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ABSTRACT: OBJECTIVES:Infants and toddlers with deformational plagiocephaly (DP) have been shown to score lower on developmental measures than unaffected children. To determine whether these differences persist, we examined development in 36-month-old children with and without a history of DP.METHODS:Participants included 224 children with DP and 231 children without diagnosed DP, all of who had been followed in a longitudinal study since infancy. To confirm the presence or absence of DP, pediatricians blinded to children's case status rated 3-dimensional cranial images taken when children were 7 months old on average. The Bayley Scales of Infant and Toddler Development, Third Edition (BSID-III) was administered as a measure of child development.RESULTS:Children with DP scored lower on all scales of the BSID-III than children without DP. Differences were largest in cognition, language, and parent-reported adaptive behavior (adjusted differences = -2.9 to -4.4 standard score points) and smallest in motor development (adjusted difference = -2.7). Children in the control group who did not have previously diagnosed DP but who were later rated by pediatricians to have at least mild cranial deformation also scored lower on the BSID-III than unaffected controls.CONCLUSIONS:Preschool-aged children with a history of DP continue to receive lower developmental scores than unaffected controls. These findings do not imply that DP causes developmental problems, but DP may nonetheless serve as a marker of developmental risk. We encourage clinicians to screen children with DP for developmental concerns to facilitate early identification and intervention.
PEDIATRICS 12/2012; · 4.47 Impact Factor
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ABSTRACT: The purpose of this study was to confirm initial reports of elevated behavior problems in children with single-suture craniosynostosis, using multiple informants, longitudinal analyses, and a control group. The authors hypothesized that children with single-suture craniosynostosis would have higher levels of maladjustment than comparison children, particularly at the older age and in selected areas of previously observed vulnerability: attention and social adjustment.
The Child Behavior Checklist was completed by 436 mothers (219 with single-suture craniosynostosis) and 371 fathers (177 with single-suture craniosynostosis) when children were aged approximately 19 months, and by 361 mothers (175 with single-suture craniosynostosis) and 303 fathers (142 with single-suture craniosynostosis) when children were aged approximately 37 months. A minimum of one caregiver/teacher report was available for 169 of these children (74 with single-suture craniosynostosis) using the Caregiver-Teacher Report Form.
Average Child Behavior Checklist/Caregiver-Teacher Report Form externalizing, internalizing, and total scores for all informants were consistently higher (worse) for children with single-suture craniosynostosis than for control group children, but most differences were small and statistically nonsignificant. No differences associated with suture site were found. At the oldest age point, both mothers and fathers (but not teachers) generated higher average scores for patients than for controls on scales measuring attention and social problems, with small to medium effect sizes (0.20 to 0.32).
On average, toddlers/preschoolers with single-suture craniosynostosis show behavioral development that is largely indistinguishable from same-aged peers of similar socioeconomic background. The predictive significance of small group differences in attention and social adjustment will be assessed in a follow-up of this cohort at age 7.
Plastic and reconstructive surgery 09/2012; 130(3):635-47. · 2.74 Impact Factor
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ABSTRACT: Objective: To evaluate associations between neurodevelopment and exposure to surgery and anesthetic agents in children with single-suture craniosynostosis (SSC). Background: Young children with SSC have unexplained neurodevelopmental delays. The possible contributions of factors related to cranial vault surgery - including anesthesia - have not been previously examined. Methods/materials: Two anesthesiologists reviewed the surgical records of 89 infants (70 had complete data). Primary exposures were duration of surgery and anesthesia and total duration of inhaled anesthesia (at age 6 months on average). Outcomes were the cognitive and motor scores from the Bayley Scales of Infant Development-II and language scores from the Preschool Language Scale, 3rd edition, given at age 36 months. Linear regression using robust standard error estimates was performed, adjusting for age at surgery and suture site. Results: Anesthesia duration ranged from 155 to 547 min. For every 30-min increase in anesthesia duration, the estimated average decrease in developmental test scores ranged from 1.1 to 2.9 (P ranged from <0.001 to 0.30). Similar, but weaker findings were observed with surgery duration and total duration of inhaled anesthesia. Inverse relations between exposure amounts and neurodevelopment were stronger in children with nonsagittal synostosis. Conclusions: Average neurodevelopmental scores were lower among children experiencing longer surgeries and higher exposures to inhaled anesthesia. These associations may be due to anesthesia exposure, nonspecific effects of surgery, or unmeasured variables that correlate with surgery duration. Further study of potential causal mechanisms is warranted.
