A Conceptual Thematic Framework of Psychological Adjustment in Caregivers of Children with Craniofacial Microsomia.

OBJECTIVE: Children with craniofacial microsomia (CFM) have complex healthcare needs, resulting in evaluations and interventions from infancy onward. Yet, little is understood about families' treatment experiences or the impact of CFM on caregivers' well-being. To address this gap, the NIH-funded 'Craniofacial microsomia: Accelerating Research and Education (CARE)' program sought to develop a conceptual thematic framework of caregiver adjustment to CFM. DESIGN: Caregivers reported on their child's medical and surgical history. Narrative interviews were conducted with US caregivers (n = 62) of children aged 3-17 years with CFM. Transcripts were inductively coded and final themes and subthemes were identified. RESULTS: Components of the framework included: 1) Diagnostic Experiences, including pregnancy and birth, initial emotional responses, communication about the diagnosis by healthcare providers, and information-seeking behaviors; 2) Child Health and Healthcare Experiences, including feeding, the child's physical health, burden of care, medical decision-making, surgical experiences, and the perceived quality of care; 3) Child Development, including cognition and behavior, educational provision, social experiences, and emotional well-being; and 4) Family Functioning, including parental well-being, relationships, coping strategies, and personal growth. Participants also identified a series of "high" and "low" points throughout their journey and shared their priorities for future research. CONCLUSIONS: Narrative interviews provided rich insight into caregivers' experiences of having a child with CFM and enabled the development of a conceptual thematic framework to guide clinical care and future research. Information gathered from this study demonstrates the need to incorporate evidence-based psychological support for families into the CFM pathway from birth onward.

Oral Structural Dysphagia in Children.

Oral causes of dysphagia in infancy may involve the lips, the tongue, or the palate. Whereas ankyloglossia is commonly diagnosed in infants with dysphagia, assessment of the need for surgical intervention may be less straightforward. Tongue size (macroglossia) may be associated with dysphagia as it may cause limitation of movement of the food or milk bolus by the lips or cheeks. Congenital conditions such as cleft lip and palate, micrognathia, or craniofacial microsomia may also be associated with dysphagia. Diagnosis and treatment of these conditions can be improved with the engagement of lactation and feeding experts as well as multidisciplinary craniofacial teams.

Laryngeal Structural Dysphagia in Children.

The upper aerodigestive system is closely intertwined from an embryologic and functional perspective. Laryngotracheal anatomic abnormalities, such as laryngomalacia, stenosis, vocal cord paralysis, and laryngeal clefts, affect not only the respiratory function but also the swallow function. Laryngotracheal pathology can interfere with the suck-swallow-breathe mechanism in infants. It can also exacerbate gastroesophageal reflux. Chronic aspiration secondary to laryngotracheal anomalies can result in respiratory and pulmonary complications. Surgical treatment of laryngotracheal anomalies can also cause transient or long-term swallow dysfunction. Multidisciplinary approaches and clinical assessment of swallowing are important in patients with laryngotracheal pathology.

Early Experiences of Parents of Children With Craniofacial Microsomia.

OBJECTIVE: To describe the early health care experiences of parents of children with craniofacial microsomia (CFM), a congenital diagnosis often identified at birth. DESIGN: Qualitative descriptive. SETTING: Homes of participants. PARTICIPANTS: Parents of 28 children with CFM from across the United States. METHODS: We interviewed participants (27 mothers individually and one mother and father together) via telephone or teleconference and used reflexive thematic analysis to derive themes that represented early health care experiences of parents of children with CFM. RESULTS: Participants' narratives included detailed recounting of their birth and early care experiences. We identified two overarching themes. The first overarching theme, Stressors, included four subthemes that represented difficulties related to emotional reactions and negative experiences with health care providers. The second overarching theme, Finding Strength, included four subthemes that represented participants' positive adjustment to stressors through independent information seeking about CFM, adaptive coping, positive experiences with health care providers, and drawing on external supports. CONCLUSION: Participants often described early experiences as challenging. Findings have implications for improving early care, including increasing open and supportive communication by health care professionals, expanding access to CFM information, screening for mental health concerns among parents, strengthening coping among parents, and linking families to resources such as reliable online CFM information and early intervention programs.

