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  • 1
    Online Resource
    Online Resource
    Methods and Challenges in a Cohort Study of Infants and Toddlers With Craniofacial Microsomia: The Clock Study
    SAGE Publications ; 2019
    In:  The Cleft Palate-Craniofacial Journal Vol. 56, No. 7 ( 2019-08), p. 877-889
    Details
    In: The Cleft Palate-Craniofacial Journal, SAGE Publications, Vol. 56, No. 7 ( 2019-08), p. 877-889
    Abstract: 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.
    Type of Medium: Online Resource
    ISSN: 1055-6656 , 1545-1569
    URL: Article
    DOI: 10.1177/1055665618821014
    RVK:
    XA 36625
    Language: English
    Publisher: SAGE Publications
    Publication Date: 2019
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  • 2
    Online Resource
    Online Resource
    Behavioral Adjustment of Preschool Children With and Without Craniofacial Microsomia
    SAGE Publications ; 2021
    In:  The Cleft Palate-Craniofacial Journal Vol. 58, No. 1 ( 2021-01), p. 42-53
    Details
    In: The Cleft Palate-Craniofacial Journal, SAGE Publications, Vol. 58, No. 1 ( 2021-01), p. 42-53
    Abstract: 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.
    Type of Medium: Online Resource
    ISSN: 1055-6656 , 1545-1569
    URL: Article
    DOI: 10.1177/1055665620947987
    RVK:
    XA 36625
    Language: English
    Publisher: SAGE Publications
    Publication Date: 2021
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  • 3
    Online Resource
    Online Resource
    Longitudinal, Three-Dimensional Analysis of Head Shape in Children with and without Deformational Plagiocephaly or Brachycephaly
    Elsevier BV ; 2012
    In:  The Journal of Pediatrics Vol. 160, No. 4 ( 2012-04), p. 673-678.e1
    Details
    In: The Journal of Pediatrics, Elsevier BV, Vol. 160, No. 4 ( 2012-04), p. 673-678.e1
    Type of Medium: Online Resource
    ISSN: 0022-3476
    URL: Article
    DOI: 10.1016/j.jpeds.2011.09.059
    Language: English
    Publisher: Elsevier BV
    Publication Date: 2012
    detail.hit.zdb_id: 2005245-5
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  • 4
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    Neurodevelopment of Infants with and without Craniofacial Microsomia
    Elsevier BV ; 2018
    In:  The Journal of Pediatrics Vol. 198 ( 2018-07), p. 226-233.e3
    Details
    In: The Journal of Pediatrics, Elsevier BV, Vol. 198 ( 2018-07), p. 226-233.e3
    Type of Medium: Online Resource
    ISSN: 0022-3476
    URL: Article
    DOI: 10.1016/j.jpeds.2018.02.076
    Language: English
    Publisher: Elsevier BV
    Publication Date: 2018
    detail.hit.zdb_id: 2005245-5
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  • 5
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    Cognitive, Motor, and Language Development of Preschool Children With Craniofacial Microsomia
    SAGE Publications ; 2021
    In:  The Cleft Palate-Craniofacial Journal Vol. 58, No. 9 ( 2021-09), p. 1169-1177
    Details
    In: The Cleft Palate-Craniofacial Journal, SAGE Publications, Vol. 58, No. 9 ( 2021-09), p. 1169-1177
    Abstract: 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.
    Type of Medium: Online Resource
    ISSN: 1055-6656 , 1545-1569
    URL: Article
    DOI: 10.1177/1055665620980223
    RVK:
    XA 36625
    Language: English
    Publisher: SAGE Publications
    Publication Date: 2021
    detail.hit.zdb_id: 2030056-6
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  • 6
    Online Resource
    Online Resource
    Speech, Language, and Communication Skills of Adolescents With Craniofacial Microsomia
    American Speech Language Hearing Association ; 2019
    In:  American Journal of Speech-Language Pathology Vol. 28, No. 4 ( 2019-11-19), p. 1571-1581
    Details
    In: American Journal of Speech-Language Pathology, American Speech Language Hearing Association, Vol. 28, No. 4 ( 2019-11-19), p. 1571-1581
    Abstract: Youth with craniofacial microsomia (CFM) have anomalies and comorbidities that increase their risk for speech, language, and communication deficits. We examined these outcomes in youth with and without CFM and explored differences as a function of CFM phenotype and hearing status. Method Participants included youth ages 11–17 years with CFM ( n = 107) and demographically similar controls ( n = 306). We assessed speech intelligibility, articulation, receptive and expressive language, and parent and teacher report measures of communication. Hearing status was also screened at the study visit. Group differences were estimated using linear regression analyses with standardized effect sizes (ES) adjusted for demographic characteristics (adjusted ES) or negative binomial regression. Results Youth with CFM scored lower than unaffected peers on most measures of intelligibility, articulation, expressive language, and parent- and-teacher-rated communication. Differences were most pronounced among participants with CFM who had mandibular hypoplasia plus microtia (adjusted ES = −1.15 to −0.18). Group differences were larger in youth with CFM who failed the hearing screen (adjusted ES = −0.73 to 0.07) than in those who passed the hearing screen (adjusted ES = −0.34 to 0.27). Conclusions Youth with CFM, particularly those with mandibular hypoplasia plus microtia and/or hearing loss, should be closely monitored for speech and language concerns. Further research is needed to identify the specific needs of youth with CFM as well as to document the course of speech and language development in children with CFM.