Pediatric Anesthesia 04/2012; · 2.10 Impact Factor
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ABSTRACT: Infants with deformational plagiocephaly (DP) have been shown to exhibit developmental delays relative to unaffected infants. Although the mechanisms accounting for these delays are unknown, one hypothesis focuses on underlying differences in brain development. In this study, we used MRI to examine brain volume and shape in infants with and without DP.
Participants included 20 infants with DP (mean age = 7.9 months, SD = 1.2; n = 12 male) and 21 controls (mean age = 7.9 months, SD = 1.3; n = 11 male). Measures included volumes of the total brain and cerebellum; midsagittal areas of the corpus callosum and cerebellar vermis; and linear distance measures used to quantify the shape of selected brain structures. We also evaluated the association between shape measures and developmental scores on the Bayley Scales of Infant and Toddler Development-III (BSID-III).
Brain volume did not distinguish cases and controls (p = .214-.976). However, cases exhibited greater asymmetry and flattening of the posterior brain (p < .001-.002) and cerebellar vermis (p = .035), shortening of the corpus callosum (p = .012), and differences in the orientation of the corpus callosum (p = .005). Asymmetry and flattening of brain structures were associated with worse developmental outcomes on the BSID-III.
Infants with DP show differences in brain shape, consistent with the skull deformity characteristic of this condition, and shape measures were associated with infant development. Longitudinal studies, beginning in the neonatal period, are needed to clarify whether developmental effects precede or follow brain deformation.
Child s Nervous System 03/2012; 28(7):1083-90. · 1.54 Impact Factor
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ABSTRACT: Abstract Objective: To evaluate the hypothesis that children with craniosynostosis and their parents have differences in psychosocial outcomes, as compared with an unaffected control group. Design: Two studies were conducted, both which followed children born with and without craniosynostosis. Study 1 ascertained affected children from clinics and Study 2 ascertained affected children from a population-based study of birth defects. Participants: Study 1 included 22 children with single-suture craniosynostosis and 18 controls, ages 4-5 years. Study 2 included 24 children with non-syndromic craniosynostosis and 124 unaffected controls, ages 5-9 years. Main Outcome Measures: Outcome measures included the Child Behavior Checklist (CBCL), Social Competence Scale (SCS), Pediatric Quality of Life Inventory (PedsQLTM), and Parenting Stress Index (PSI). Results: We observed lower scores on measures of health-related quality of life in cases versus controls, with adjusted effect sizes ranging from -0.72 to -0.44 (p<0.05) on summary measures. Small but statistically non-significant increases in behavioral problems were observed in cases versus controls, with no apparent differences in social competence or parenting stress. Conclusions: Results provide preliminary evidence suggesting that children with non-syndromic craniosynostosis may have elevated risk for psychosocial difficulties, particularly health-related quality of life. Continued follow-up through pre-adolescence and adolescence is warranted. Key Words: Craniosynostoses; Health-Related Quality of Life; Neurobehavioral; National Birth Defects Prevention Study.
The Cleft Palate-Craniofacial Journal 02/2012; · 0.82 Impact Factor
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ABSTRACT: To evaluate the hypothesis that 3-year-old children with single-suture craniosynostosis would receive lower neurodevelopmental scores than a comparable group of children born with patent sutures.
Longitudinal comparison study.
Five tertiary care craniofacial centers.Participants: Patients with craniosynostosis (cases) and a comparison group of children without craniosynostosis(controls). Patients diagnosed with single-suture craniosynostosis from 2002 to 2006 were eligible as cases.Controls were frequency-matched to cases on age, sex, race, socioeconomic status, and study site.
Craniosynostosis.
We administered the Bayley Scales of Infant Development, Second Edition, mental and motor development indices and the Preschool Language Scales, Third Edition, receptive and expressive communication scales. Children were evaluated at baseline (before surgery in cases and at a similar age in controls)and at 18 and 36 months of age. We compared the groups' performances at 36 months by fitting adjusted linear and logistic regression models. We also estimated adjusted associations between age at surgery and neurodevelopmental scores.
Adjusted mean case deficits ranged from 3 to 6 points (P≤ .008 for all comparisons). Compared with controls, the odds of cases being delayed ranged from 1.5 to 2.0, depending on the neurodevelopmental scale (P values ranged from .03 to .09). Cases' ages at craniosynostosis repair were not strongly related to neurodevelopmental performance.
