Christy Michelle McKinney

Objective: To develop and compare models predicting which infants with cleft lip and/or palate (CL/P) will be underweight at 4 months, using multicenter electronic health record data. Design: Using data from birth to 3 months, we evaluated 63 anthropometric, clinical, healthcare-utilization, and demographic variables across 6 regression and machine-learning models to identify the strongest predictors of underweight status, overall and by cleft type. Setting: Ten US children's hospitals in PEDSnet, a national pediatric learning health network, 2009 to 2024. Patients: Infants with CL/P aged 0 to 4.5 months. Main Outcome Measure: Underweight status at 4 months (range 3.5-4.5), defined as weight-for-age z-score (WAZ) < −2. Results: Among 5717 infants with CL/P (19% cleft lip, 35% cleft lip and palate, 46% cleft palate), 25.9% were underweight at 4 months. The strongest predictors were early (0-3 months) growth measures: mean WAZ (58% of predictive value), WAZ velocity, mean length-for-age z-score (LAZ), and LAZ velocity, plus the Child Opportunity Index (a neighborhood socioeconomic measure; ∼2%). These 5 predictors accounted for >85% of predictive value in every cleft type. All 6 models discriminated similarly (area under the receiver operating characteristic curve 0.87-0.90); GPBoost, which also accounts for hospital differences, produced risk estimates closest to the actual proportion of underweight infants. Conclusions: Routine early-life growth measures (WAZ, LAZ, and their trajectories), together with neighborhood socioeconomic status, can identify infants with cleft at elevated risk of being underweight before 4 months, helping craniofacial and primary care teams provide timelier feeding and nutrition support and referrals to the highest-risk infants.

Objectives: To evaluate growth patterns in US children with cleft overall and by cleft lip (CL), CL and palate (CLP), and cleft palate (CP) across US children’s hospitals from 0 to 18 mo of age. Design: We conducted a retrospective cohort study leveraging medical records from 2009 to 2022 in children with a cleft seen at 8 US children’s hospitals. Participants: Individuals with a cleft seen at a PEDSnet consortium hospital. Exposure: We examined growth over time, by cleft type, and across hospitals. Main Outcomes: We used electronic health data to generate weight-for-age z scores (WAZ) and length-for-age z scores (LAZ), as well as underweight (WAZ <–2) and stunting (LAZ <–2). We tracked longitudinal growth using generalized linear mixed models to estimate mean WAZ and LAZ from 0 to 18 mo of age. We compared the prevalence of underweight and stunting against World Health Organization growth standards. Results: Our sample included 10,223 children: 19.1% with CL, 31.7% with CLP, and 49.2% with CP. WAZ and LAZ trajectories showed a fast rate of decline in the first 4 mo of life (P < 0.001). Between 6 and 12 mo and between 12 and 18 mo, WAZ showed a significant nonlinear increase in growth (P < 0.01) while LAZ remained unchanged (P > 0.05). All children had a higher prevalence of underweight and stunting relative to World Health Organization growth standards. Underweight and stunting were highest among those with CP, followed by CLP and then CL, and varied across hospitals. Conclusions: Growth trajectories were slower in the first 4 mo of life, followed by rapid catch-up in weight to 12 mo of age. Children with cleft had a high prevalence of underweight and stunting at all ages. Variability across hospitals suggests that care patterns may affect growth. Close growth monitoring in the first 4 mo in children with cleft may be warranted. Knowledge Transfer Statement: US children with a cleft of the lip and/or palate have substantial growth deficits that vary by cleft type, comorbidities, and across hospitals, particularly in the first 4 mo of life.

Objective: To propose recommendations for ethical, participant-centered clinical data sharing in craniofacial research. Design: Series of deliberative multidisciplinary expert working group meetings to develop recommendations. Setting: Two 1-h virtual meetings; one all-day hybrid meeting. Patients, Participants: Working group ( n = 16) comprised individuals with expertise in craniofacial research, bioethics, and patient/caregiver advocacy, as well as lived experience of craniofacial conditions. Interventions: The working group first reviewed prior empirical data about research participant attitudes about data sharing then drafted initial recommendations that were built on the data and the group's collective expertise. Recommendations were iteratively refined until the group agreed upon their final presentation. Main outcome measures: Working group endorsement of recommendations. Results: The working group produced 16 recommendations that addressed considerations for primary and secondary researchers, data repositories, and the craniofacial research community across 5 domains. These domains address: (1) research team communication with participants, (2) data collection and protections, (3) data governance, (4) education for researchers, and (5) remaining research gaps. Recommendations highlight the importance of prioritizing the experiences of those with lived experience of craniofacial conditions in decision-making about data sharing, navigating varied perspectives on privacy protections for facial images, and striving to implement trustworthy data sharing and governance practices. Conclusions: This summary of recommendations offers guidance for the craniofacial research community to advance participant-centered clinical data sharing practices. Ethical data sharing that accounts for participants’ experiences and values has potential to advance scientific research and improve outcomes for individuals with craniofacial conditions, their families, and their communities.