Establishing an International Interdisciplinary Research Network in Craniofacial Microsomia: The CARE Program.

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.

Building diverse leadership in an academic medical center: The ACCLAIM program.

As healthcare systems become more complex, medical education needs to adapt in many ways. There is a growing need for more formal leadership learning for healthcare providers, including greater attention to health disparities. An important challenge in addressing health disparities is ensuring inclusive excellence in the leadership of healthcare systems and medical education. Women and those who are underrepresented in medicine (URMs) have historically had fewer opportunities for leadership development and are less likely to hold leadership roles and receive promotions. One successful initiative for improved learning of medical leadership-presented as a case example here-is the Academic Career Leadership Academy in Medicine (ACCLAIM) at the University of North Carolina at Chapel Hill School of Medicine. ACCLAIM is uniquely designed for faculty identified as having emerging leadership potential, with an emphasis on women and URMs. Using a leadership learning system approach, annual cohorts of participants (Scholars) interactively participate in a multi-faceted nine-month long learning experience, including group (e.g., guest-speaker weekly presentations and exercises) and individual learning components (e.g., an individual leadership project). Since its initiation in 2012 and through 2021, 111 Scholars have participated in ACCLAIM; included were 57% women and 27% URMs. Two important outcomes described are: short-term impact as illustrated by consistent improvements in quantitively measured leadership knowledge and capabilities; and long-term leadership growth, whereby half of the ACCLAIM graduates have received academic rank promotions and almost two-thirds have achieved new leadership opportunities, with even higher percentages observed for women and URMs; for example, 87% of URMs were either promoted or achieved new leadership positions. Also consistently noted, through qualitative assessments, are broader healthcare system knowledge and shared tactics for addressing common challenges among Scholars. This case example shows that the promotion of leadership equity may jointly enhance professional development while creating opportunities for systems change within academic medical centers. Such an approach can be a potential model for academic medical institutions and other healthcare schools seeking to promote leadership equity and inclusion.

Body Dysmorphic Disorder in Adult Patients With an Orofacial Cleft: An Unseen Psychological Burden.

OBJECTIVES: Facial dysmorphic disorder (FDD), a variant of body dysmorphic disorder, occurs when individuals are preoccupied with perceived defects in their facial appearance. Cleft lip and/or palate (CL/P) requires many clinical interventions and has significant psychological impacts on a patient's perception of appearance. This study identified psychological burdens related to living as an adult with CL/P and characterizes the degree of FDD symptoms in an adult craniofacial population. METHODS: This was a prospective, single-center, cross-sectional case-control study using semi-structured interviews and symptom assessments at a university-based craniofacial center. Patients without CL/P undergoing non-cosmetic facial surgery were recruited as controls (n = 20). Patients with an orofacial cleft (n = 30) were recruited from medical and dental providers at the University of North Carolina. Body Dysmorphic Disorder-Yale Brown Obsessive Compulsive Scale (BBD-YBOCS) scores were collected from a control population and patients with CL/P to assess FDD severity. RESULTS: Demographic factors such age, biological sex, and ethnicity had no significant impact on FDD symptom scores. Patient with CL/P were more likely to have significant FDD symptoms (BDD-YBOCS greater than 16) than patients without CL/P (OR 10.5, CI95 2.7-41.1), and had a mean difference in FDD symptoms scores of 10.04 (p < 0.0001; CI95 5.5-14.6). Patients with CL/P seen by a mental health provider in the past 3 months had 3-fold lower overall FDD symptom scores (OR 0.081; CI95 0.0085-0.77). CONCLUSIONS: Adults with CL/P would benefit from treatment for cleft-specific needs and psychological support as they face unique stressors related to their appearance, including an increase in FDD-associated symptoms. This study emphasizes the importance of recognizing psychological symptoms and providing ongoing multidisciplinary care to adults with CL/P. LEVEL OF EVIDENCE: 3; Individual case-control study Laryngoscope, 133:818-821, 2023.