In this large, carefully controlled, multicenter study, we observed consistently lower mean neurodevelopmental scores in children with single-suture craniosynostosis compared with controls. These results provide further support for neurodevelopmental screening in young children with single-suture craniosynostosis.
Archives of pediatrics & adolescent medicine 02/2012; 166(6):536-42. · 3.73 Impact Factor
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ABSTRACT: To assess 3-dimensional (3D) changes in head shape in infancy and at age 18 months in children with and without plagiocephaly or brachycephaly.
Using a longitudinal design, we evaluated head shape using 3D surface imaging. We compared the head shapes of children with (1) diagnosed deformational plagiocephaly or brachycephaly (cases; n=233); (2) unaffected controls, with no evidence of dysmorphology (n=167); and (3) affected controls, who despite having no previous diagnosis demonstrated skull dysmorphology on 3D surface imaging (n=70).
Cases had greater skull flattening and asymmetry than unaffected controls at both time points, as did controls with skull dysmorphology. In all groups, head shapes became less flat and more symmetric over time. Among cases, symmetry improved slightly more for those who received orthotic treatment.
Although head shape improves over time for children with deformational plagiocephaly or brachycephaly, skull dysmorphology persists relative to unaffected controls. Further research is needed to clarify the extent to which these differences are detectable to clinicians and lay observers.
The Journal of pediatrics 11/2011; 160(4):673-678.e1. · 4.02 Impact Factor
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ABSTRACT: Objective : To compare relative levels of stress reported by mothers and fathers in families containing infants with and without single-suture craniosynostosis. Design : Case-control study. Participants : Mothers and fathers of 246 infants with recently diagnosed single-suture craniosynostosis and 253 frequency-matched control infants completed the Parenting Stress Index just prior to their infant's cranioplastic surgery. Family demographic information and mothers' ratings of the severity of their child's single-suture craniosynostosis were obtained. Results : Average Parent Domain scores for parents of infants with single-suture craniosynostosis differed little from those reported by parents of control infants; however, Child Domain scores among parents of infants with single-suture craniosynostosis were higher on some subscales, primarily related to unexpected infant health and appearance issues. In both groups, fathers reported higher Child Domain stress than mothers, and mothers reported higher Parent Domain stress than fathers. Case mothers reported greater stress if they perceived their child's condition as more noticeable to others. Conclusions : Prior to cases' cranioplastic surgery, parents of children with and without single-suture craniosynostosis reported similar levels of stress in relation to their parenting roles and the behavioral characteristics of their infants. Visibility of condition should be considered a risk for increased stress for mothers of infants with single-suture craniosynostosis. Stress differences between mothers and fathers were far more discernible than those associated with the presence or absence of single-suture craniosynostosis.
The Cleft Palate-Craniofacial Journal 09/2011; 48(5):509-18. · 0.82 Impact Factor
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ABSTRACT: To determine whether the heightened risk of developmental delays seen in infancy in patients with deformational plagiocephaly (DP) continues into the toddler years.
Longitudinal study comparing the development of children with and without DP, with assessments in infancy (mean age, 7 months) and at age 18 months.
Infants with DP were recruited from a large craniofacial center, and unaffected infants were recruited from a research registry.
The study included 227 children with DP and 232 children without previously diagnosed DP.
Diagnosis of DP by a craniofacial specialist.
Bayley Scales of Infant and Toddler Development, Third Edition, scores.
Toddlers with DP scored lower than did unaffected children on all the scales of the Bayley Scales of Infant and Toddler Development, Third Edition. Motor score differences were smaller and cognitive and language score differences were greater than those observed in infancy.
Toddlers with DP continue to exhibit evidence of developmental delays relative to toddlers without DP. These findings do not necessarily imply a causal relationship between DP and development because children with delays may be more likely to develop DP. Nonetheless, it seems that increased developmental surveillance is warranted in this population.