Objective: To conduct a systematic review on growth in young children with a cleft of the lip and/or palate (“cleft”). Design: We searched 6 electronic databases for articles published on or before February 9, 2025 using MESH terms cleft lip/palate, growth, body weights/measures. We screened titles and abstracts iteratively using Rayyan's screening tool and manual review. Eligible studies underwent full-text review and anthropometric data extraction and synthesis. We excluded studies/estimates with N < 20. Setting: All published studies that reported anthropometric measures on children with cleft. Patients/Participants: Children with cleft of age 0-24 months. Main Outcome Measures: Comparable measures including weight and length percentiles, failure to thrive (typically z -scores (WAZ), length-for-age z -scores (LAZ), moderate/severe underweight (WAZ < −2), and moderate/severe stunting (LAZ < −2). Results: 289 articles were selected for full-text review following title/abstract screening of the initially identified 5561 unique articles. 143 met the eligibility criteria, 46 of which had comparable anthropometric estimates. The estimates in most studies were lower than expected based on referent growth charts, and demonstrated mean WAZ near one standard deviation below the mean or between 10% and 50% of infants had failure to thrive and/or were underweight. Measures of growth related to length showed deficits but to a lesser degree than weight-based measures. Data beyond age 12 months were sparse. Conclusions: Regardless of cleft type, young children with a cleft have worse attained growth than peers based on referent growth charts. Children with a cleft palate, with or without a cleft lip, had lower estimates than children with cleft lip.

Retrospective cohort studies, defined as a follow-up study in which outcome occurred prior to study onset, are common in craniofacial outcomes research and will continue to be prevalent given the increasing availability of secondary datasets and inherent prospective study limitations. However, if available data are not adequately measured, or necessary variables are absent, retrospective cohort studies can be particularly prone to bias. This brief communication aims to highlight the primary sources of bias, including measurement error, selection bias, and confounding. Each source is clearly defined, examples pertinent to craniofacial outcomes are provided, and mitigation strategies are discussed.

Background: Dentists often use sedative medications such as nitrous oxide inhaled sedation and general anesthesia (GA) to help decrease patient fear and manage paediatric patients' behavior during treatment.Aim: The goal of this study was to examine factors associated with dental fear changes after restorative dental treatment under nitrous oxide or GA in children 4-12 years old.Design: A prospective cohort study of 124 children examined changes in dental fear, number of treatment visits, and parental factors among children receiving restorative dental treatment under nitrous oxide (n = 68) or GA (n = 56) sedation. Data were collected at pretreatment (T1), 16 weeks post-treatment (T2), and at 29-month follow-up (T3).Results: Dental fear increased slightly, but not significantly, under both forms of sedation between T1 and T3. Children's dental fear was associated with parents' poor dental experiences and oral health, but not with number of treatment visits.Conclusions: Progression of children's dental fear seems not dependent solely on the type of sedation used but is likely predicted by factors including pretreatment dental fear and dental needs. Dentists recommending sedation for children's dental care may consider pretreatment dental need, fear levels, and parental factors when determining which type of sedation to use.

Objective: Craniofacial microsomia (CFM) is a broad clinical term used to describe a congenital condition most commonly involving the underdevelopment of the external ear, mandible, soft tissues, and facial nerve. Despite medical advances, understanding of the psychological health and healthcare experiences of individuals with CFM and their caregivers remains limited. This article describes a research program designed to address these knowledge gaps, and identify opportunities for psychosocial intervention and improved healthcare provision. Design: The Craniofacial microsomia: Accelerating Research and Education (CARE) research program aims to: 1) Conduct up to 160 narrative interviews with individuals and caregivers to validate a conceptual framework; 2) Administer an online international survey of up to 800 individuals with CFM and caregivers to identify predictors of psychological distress; 3) Perform up to 60 semi-structured interviews with healthcare providers and advocacy leaders to examine the extent to which current healthcare provisions address identified patient needs; and 4) Establish a participant registry to build a longitudinal database and develop an international community. Results: Teams in the USA and UK have been established, alongside an international, interdisciplinary Advisory Committee. Data analysis for Aim 1 is ongoing and informing the delivery of Aims 2-3. Aim 4 is also in development. A dedicated website serves as a recruitment tool, educational resource, and mechanism for engaging with the CFM community. Conclusions: The CARE program provides a comprehensive approach to understanding the experiences of individuals with CFM and their caregivers. Challenges encountered and lessons learned are shared for the benefit of the community.

Objective: Our goal was to compare data collected from 3- and 7-day Infant with Clefts Observation Outcomes (iCOO) diaries. Design: Secondary data analysis of an observational longitudinal cohort study. Caregivers completed the daily iCOO for 7 days before cleft lip surgery (T0) and for 7 days after cleft lip repair (T1). We compared 3- and 7-day diaries collected at T0 and 3- and 7-day diaries collected at T1. Setting: United States. Participants: Primary caregivers of infants with cleft lip with and without cleft palate (N = 131) planning lip repair and enrolled in original iCOO study. Main Outcomes Measure(s): Mean differences and Pearson correlation coefficients. Results: Correlation coefficients were high for global impressions (>0.90) and scaled scores (0.80–0.98). Mean differences were small across iCOO domains at T0. T1 comparisons reflected the same pattern. Conclusions: Three-day diary data is comparable to 7-day diaries for measuring caregiver observations using iCOO across T0 and T1.