Matthew L Speltz1, Kathleen A Kapp-Simon2, Alexis L Johns3, Erin R Wallace4, Brent R Collett5, Leanne Magee6, Brian G Leroux7, Daniela V Luquetti5, Carrie L Heike5. 1. Centers for Child Health, Behavior and Development, Developmental Biology & Regenerative Medicine, and Clinical and Translational Research, Seattle Children's Research Institute, Seattle, WA; Craniofacial Center, Seattle Children's Hospital, Seattle, WA; Departments of Pediatrics and Psychiatry and Behavioral Medicine, University of Washington School of Medicine, Seattle, WA. Electronic address: matt.speltz@seattlechildrens.org. 2. Cleft-Craniofacial Center, Shriners Hospital for Children, Chicago, IL; Craniofacial Center, Department of Surgery, University of Illinois, Chicago, IL. 3. Division of Plastic and Maxillofacial Surgery, Children's Hospital of Los Angeles, Los Angeles, CA. 4. Centers for Child Health, Behavior and Development, Developmental Biology & Regenerative Medicine, and Clinical and Translational Research, Seattle Children's Research Institute, Seattle, WA. 5. Centers for Child Health, Behavior and Development, Developmental Biology & Regenerative Medicine, and Clinical and Translational Research, Seattle Children's Research Institute, Seattle, WA; Craniofacial Center, Seattle Children's Hospital, Seattle, WA; Departments of Pediatrics and Psychiatry and Behavioral Medicine, University of Washington School of Medicine, Seattle, WA. 6. Division of Plastic and Reconstructive Surgery, Children's Hospital of Philadelphia, Philadelphia, PA. 7. University of Washington School of Dentistry, Seattle, WA.
Abstract
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.
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.
Authors: M S Cohen; C A Samango-Sprouse; H J Stern; D A Custer; D R Vaught; H M Saal; C J Tifft; K N Rosenbaum Journal: Am J Med Genet Date: 1995-12-18
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Authors: Alexis L Johns; Erin R Wallace; Brent R Collett; Kathleen A Kapp-Simon; Amelia F Drake; Carrie L Heike; Sara L Kinter; Daniela V Luquetti; Leanne Magee; Susan Norton; Kathleen Sie; Matthew L Speltz Journal: Cleft Palate Craniofac J Date: 2020-08-12