Mentoring Underrepresented Minority Physician-Scientists to Success.

As the nation seeks to recruit and retain physician-scientists, gaps remain in understanding and addressing mitigatable challenges to the success of faculty from underrepresented minority (URM) backgrounds. The Doris Duke Charitable Foundation Fund to Retain Clinical Scientists program, implemented in 2015 at 10 academic medical centers in the United States, seeks to retain physician-scientists at risk of leaving science because of periods of extraordinary family caregiving needs, hardships that URM faculty-especially those who identify as female-are more likely to experience. At the annual Fund to Retain Clinical Scientists program directors conference in 2018, program directors-21% of whom identify as URM individuals and 13% as male-addressed issues that affect URM physician-scientists in particular. Key issues that threaten the retention of URM physician-scientists were identified through focused literature reviews; institutional environmental scans; and structured small- and large-group discussions with program directors, staff, and participants. These issues include bias and discrimination, personal wealth differential, the minority tax (i.e., service burdens placed on URM faculty who represent URM perspectives on committees and at conferences), lack of mentorship training, intersectionality and isolation, concerns about confirming stereotypes, and institutional-level factors. The authors present recommendations for how to create an environment in which URM physician-scientists can expect equitable opportunities to thrive, as institutions demonstrate proactive allyship and remove structural barriers to success. Recommendations include providing universal training to reduce interpersonal bias and discrimination, addressing the consequences of the personal wealth gap through financial counseling and benefits, measuring the service faculty members provide to the institution as advocates for URM faculty issues and compensating them appropriately, supporting URM faculty who wish to engage in national leadership programs, and sustaining institutional policies that address structural and interpersonal barriers to inclusive excellence.

Parental Reports of Intervention Services and Prevalence of Teasing in a Multinational Craniofacial Microsomia Pediatric Study.

ABSTRACT: Children with craniofacial microsomia (CFM) are at increased risk for educational and social concerns. This study describes intervention services and frequency of teasing in a multinational population of children with CFM. Caregivers of children with CFM ages 3 to 18 years in the US and South America were administered a questionnaire. Additional information was gathered from medical charts and photographs. Participants (N = 169) had an average age of 10.1 ±â€Š6.2 years, were primarily male (60%), and from the US (46%) or Colombia (32%). Most participants had microtia and mandibular hypoplasia (70%). They often had unilateral (71%) or bilateral (19%) hearing loss and 53% used a hearing aid. In the US, special education services were provided for 48% of participants enrolled in school; however, similar services were rare (4%) in South America and reflect differences in education systems. Access to any intervention service was higher in the US (80%) than in South America (48%). Caregivers reported children showed diagnosis awareness by an average age of 4.4 ±â€Š1.9 years. Current or past teasing was reported in 41% of the children, starting at a mean age of 6.0 ±â€Š2.4 years, and most often took place at school (86%). As half of the US participants received developmental and academic interventions, providers should screen for needs and facilitate access to services. Given diagnosis awareness at age 4 and teasing at age 6, providers are encouraged to assess for psychosocial concerns and link to resources early in treatment.

Insights from an Intervention to Support Early Career Faculty with Extraprofessional Caregiving Responsibilities.

Background: Insufficient support for balancing career and family responsibilities hinders retention of physician-scientists. Programs to improve retention of this important group of faculty are crucial. Understanding the experiences of program implementers is key to refining and improving program offerings. Methods: We conducted an interpretive, descriptive, and qualitative study as part of an ongoing evaluation of the Doris Duke Charitable Foundation's Fund to Retain Clinical Scientists (FRCS) awards. We conducted telephone interviews with 12 program directors representing all 10 US medical schools who received the Doris Duke funding in 2016. Results: Of the 12 participants, 10 were women (83.3%). Participating program directors perceived the FRCS award as capable of producing paradigmatic changes regarding how responsibilities at home and work in academic medicine are viewed and integrated by early-career faculty members. The main qualitative themes that captured directors' experiences implementing the program were as follows: (1) championing a new paradigm of support, (2) lessons learned while implementing the new paradigm, (3) results of the new paradigm, and (4) sustaining the paradigm. Conclusions: These findings may help to inform development of similar programs to retain and support the career progress of physician-scientists with extraprofessional caregiving responsibilities. The interviews illuminate ways in which the Doris Duke FRCS award has driven institutional culture change by normalizing discussion and prompted reassessment of extraprofessional challenges and how best to aid early-career faculty members in overcoming these challenges.