Archives of pediatrics & adolescent medicine 07/2011; 165(7):653-8. · 3.73 Impact Factor
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ABSTRACT: Abstract Objective. To test the hypothesis that children with orofacial clefts and their parents show poorer psychosocial adaptation than controls. Design. A case-control design was utilized to examine psychosocial outcomes in children with and without oral clefts and their parents. Participants. Outcomes were evaluated in 93 cases with orofacial clefts and 124 controls, ages 5-9 years, who were part of the Massachusetts Center for Birth Defects Research and Prevention registry. Main Outcome Measures. Measures included the Child Behavior Checklist (CBCL), the PedsQL 4.0, the Social Competence Scale (SCS), and the Parenting Stress Inventory (PSI). Results. Group differences were negligible for all measures (p > .05), and findings changed little with the application of inverse probability weighting to examine the effects of response bias. Stratified analyses revealed differences according to both sex and age, with worse outcomes observed in male cases and cases ages 7-9 years. Conclusions. We found minimal support for the notion that children with orofacial clefts and their parents show worse psychosocial adaptation than controls. Social-emotional deficits may become apparent in older school-age children, and the preschool and early elementary school years might be an optimal time for preventive interventions. Key words: orofacial cleft, craniofacial, psychological, psychosocial, parent.
The Cleft Palate-Craniofacial Journal 02/2011; · 0.82 Impact Factor
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ABSTRACT: To determine whether children with hemifacial microsomia (HFM) have higher risk for psychosocial problems than children without HFM.
One hundred and thirty-six children with HFM (64% male, mean age = 6.9 years) were compared to 568 matched controls (50% male, mean age = 7.0 years) on parent and teacher measures of behavior problems and social competence, and teacher rankings of peer acceptance.
Parents of cases and controls reported similar levels of behavior problems and social competence. Teachers reported higher frequencies of internalizing problems, lower social competence and less peer acceptance for cases. Relative to controls, teacher-rated outcomes were worse for female cases, those with younger mothers at the time of birth, those with eye anomalies, and those with one or more malformations in addition to the core features of HFM.
This study provides the first evidence of relatively poor psychosocial outcomes among children with HFM.
Journal of Pediatric Psychology 02/2011; 36(7):794-805. · 2.91 Impact Factor
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ABSTRACT: To determine whether preadolescent children with hemifacial microsomia (HFM) have higher risk of neurodevelopmental delays than unaffected control individuals.
Case-control follow-up study of neurodevelopment in children with and without HFM.
Case individuals were originally recruited from 26 craniofacial centers across the United States and Canada, and controls were recruited through community pediatricians.
One hundred thirty-six children with HFM (cases) and 568 unaffected children (controls). Main Exposure History of HFM.
The Peabody Picture Vocabulary Test-Third Edition, the Beery-Buktenica Developmental Test of Visual Motor Integration-Fifth Edition, and the Academic Competence scales from the Child Behavior Checklist and the Teacher Report Form.
Children with HFM scored lower than controls on all measures (effect size = -0.27 to -0.45; P < .001 to P = .008). Compared with controls, cases were 2 to 3 times as likely to score in the at-risk range. Relative to controls, outcomes were worse for male cases and those whose mothers were 25 years or younger at the time of their birth. Cases with HFM plus other malformations had poorer outcomes, as did cases with hearing, vision, or speech impairments.
This is the first study, to our knowledge, to show that children with HFM have poorer neurodevelopmental outcomes than unaffected children, but further study using more detailed assessments is indicated. Clinically, the findings suggest that early neurodevelopmental screening is warranted for all children with HFM.
Archives of pediatrics & adolescent medicine 02/2011; 165(2):134-40. · 3.73 Impact Factor
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ABSTRACT: The genetic contribution to the pathogenesis of isolated single suture craniosynostosis is poorly understood. The role of mutations in genes known to be associated with syndromic synostosis appears to be limited. We present our findings of a candidate gene resequencing approach to identify rare variants associated with the most common forms of isolated craniosynostosis. Resequencing of the coding regions, splice junction sites, and 5' and 3' untranslated regions of 27 candidate genes in 186 cases of isolated non-syndromic single suture synostosis revealed three novel and two rare sequence variants (R406H, R595H, N857S, P190S, M446V) in insulin-like growth factor I receptor (IGF1R) that are enriched relative to control samples. Mapping the resultant amino acid changes to the modeled homodimer protein structure suggests a structural basis for segregation between these and other disease-associated mutations found in IGF1R. These data suggest that IGF1R mutations may contribute to the risk and in some cases cause single suture craniosynostosis.
