups with shared phenotypic features. METHODS: Participants were enrolled between 1996 and 2002. We classified the facial phenotype from photographs, ratings using a modified version of the Orbital, Ear, Mandible, Nerve, Soft tissue (OMENS) pictorial system, data from medical record abstraction, and health history questionnaires. RESULTS: The participant sample included 142 cases and 290 controls. The average age was 13.5 years (standard deviation, 1.3 years; range, 11.1-17.1 years). Sixty-one percent of cases were male, 74% were white non-Hispanic. Among cases, the most common features were microtia (66%) and mandibular hypoplasia (50%). Case subgroups with meaningful group definitions included: (1) microtia without other CFM-related features (n = 24), (2) microtia with mandibular hypoplasia (n = 46), (3) other combinations of CFM- related facial features (n = 51), and (4) atypical features (n = 21). CONCLUSION: We developed a standardized approach for integrating multiple data sources to phenotype individuals with CFM, and created subgroups based on clinically-meaningful, shared characteristics. We hope that this system can be used to explore associations between phenotype and clinical outcomes of children with CFM and to identify the etiology of CFM. Birth Defects Research (Part A) 106:915-926, 2016.© 2016 Wiley Periodicals, Inc.

Observed child and parent toothbrushing behaviors and child oral health.

BACKGROUND: Parent-led toothbrushing effectively reduces early childhood caries. Research on the strategies that parents use to promote this behavior is, however, lacking. AIM: To examine associations between parent-child toothbrushing interactions and child oral health using a newly developed measure, the Toothbrushing Observation System (TBOS). DESIGN: One hundred children ages 18-60 months and their parents were video-recorded during toothbrushing interactions. Using these recordings, six raters coded parent and child behaviors and the duration of toothbrushing. We examined the reliability of the coding system and associations between observed parent and child behaviors and three indices of oral health: caries, gingival health, and history of dental procedures requiring general anesthesia. RESULTS: Reliabilities were moderate to strong for TBOS child and parent scores. Parent TBOS scores and longer duration of parent-led toothbrushing were associated with fewer decayed, missing or filled tooth surfaces and lower incidence of gingivitis and procedures requiring general anesthesia. Associations between child TBOS scores and dental outcomes were modest, suggesting the relative importance of parent versus child behaviors at this early age. CONCLUSIONS: Parents' child behavior management skills and the duration of parent-led toothbrushing were associated with better child oral health. These findings suggest that parenting skills are an important target for future behavioral oral health interventions.

Predicting developmental delay in a longitudinal cohort of preschool children with single-suture craniosynostosis: is neurobehavioral assessment important?

AIM: The aim of this study was to determine whether neurobehavioral assessment before and after cranial vault surgery can improve prediction of developmental delay in children with single-suture craniosynostosis (SSC), after accounting for 'baseline' demographic and clinical variables (SSC diagnosis and surgery age). METHOD: Children with SSC were referred by the treating surgeon or pediatrician before surgery. Neurobehavioral assessments were performed at ages of approximately 6, 18, and 36 months. Iterative models were developed to predict delay, as determined by one or more tests of cognitive, motor, and language skills at 36 months. We selected from groups of variables entered in order of timing (before or after corrective surgery), and source of information (parent questionnaire or psychometric testing). RESULTS: Good predictive accuracy as determined by area under the receiver operating characteristic curve (AUC), was obtained with the baseline model (AUC=0.66), which incorporated age at surgery, sex, and socio-economic status. However, predictive accuracy was improved by including pre- and post-surgery neurobehavioral assessments. Models incorporating post-surgery neurobehavioral testing (AUC=0.79), pre-surgery testing (AUC=0.74), or both pre- and post-surgery testing (AUC=0.79) performed similarly. However, the specifity of all models was considered to be moderate (≤0.62). INTERPRETATION: Prediction of delay was enhanced by assessment of neurobehavioral status. Findings provide tentative support for guidelines of care that call for routine testing of children with SSC.

Sex differences in the neurodevelopment of school-age children with and without single-suture craniosynostosis.

PURPOSE: Previous studies have indicated that infants and school-age children with single-suture craniosynostosis (SSC, cases) score modestly but consistently lower than unaffected children (controls) on neurodevelopmental tests. However, sex differences in these functions rarely have been examined, and it is unknown whether potential sex differences vary by case status (cases vs. controls) or location of suture fusion. METHODS: We tested 182 cases and 183 demographically matched controls at a mean age of 7.4 years. We measured intellectual abilities with the Wechsler Scale of Intelligence for Children-Fourth Edition. We assessed reading, spelling, and math with a combination of the Wide Range Assessment Test-Fourth Edition, the Test of Word Reading Efficiency, and the Comprehensive Test of Phonological Processing. RESULTS: Among both cases and controls, males scored lower on all measures than females with standard score differences ranging from -1.2 to -7.8 for controls (p values from <0.001 to 0.55) and -2.3 to -8.5 for cases (p values from <0.001 to 0.33). For all but one measure, sex differences were slightly larger for cases than controls. Among cases, males were more likely than females to have learning problems (50 vs. 30%, respectively), with the highest level observed among males with unicoronal synostosis (86%). CONCLUSIONS: Sex differences in neurodevelopmental abilities among children with SSC are substantial, but not a unique correlate of this disorder as similar differences were observed among controls. Girls and those with sagittal synostosis have the lowest risk for academic problems. Boys with unicoronal synostosis warrant close developmental surveillance.

Longitudinal analysis of parenting stress in mothers and fathers of infants with and without single-suture craniosynostosis.

OBJECTIVE: To examine longitudinal differences in reported stress between parents of children with and without single-suture craniosynostosis and to compare the stress reports of mothers and fathers. DESIGN: Multi-site, nonrandomized prospective study. SETTING/PARTICIPANTS: Children with single-suture craniosynostosis (cases) were identified via referral of the treating surgeon or physician at the time of diagnosis, and those without single-suture craniosynostosis (controls) were recruited from pediatric practices, birthing centers, and announcements in print media. When children were aged 6, 18, and 36 months (on average), mothers and fathers of children with and without single-suture craniosynostosis completed the Parenting Stress Index. For cases, 247 mothers and 211 fathers completed the Parenting Stress Index at the first visit; corresponding numbers for controls were 254 and 220, respectively. MAIN OUTCOME MEASURES: The Parenting Stress Index Parent and Child Domains and subscales scores. RESULTS: We found few differences between parents of infants with and without single-suture craniosynostosis, regardless of parent gender. Irrespective of case status, mothers consistently reported higher stress than fathers on the Parent Domain. Within the Parent Domain, mothers reported more stress than fathers on the Role Restriction and Spousal Support subscales. CONCLUSIONS: The parents of children with single-suture craniosynostosis reported levels of stress similar to those reported by parents of same-aged, unaffected children. Mothers reported greater stress than fathers, and these differences remained remarkably stable over time. This may reflect widely held perceptions of gender differences in parenting roles.