Haploinsufficiency of SF3B2 causes craniofacial microsomia.

Craniofacial microsomia (CFM) is the second most common congenital facial anomaly, yet its genetic etiology remains unknown. We perform whole-exome or genome sequencing of 146 kindreds with sporadic (n = 138) or familial (n = 8) CFM, identifying a highly significant burden of loss of function variants in SF3B2 (P = 3.8 × 10-10), a component of the U2 small nuclear ribonucleoprotein complex, in probands. We describe twenty individuals from seven kindreds harboring de novo or transmitted haploinsufficient variants in SF3B2. Probands display mandibular hypoplasia, microtia, facial and preauricular tags, epibulbar dermoids, lateral oral clefts in addition to skeletal and cardiac abnormalities. Targeted morpholino knockdown of SF3B2 in Xenopus results in disruption of cranial neural crest precursor formation and subsequent craniofacial cartilage defects, supporting a link between spliceosome mutations and impaired neural crest development in congenital craniofacial disease. The results establish haploinsufficient variants in SF3B2 as the most prevalent genetic cause of CFM, explaining ~3% of sporadic and ~25% of familial cases.

Reduced pharyngeal dimensions and obstructive sleep apnea in adults with cleft lip/palate and Class III malocclusion.

Objective: To three-dimensionally evaluate the upper airway of individuals with cleft lip and palate (CLP) and Class III malocclusion and the occurrence of obstructive sleep apnea (OSA).Methods: Twenty-one CLP individuals with Class III malocclusion, 20-29 years of age, who underwent computed tomography for orthognathic surgery planning, were prospectively evaluated. All participants underwent polysomnography, and the apnea-hypopnea index ≥ 5 events/hour was considered indicative of OSA. The total upper airway and its subdivisions volumes, as well as the minimum pharyngeal cross-sectional area (CSA), were assessed using Mimics software.Results: Among the 21 individuals analyzed, 6 (29%) presented with OSA. The total upper airway and the oropharynx mean volumes were significantly decreased in subjects with OSA when compared to individuals without OSA. Mean CSA was not statistically different between groups.Conclusion: CLP individuals with Class III malocclusion and OSA have an upper airway significantly smaller than individuals without OSA.

Cognitive, Motor, and Language Development of Preschool Children With Craniofacial Microsomia.

OBJECTIVE: 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.

Behavioral Adjustment of Preschool Children With and Without Craniofacial Microsomia.

OBJECTIVE: 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.

Microtia and craniofacial microsomia: Content analysis of facebook groups.

OBJECTIVE: An increasing number of patients use social media for health-related information and social support. This study's objective was to describe the content posted on Facebook groups for individuals with microtia and/or craniofacial microsomia (CFM) and their families in order for providers to gain insight into patient and family needs and experiences to inform clinical care. METHODS: Two months of posts, images, comments, and "like" responses from two Facebook groups in the US and the UK were recorded and analyzed using content analysis. A secondary analysis identified statements of emotion. RESULTS: Posts (N = 254) had a total of 7912 "like" responses, 2245 comments, and 153 images. There were three categories of posts: seeking guidance (43%; 9 themes), promoting events/news (33%; 5 themes), and sharing experiences (24%; 3 themes). Across categories, 16% of posts had emotional content. Most comments were responding to posts seeking guidance, including medical care (20%), surgical care (9%), and hearing aids (5%). Promotional posts often aimed to increase CFM awareness. Posts sharing experiences were generally positive, with the highest number of "likes". CONCLUSIONS: Facebook groups members frequently exchanged health-related information, suggesting value placed on input from other families and the convenience of seeking information online. Posts also promoted awareness and shared experiences. Clinical care implications include the need for easily accessible accurate and tailored CFM-related health education. Additionally, providers should demonstrate awareness of health information on social media and may address the potential emotional impact of CFM by facilitating access to resources for social support.