American Journal of Medical Genetics Part A 01/2011; 155A(1):91-7. · 2.39 Impact Factor
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ABSTRACT: Abstract Objective: Compare relative levels of stress reported by mothers and fathers in families containing infants with and without single suture craniosynostosis (SSC). Design/Participants: Mothers and fathers of 246 infants with recently-diagnosed SSC and 253 frequency-matched control infants completed the Parenting Stress Index (PSI) just prior to their infant's cranioplastic surgery. Family demographic information and mothers' ratings of the severity of their child's SSC were obtained. Results: Average Parent Domain scores for parents of infants with SSC differed little from those reported by parents of control infants; however, Child Domain scores among parent of infants with SSC were higher on some subscales, primarily related to unexpected infant health and appearance issues. In both groups, fathers reported higher Child Domain stress than mothers; mothers reported higher Parent Domain stress. Case mothers reported greater stress if they perceived their child's condition as more noticeable to others. Conclusions: Prior to cases' cranioplastic surgery, parents of children with and without SSC reported similar levels of stress in relation to their parenting roles and the behavioral characteristics of their infants. Visibility of condition should be considered a risk for increased stress for mothers of infants with SSC. Stress differences between mothers and fathers were far more discernible than those associated with the presence or absence of SSC. Key words: craniosynostosis; parenting stress; infancy; craniofacial condition.
The Cleft Palate-Craniofacial Journal 01/2011; · 0.82 Impact Factor
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Heather C Mefford,
Neil Shafer,
Francesca Antonacci,
Jesse M Tsai,
Sarah S Park,
Anne V Hing,
Mark J Rieder,
Matthew D Smyth, Matthew L Speltz,
Evan E Eichler,
Michael L Cunningham
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ABSTRACT: Little is known about genes that underlie isolated single-suture craniosynostosis. In this study, we hypothesize that rare copy number variants (CNV) in patients with isolated single-suture craniosynostosis contain genes important for cranial development. Using whole genome array comparative genomic hybridization (CGH), we evaluated DNA from 186 individuals with single-suture craniosynostosis for submicroscopic deletions and duplications. We identified a 1.1 Mb duplication encompassing RUNX2 in two affected cousins with metopic synostosis and hypodontia. Given that RUNX2 is required as a master switch for osteoblast differentiation and interacts with TWIST1, mutations in which also cause craniosynostosis, we conclude that the duplication in this family is pathogenic, albeit with reduced penetrance. In addition, we find that a total of 7.5% of individuals with single-suture synostosis in our series have at least one rare deletion or duplication that contains genes and that has not been previously reported in unaffected individuals. The genes within and disrupted by CNVs in this cohort are potential novel candidate genes for craniosynostosis.
American Journal of Medical Genetics Part A 09/2010; 152A(9):2203-10. · 2.39 Impact Factor
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ABSTRACT: To measure severity of trigonocephaly among infants with single-suture metopic craniosynostosis by using a novel shape descriptor, the trigonocephaly severity index (TSI), and to evaluate whether degree of trigonocephaly correlates with their neurodevelopmental test scores.
We conducted a multicenter cross-sectional and longitudinal study, identifying and recruiting 65 infants with metopic synostosis before their corrective surgery. We obtained computed tomography images for 49 infants and measured the presurgical TSI, a 3-dimensional outline-based cranial shape descriptor. We evaluated neurodevelopment by administering the Bayley Scales of Infant Development, Second Edition, and the Preschool Language Scale, Third Edition, before surgery and at 18 and 36 months of age. We fit linear regression models to estimate associations between test scores and TSI values adjusted for age at testing and race/ethnicity. We fit logistic regression models to estimate whether the odds of developmental delay were increased among children with more severe trigonocephaly.
We observed little adjusted association between neurodevelopmental test scores and TSI values, and no associations that persisted at 3 years. Trigonocephaly was less severe among children referred at older ages.
We observed little evidence of an association between the severity of trigonocephaly among metopic synostosis patients and their neurodevelopmental test scores. Detecting such a relationship with precision may require larger sample sizes or alternative phenotypic quantifiers. Until studies are conducted to explore these possibilities, it appears that although associated with the presence of metopic synostosis, the risk of developmental delays in young children is unrelated to further variation in trigonocephalic shape.
Neurosurgery 08/2010; 67(2):408-15; discussion 415-6. · 2.79 Impact Factor
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ABSTRACT: To test the hypothesis that children with orofacial clefts score lower than controls on measures of language and reading and to examine predictors of these outcomes.
Longitudinal study tracking the development of children with and without orofacial clefts from infancy through age 7 years.
Children with isolated cleft lip and palate (n = 29) and cleft palate only (n = 28) were recruited from the craniofacial program in an urban medical center. Seventy-seven demographically similar, unaffected controls were recruited via advertisements placed in area pediatric clinics and community centers.
