Body image and psychosocial outcomes in youth and young adults with differences of sex development: a multi-method study.

OBJECTIVE: Differences of sex development (DSD) can affect the physical health, appearance, and psychosocial functioning of affected individuals, but little is known about how subjective appearance perceptions (body image) impact psychosocial outcomes. This study evaluated body image and its associations with psychosocial outcomes including quality of life, resilience, and psychosocial adjustment. METHODS: This cross-sectional, multi-method study assessed body image and psychosocial outcomes including quality of life, adjustment, and resilience in 97 youth and young adults with DSD (mean age = 17 ± 3.7 years; 56% assigned female in infancy) using psychometrically sound instruments. A subsample (n = 40) completed qualitative interviews. RESULTS: Quantitative results indicated that overall, participants were satisfied with their physical appearance, although less so with their primary sex characteristics. Body image dissatisfaction was associated with poorer psychosocial adjustment, quality of life, and resilience. Qualitatively, youth and young adults reported a variety of perceptions, both positive and negative, related to their body image and the impact of living with a DSD condition. Themes identified included appearance management; effects of DSD on body image; diagnostic factors and features; attitudes about diagnosis; and treatment. CONCLUSIONS: Body image is significantly associated with psychosocial outcomes in youth and young adults with DSD, with qualitative findings highlighting both positive and negative body image experiences. Results have implications for clinical care including screening for appearance concerns, normalization of appearance variations, and intervention development to better support healthy body image and psychosocial functioning in youth and young adults with DSD.

Implications of Cardiac Scarring on Body Image Disturbance Among Young Adults With Congenital Heart Defects.

BACKGROUND: Many individuals with congenital heart disease (CHD) undergo open heart surgery, resulting in prominent scarring. However, little research has assessed the impact of surgical scarring on body image in this population. OBJECTIVE: Within this cross-sectional study, associations between body image concerns, anxiety and depression symptoms, and health-related quality of life among individuals with CHD were evaluated. METHODS: Young adults with CHD (N = 138) completed the Body Image Disturbance Questionnaire along with other patient-reported outcome measures. RESULTS: Scores on the Body Image Disturbance Questionnaire were lower (mean, 1.3 ± 0.5) than populations with diabetes amputations, craniofacial conditions, idiopathic scoliosis, and clinical controls. Higher Body Image Disturbance Questionnaire scores were reported among women (P = .003) and were associated with greater anxiety (P = .000) and depression (P = .13) after controlling for sex. CONCLUSIONS: Body image concerns related to cardiac surgery scarring are especially prominent in women and associated with greater emotional distress, suggesting potential clinical relevance for evaluating body image concerns in persons with CHD.

Prospective Evaluation of Health-Related Quality-of-Life in Children with Craniosynostosis.

OBJECTIVE: To investigate health-related quality of life (HRQL) in children aged 2 to 7 years, who have undergone surgery for craniosynostosis. DESIGN: Cross-sectional survey. SETTING: A tertiary pediatric academic medical center. PARTICIPANTS: Children with craniosynostosis who underwent surgical correction, and who were 2-7 years old at the time of the study. Children from families that did not speak English were excluded. INTERVENTIONS: Caregivers were asked to fill out the Pediatric Quality of Life Inventory (PedsQL) Core Parent Report and the PedsQL Cognitive Functioning Scale. MAIN OUTCOME MEASURES: PedsQL: Psychosocial Health Summary Score, Physical Health Summary Score, Total Core Score, Cognitive Functioning Scale Score. Scores range from 0 to 100, with higher scores reflecting greater QoLSubject factors: comorbidities, syndromic status, type of craniosynostosis, type of surgery. RESULTS: The study included 53 subjects, of whom 13.2% had a syndrome. Core and cognitive scores did not depend on presence of a syndrome or suture involved. Subjects who underwent posterior cranial distraction achieved higher Total Core Scores than subjects who underwent open vault remodeling. Among subjects with sagittal craniosynostosis, there was a tendency for higher scores among children who underwent minimally-invasive surgery compared to those who underwent open vault remodeling. CONCLUSIONS: This study demonstrates similar HRQL among children with and without a syndrome, higher HRQL among children undergoing posterior cranial distraction than those undergoing open vault remodeling, and trends towards higher HRQL in children with sagittal craniosynostosis who underwent minimally-invasive surgery compared to those who underwent open vault remodeling.

Screening for Academic Risk Among Students With Cleft Lip and/or Palate: Patterns of Risk and Qualities of Effective Tools.