    Type of Medium: Online Resource
    ISSN: 1058-0360 , 1558-9110
    URL: Article
    DOI: 10.1044/2019_AJSLP-19-0089
    Language: English
    Publisher: American Speech Language Hearing Association
    Publication Date: 2019
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  • 7
    Online Resource
    Online Resource
    Development at Age 36 Months in Children With Deformational Plagiocephaly
    American Academy of Pediatrics (AAP) ; 2013
    In:  Pediatrics Vol. 131, No. 1 ( 2013-01-01), p. e109-e115
    Details
    In: Pediatrics, American Academy of Pediatrics (AAP), Vol. 131, No. 1 ( 2013-01-01), p. e109-e115
    Abstract: 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.
    Type of Medium: Online Resource
    ISSN: 0031-4005 , 1098-4275
    URL: Article
    DOI: 10.1542/peds.2012-1779
    Language: English
    Publisher: American Academy of Pediatrics (AAP)
    Publication Date: 2013
    detail.hit.zdb_id: 1477004-0
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  • 8
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    Online Resource
    Characterizing facial features in individuals with craniofacial microsomia: A systematic approach for clinical research
    Wiley ; 2016
    In:  Birth Defects Research Part A: Clinical and Molecular Teratology Vol. 106, No. 11 ( 2016-11), p. 915-926
    Details
    In: Birth Defects Research Part A: Clinical and Molecular Teratology, Wiley, Vol. 106, No. 11 ( 2016-11), p. 915-926
    Abstract: 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 O rbital, E ar, M andible, N erve, S oft 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.
    Type of Medium: Online Resource
    ISSN: 1542-0752 , 1542-0760
    URL: Issue
    URL: Article
    DOI: 10.1002/bdra.v106.11
    DOI: 10.1002/bdra.23560
    Language: English
    Publisher: Wiley
    Publication Date: 2016
    detail.hit.zdb_id: 2108606-0
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  • 9
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    Online Resource
    Behavioral-Social Adjustment of Adolescents with Craniofacial Microsomia
    SAGE Publications ; 2018
    In:  The Cleft Palate-Craniofacial Journal Vol. 55, No. 5 ( 2018-05), p. 664-675
    Details
    In: The Cleft Palate-Craniofacial Journal, SAGE Publications, Vol. 55, No. 5 ( 2018-05), p. 664-675
    Abstract: 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.
    Type of Medium: Online Resource
    ISSN: 1055-6656 , 1545-1569
    URL: Article
    DOI: 10.1177/1055665617750488
    RVK:
    XA 36625
    Language: English
    Publisher: SAGE Publications
    Publication Date: 2018
    detail.hit.zdb_id: 2030056-6
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  • 10
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    Intelligence and Academic Achievement of Adolescents with Craniofacial Microsomia
    Ovid Technologies (Wolters Kluwer Health) ; 2017
    In:  Plastic & Reconstructive Surgery Vol. 140, No. 3 ( 2017-09), p. 571-580
    Details
    In: Plastic & Reconstructive Surgery, Ovid Technologies (Wolters Kluwer Health), Vol. 140, No. 3 ( 2017-09), p. 571-580
    Abstract: The authors compared the IQ and academic achievement of adolescents with craniofacial microsomia (cases) and unaffected children (controls). Among cases, the authors analyzed cognitive functioning by facial phenotype. Methods: The authors administered standardized tests of intelligence, reading, spelling, writing, and mathematics to 142 cases and 316 controls recruited from 26 cities across the United States and Canada. Phenotypic classification was based on integrated data from photographic images, health history, and medical chart reviews. Hearing screens were conducted for all participants. Results: After adjustment for demographics, cases’ average scores were lower than those of controls on all measures, but the magnitude of differences was small (standardized effect sizes, −0.01 to −0.3). There was little evidence that hearing status modified case-control group differences (Wald p 〉 0.05 for all measures). Twenty-five percent of controls and 38 percent of cases were classified as having learning problems (adjusted OR, 1.5; 95 percent CI, 0.9 to 2.4). Comparison of cases with and without learning problems indicated that those with learning problems were more likely to be male, Hispanic, and to come from lower income, bilingual families. Analyses by facial phenotype showed that case-control group differences were largest for cases with both microtia and mandibular hypoplasia (effect sizes, −0.02 to −0.6). Conclusions: The highest risk of cognitive-academic problems was observed in patients with combined microtia and mandibular hypoplasia. Developmental surveillance of this subgroup is recommended, especially in the context of high socioeconomic risk and bilingual families. Given the early stage of research on craniofacial microsomia and neurodevelopment, replication of these findings is needed. CLINICAL QUESTION/LEVEL OF EVIDENCE: Risk, II.
    Type of Medium: Online Resource
    ISSN: 0032-1052
    URL: Article
    DOI: 10.1097/PRS.0000000000003584
    Language: English
    Publisher: Ovid Technologies (Wolters Kluwer Health)
    Publication Date: 2017
    detail.hit.zdb_id: 2037030-1
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