Infant measures assessed child development and mother-child interactions during feeding and teaching tasks. At ages 5 and 7 years, measures of language functioning and academic achievement were completed along with an interview to collect school placement data and information on speech services received.
There were no significant group differences in language at ages 5 and 7 years. Children with clefts scored significantly higher than controls on measures of early reading at age 7 years. Outcomes were predicted by demographic factors, the quality of mother-child interactions during teaching and feeding tasks, and cognitive development scores at age 24 months.
Findings do not support the hypothesis that children with clefts score lower than controls on neurocognitive and academic achievement measures. Predictive analyses revealed several dimensions that may be used in clinical practice to identify children at risk for learning and developmental concerns.
The Cleft Palate-Craniofacial Journal 05/2010; 47(3):284-92. · 0.82 Impact Factor
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ABSTRACT: We assessed the neurodevelopment of infants with and without deformational plagiocephaly (DP), at an average age of 6 months.
The Bayley Scales of Infant Development III (BSID-III) were administered to 235 case subjects and 237 demographically similar, control participants. Three-dimensional head photographs were randomized and rated for severity of deformation by 2 craniofacial dysmorphologists who were blinded to case status.
We excluded 2 case subjects with no photographic evidence of DP and 70 control subjects who were judged to have some degree of DP. With control for age, gender, and socioeconomic status, case subjects performed worse than control subjects on all BSID-III scales and subscales. Case subjects' average scores on the motor composite scale were approximately 10 points lower than control subjects' average scores (P < .001). Differences for the cognitive and language composite scales were approximately 5 points, on average (P < .001 for both scales). In subscale analyses, case subjects' gross-motor deficits were greater than their fine-motor deficits. Among case subjects, there was no association between BSID-III performance and the presence of torticollis or infant age at diagnosis.
DP seems to be associated with early neurodevelopmental disadvantage, which is most evident in motor functions. After follow-up evaluations of this cohort at 18 and 36 months, we will assess the stability of this finding. These data do not necessarily imply that DP causes neurodevelopmental delay; they indicate only that DP is a marker of elevated risk for delays. Pediatricians should monitor closely the development of infants with this condition.
PEDIATRICS 02/2010; 125(3):e537-42. · 4.47 Impact Factor
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ABSTRACT: Children with craniofacial anomalies are at high risk for sleep-disordered breathing (SDB), yet its prevalence among children with craniofacial conditions is not known. Children with hemifacial microsomia (HFM) are likely particularly vulnerable to SDB as a result of underdevelopment of the mandible and oropharynx. Nevertheless, most children with HFM are not referred for sleep studies. We hypothesized that sleep outcomes would be worse in children with HFM versus control subjects.
We conducted a follow-up study among 124 case participants and 349 control subjects who previously participated in a study of HFM risk factors. Parents completed the Pediatric Sleep Questionnaire (PSQ) regarding symptoms of SDB and sleep habits. Regression models were adjusted for region, age, sex, race/ethnicity, and maternal education.
Snoring was more commonly reported for children with HFM (29%) than for control subjects (17%). Compared with control subjects, children with HFM more often had symptoms consistent with SDB. On average, case participants' parents reported 1.9 times as many symptoms on the PSQ breathing scale and 1.3 times more symptoms on the PSQ sleepiness scale than did control subjects' parents, with little difference on the PSQ behavior scale. Parents of children with HFM reported 1.4 times more night awakenings than did control subjects' parents.
Children with HFM experienced more snoring and other symptoms of SDB than did control subjects. Pediatricians should be aware of the increased vulnerability for SDB among children with mandibular or external ear underdevelopment or asymmetry and should refer to a sleep specialist as needed.
PEDIATRICS 09/2009; 124(2):e313-21. · 4.47 Impact Factor
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ABSTRACT: To examine reading and related skills in children with and without orofacial clefts.
Forty-two children with orofacial clefts were recruited from an urban craniofacial center. A demographically similar sample of 43 children without clefts was recruited using community advertisements and a research registry. Participants completed assessments of basic reading, phonological awareness, phonological memory, reading fluency, and rapid naming. Parents completed a semi-structured interview regarding educational and medical history.
Children with clefts scored significantly lower than controls on measures of basic reading, phonological memory, and reading fluency.
This is one of the first studies of reading in children with orofacial clefts to include a control sample. The findings suggest that children with clefts are less adept readers than demographically matched peers without clefts, supporting the need to monitor academic achievement in this population.
Journal of Pediatric Psychology 07/2009; 35(2):199-208. · 2.91 Impact Factor
