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  • Magee, Leanne  (5)
  • Speltz, Matthew L.  (5)
  • 1
    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
    RVK:
    Language: English
    Publisher: SAGE Publications
    Publication Date: 2019
    detail.hit.zdb_id: 2030056-6
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  • 2
    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
    RVK:
    Language: English
    Publisher: SAGE Publications
    Publication Date: 2021
    detail.hit.zdb_id: 2030056-6
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  • 3
    In: The Journal of Pediatrics, Elsevier BV, Vol. 198 ( 2018-07), p. 226-233.e3
    Type of Medium: Online Resource
    ISSN: 0022-3476
    Language: English
    Publisher: Elsevier BV
    Publication Date: 2018
    detail.hit.zdb_id: 2005245-5
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  • 4
    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
    RVK:
    Language: English
    Publisher: SAGE Publications
    Publication Date: 2021
    detail.hit.zdb_id: 2030056-6
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  • 5
    In: Birth Defects Research, Wiley, Vol. 111, No. 11 ( 2019-07), p. 649-658
    Abstract: Craniofacial microsomia (CFM) is a congenital condition that typically involves hypoplasia of the ear and jaw. It is often associated with adverse effects such as hearing loss and sleep‐disordered breathing. There is little research on its etiology. Methods We conducted a case–control study from maternal interview data collected from mothers of infants with and without CFM. The study included 108 children with and 84 children without CFM. Logistic regression with adjustment for demographic factors was used to evaluate associations between maternal exposures of interest and risk for CFM overall, as well as for different phenotypic sub‐groups of children on the CFM spectrum. Results We found a statistically significant association between diabetes mellitus (DM) and CFM (OR 4.01, 95% CI 1.6–10.5). The association was slightly attenuated after adjustment for BMI. Higher parity was also associated with increased risk for CFM (OR 2.0, 95% CI 1.0–4.0). Vitamin A consumption and/or liver consumption was associated with a 70% lower risk compared with non‐users (OR 0.3, 95% 0.1–0.8). Maternal age at the time of pregnancy was not associated with CFM. Conclusions These analyses contribute evidence linking maternal DM with an elevated risk of having an infant with CFM, which is consistent with previous research and adds to the body of knowledge about the strength of this association. Further study is warranted to understand the potential mechanisms underlying the effect of DM in the developing embryo.
    Type of Medium: Online Resource
    ISSN: 2472-1727 , 2472-1727
    URL: Issue
    Language: English
    Publisher: Wiley
    Publication Date: 2019
    detail.hit.zdb_id: 2884154-2
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