This study evaluated the effectiveness of academic screening measures in relation to parent-reported diagnoses.Multicenter, retrospective cohort study including structured interviews, questionnaires, and chart reviews.Six North American cleft centers.Children (n = 391) with cleft lip and/or palate, ages 8 to 10 years of age (192 male) and their guardians were recruited during regular clinic visits.Parent and child ratings on the Pediatric Quality of Life Inventory (PedsQL) School Scale, child report on CleftQ School Scale, parent report on the Adaptive Behavior Assessment System-Third Edition Functional Academics (ABAS-FA) Scale and Child Behavior Checklist (CBCL) School Competency Scale, parent interview, and medical chart review.Risk for concerns ranged from 12% to 41%, with higher risk reflected on the CBCL-SC compared to other measures. Males with cleft palate were consistently at the highest risk. Only 9% of the sample had a parent-reported diagnosis of a learning or language disability. Ratings from the ABAS-FA and CBCL-SC had the highest utility in identifying those with language and/or learning concerns.As cleft teams work to develop standardized batteries for screening and monitoring of patients, it is important to evaluate the effectiveness of measures in identifying those at highest risk. When screening for language and learning disorders, questions related to potential academic struggles, such as increased school effort or increased school distress, are most useful. Referrals for follow-up evaluation are recommended for those identified at high risk.

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.

Neuropsychological and mental health concerns in a multicenter clinical sample of youth with turner syndrome.

Clinical practice guidelines for individuals with Turner syndrome (TS) recommend screening for neuropsychological concerns (NC) and mental health concerns (MHC). However, current provider screening and referral patterns for NC and MHC are not well characterized. Additionally, prevalence of and risk factors for NC and MHC vary across studies. This multicenter chart review study examined the prevalence, risk factors for, and management of NC and MHC in a cohort of 631 patients with TS from three pediatric academic medical centers. NC and/or MHC were documented for 48.2% of patients. Neuropsychological evaluation recommendations were documented for 33.9% of patients; 65.4% of the sample subsequently completed these evaluations. Mental health care recommendations were documented in 35.0% of records; subsequent documentation indicated that 69.7% of these patients received such services. Most notably, rates of documented MHC, NC, and related referrals differed significantly by site, suggesting the need for standardized screening and referral practices. TS diagnosis in early childhood was associated with an increased risk of NC. Spontaneous menarche was associated with increased risk of MHC. Younger age at growth hormone initiation was associated with both increased risk of isolated NC and co-occurring NC and MHC. Mosaic karyotype was associated with decreased risk of NC and MHC.

Psychosocial and Health-Related Experiences of Individuals With Microtia and Craniofacial Microsomia and Their Families: Narrative Review Over 2 Decades.

This paper describes 20 years of microtia and craniofacial microsomia (CFM) psychosocial and healthcare studies and suggests directions for clinical care and research.A narrative review of papers January 2000 to July 2021 related to psychosocial and healthcare experiences of individuals with microtia and CFM and their families.Studies (N = 64) were mainly cross-sectional (69%), included a range of standardized measures (64%), and were with European (31%), American (27%), or multinational (23%) samples. Data were generally collected from both patients and caregivers (38%) or patient self-report (35%). Sample sizes were 11 to 25 (21%), 26 to 50 (19%), 51 to 100 (22%), or over 100 (38%). Studies addressed 5 primary topics: (1) Healthcare Experiences, including Medical Care, Hearing Loss/Amplification, Diagnostic Experiences, and Information Preferences; (2) Psychosocial Experiences, including Teasing, Behavioral Adjustment, Psychosocial Support, and Public Perception; (3) Neurocognitive Functioning and Academic Assistance; (4) Pre- and Post-Operative Psychosocial Outcomes of Ear Reconstruction/Canaloplasty; and (5) Quality of Life and Patient Satisfaction.Care involved multiple specialties and was often experienced as stressful starting at diagnosis. Psychosocial and neurocognitive functioning were generally in the average range, with possible risk for social and language concerns. Coping and resiliency were described into adulthood. Satisfaction and positive benefit of ear reconstruction/canaloplasty were high. Care recommendations include increasing: hearing amplification use, microtia and CFM knowledge among providers, efficient treatment coordination, psychosocial support, academic assistance, and advances to minimize surgical scarring. This broad literature overview informs clinical practice and research to improve psychosocial outcomes.

A Multisite Study Investigating Child and Parent Proxy Reported Quality of Life in Children With Cleft Lip and/or Palate.

This observational, multisite cohort study explored health-related quality of life (HRQoL) in children with cleft lip and/or palate (CL/P), including interrater agreement and ratings for this group relative to clinical cutoff scores and published means for healthy and chronically ill children.Participants (338 children ages 8-10 years, 45.9% male and their parents, 82.0% female) across 6 sites completed the Pediatric Quality of Life Inventory Generic Core Scales (PedsQL).Intraclass correlation revealed poor interrater agreement for most HRQoL domains. Although ratings were generally higher than those expected for children with a chronic illness, child ratings were below healthy means for school functioning, and parent proxy ratings were below healthy means for all domains except physical functioning. Lower ratings consistent with chronic illness means were found for self-reported emotional and psychosocial functioning in children with cleft lip and palate (CLP), as well as parent proxy-reported emotional, school, and psychosocial functioning for children with cleft palate (CP). Scores were most likely to be in the clinical range for children with CP for social, school, and total functioning.Although parent proxy report provides important information about observed functioning, poor interrater agreement indicates that both child and parent proxy reported HRQoL should be included in outcomes assessment for CL/P. HRQoL ratings may be higher for children with CL/P compared to youth with other chronic illnesses, but psychosocial functioning may be negatively impacted when compared with healthy youth, particularly for emotional, social, and school functioning in children with CLP or CP.

