Neurodevelopment of Infants with and without Craniofacial Microsomia

Abstract

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.

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

“…T1 assessments are summarized in the Supplemental Material. The results of case–control comparisons of neurodevelopment and facial expressivity at time 1 have been published elsewhere (Hammal et al, 2018; Speltz et al, 2018, respectively). Future articles will address group comparisons of other outcomes collected at T1 (eg, hearing, speech), group comparisons of multiple outcomes at T3, and the results of predictive analyses of T3 outcomes from T1 data (eg, facial expressiveness at T1 to measures of socialization and behavioral adjustment at T3).…”

“…Craniofacial microsomia can lead to several functional problems of significance (eg, impaired speech and hearing), but none may be as poorly understood or as potentially modifiable as those associated with early neurodevelopment (Speltz et al, 2018). For this reason, the CLOCK project has prioritized the longitudinal collection of cognitive, motor, and language outcomes.…”

“…As the present article is a descriptive study, we did not conduct statistical comparisons. Case-control comparisons of initial (T1) neurodevelopmental outcomes have been previously published (Speltz et al, 2018) and follow-up comparisons of group differences in neurodevelopment and other outcomes at T3 will be presented in future articles.…”

“…The long-term goal of our research consortium is to improve health for individuals with CFM as defined by the World Health Organization: “a state of complete physical, mental, and social well-being” (World Health Organization, 1946). This project is using a multicenter longitudinal cohort design to investigate the neurodevelopmental, social, and behavioral outcomes of infants and toddlers with and without CFM (Hammal et al, 2018; Speltz et al, 2018). The rationale underlying this study is that better understanding of the direct and indirect effects of CFM on early neurodevelopment may help clinicians identify children at risk for poor neurodevelopment and lead to the design of early interventions to reduce such risk.…”

Dynamics of Face and Head Movement in Infants with and without Craniofacial Microsomia: An Automatic Approach

“…We classified the participant’s phenotype with a case-by-case integration of standardized photographic ratings of facial features and data taken from a medical history interview and medical charts. 26 The photographic protocol and classification method 27 generated 3 phenotypic subgroups: Microtia only (absence of other CFM-related features; n = 16); Microtia plus mandibular hypoplasia (n = 38); and Other combinations of CFM- associated malformations (n = 9).…”