From Stigma to Validation: A Qualitative Assessment of a Novel National Program to Improve Retention of Physician-Scientists with Caregiving Responsibilities.

Background: Research is needed to improve understanding of work-life integration issues in academic medicine and to guide the implementation of the Doris Duke Charitable Foundation's Fund to Retain Clinical Scientists (FRCS), a national initiative offering financial support to physician-scientists facing caregiving challenges. Materials and Methods: In 2018, as part of a prospective program evaluation, the authors conducted a qualitative study to examine FRCS program participants' initial impressions, solicit descriptions of their career and caregiving experiences, and inquire how such factors might influence their professional advancement. The authors invited all 33 awardees who had been granted FRCS funding in the first year of the program to participate in the study, of whom 28 agreed to complete an interview. Analysts evaluated de-identified transcripts and explicated the data using a thematic analysis approach. Results: While participants described aspects of a culture that harbor stigma against caregivers and impede satisfactory work-life integration, they also perceived an optimistic cultural shift taking place as a result of programs like the FRCS. Their comments indicated that the FRCS has the potential to influence culture if institutional leadership simultaneously fosters a community that validates individuals both as caregivers and as scientists. Conclusions: Insights garnered from this qualitative study suggest that there is a pressing need for institutional leaders to implement programs that can foster awareness and normalization of caregiving challenges. In addition to providing funding and other tangible resources, interventions should strive to reinforce a broader culture that affirms the presence of work-life integration challenges and openly embraces solutions.

Craniofacial Interventions in Children.

Craniofacial interventions are common and the surgical options continue to grow. The issues encountered include micrognathia, macroglossia, midface hypoplasia, hearing loss, facial nerve palsy, hemifacial microsomia, and microtia. In addition, a unifying theme is complex upper airway obstruction. Throughout a child's life the focus of interventions may change from airway management to speech, hearing, and language optimization, and finally to decannulation and procedures aimed at social integration and self-esteem. Otolaryngologists play an important role is this arena and provide high-quality care while continuing to expand what can be done for our patients.

The Most Valuable Resource Is Time: Insights From a Novel National Program to Improve Retention of Physician-Scientists With Caregiving Responsibilities.

PURPOSE: To enhance understanding of challenges related to work-life integration in academic medicine and to inform the ongoing implementation of an existing program and the development of other interventions to promote success of physician-scientists. METHOD: This study is part of a prospective analysis of the effects of the Fund to Retain Clinical Scientists (FRCS), a national program launched by the Doris Duke Charitable Foundation at 10 U.S. institutions, which provides financial support to physician-scientists facing caregiving challenges. In early 2018, 28 of 33 program awardees participated in semistructured interviews. Questions were about challenges faced by physician-scientists as caregivers and their early perceptions of the FRCS. Multiple analysts reviewed deidentified transcripts, iteratively revised the coding scheme, and interpreted the data using qualitative thematic analysis. RESULTS: Participants' rich descriptions illuminated 5 interconnected themes: (1) Time is a critical and limited resource, (2) timing is key, (3) limited time resources and timing conflicts may have a particularly adverse effect on women's careers, (4) flexible funds enable reclamation and repurposing of time resources, and (5) FRCS leaders should be cognizant of time and timing conflicts when developing program-related offerings. CONCLUSIONS: Programs such as the FRCS are instrumental in supporting individuals to delegate time-consuming tasks and to control how they spend their valuable time. Qualitative analysis suggests that access to and command of valuable time resources are crucial to career advancement, research productivity, and work-life flexibility, especially during critical time points along the physician-scientist trajectory.

Evaluation of prenatal diabetes mellitus and other risk factors for craniofacial microsomia.

OBJECTIVES: 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.