Psychosocial Outcomes in Children with Cleft Lip and/or Palate: Associations of Demographic, Cleft Morphologic, and Treatment-Related Variables.

OBJECTIVE: To determine associations of demographic, morphologic, and treatment protocol parameters with quality of life (QoL), appearance/speech satisfaction, and psychological adjustment. DESIGN: Observational study utilizing retrospective report of protocol variables and current outcome variables. SETTING: Six North American cleft treatment clinics. PARTICIPANTS: Children, ages 8.0-10.99 years, with Cleft Lip ± Alveolus, Cleft Palate, Cleft Lip and Palate, and parents (N = 284). OUTCOME MEASURES: Pediatric QoL Inventory (PedsQL): Parent, Child, Family Impact Module (FIM); Patient Reported Outcome Measurement Information System (PROMIS); Child Behavior Checklist (CBCL); CLEFT-Q. RESULTS: Outcome scores were average with few differences by cleft type. Multiple regression analyses yielded significant associations (Ps < .05) between socioeconomic status, race, and age at assessment and parent- and self-reported measures. Females had higher PROMIS Depression (ß=.20) but lower CBCL Affective (ß = -.16) and PROMIS Stigma scores (ß= -.24). Incomplete cleft lip was associated with lower PROMIS Depression, and more positive ratings of CLEFT-Q: Nose, Nostril, Lip Scar; CBCL Competence scores, (ßs = -.17 to .17). Younger Age at Lip Closure was associated with higher CBCL School Competence (ß= -.18). Younger Age at Palate Closure was associated with higher Child PedsQL Total, Physical, Psychosocial QoL, and better CLEFT-Q Speech Function (ßs = -.18 to -.15). Furlow Palatoplasty was associated with more CBCL Externalizing Problems (ß = .17) higher CBCL Activities (ß = .16). For all diagnoses, fewer Total Cleft-Related Surgeries was associated with lower PROMIS Stigma and higher CBCL Total Competence and Activities (ßs = -.16 to .15). CONCLUSIONS: Demographic characteristics, lip morphology, and treatment variables are related to later psychological functioning.

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.

Implementation of a Post-Baccalaureate Program at a Pediatric Academic Medical Center: A Pathway to Graduate School.

Post-baccalaureate programs are increasingly common and have demonstrated success in preparing students for graduate programs. Most post-baccalaureate programs have a pre-medical focus, and the few existing clinical psychology-focused programs are centered on coursework, situated within a university, costly to students, and/or geared towards career change. The post-baccalaureate program at Nationwide Children's Hospital was created in 2017 and is uniquely positioned within a pediatric academic medical center. The program provides research skill training, mentorship, professional development, and graduate school preparation to employed clinical research coordinators. An early program assessment, based on self-reports, revealed the program attracts diverse participants that are primarily interested in pediatric or child psychology and rate themselves as "somewhat confident" in their current research skills. This manuscript summarizes the three phases of development and key components of the post-baccalaureate program, which can serve as a model to other medical centers interested in developing training programs for research staff, particularly underrepresented racial and ethnic minorities, and those from disadvantaged backgrounds.

Improving Psychosocial Risk Assessment and Service Provision for Craniofacial Team Patients: A Quality Improvement Project.

To improve psychosocial risk assessment and service provision for children with craniofacial conditions presenting for annual interdisciplinary team visits.Institute for Healthcare quality improvement model.U.S. pediatric academic medical center.Caregivers of children ages 0-17 years with craniofacial conditions presenting for 1692 team visits between August 2017 and July 2019.Key drivers included: (1) standardizing pre-visit triage processes; (2) administering the Psychosocial Assessment Tool-Craniofacial Version (PAT-CV); (3) utilizing PAT-CV scores in real time to add patients to psychosocial provider schedules; and (4) family education. Interventions included improving patient screening, increasing PAT-CV completion rate, altering clinic flow, providing patient and parent education about psychosocial services, and altering team member roles to fully integrate PAT-CV administration and scoring in the clinic.The primary outcome was the percentage of patients identified for psychosocial consultations via nurse triage, PAT-CV score, family or provider request who completed consultations. The secondary outcome was the percentage of patients completing needed psychosocial consultations based on elevated PAT-CV scores.Use of the PAT-CV resulted in an increase in the percentage of patients with elevated psychosocial risk who received a psychosocial consultation from 86.7% to 93.4%. The percentage of children receiving psychosocial consultation at their annual team visit due to elevated PAT-CV scores increased from 72% to 90%.Integrating a validated psychosocial risk screening instrument can improve risk identification and psychosocial consultation completion. A combination of risk screening approaches may be indicated to identify patients in need of psychosocial services.

Clinical phenotype and management of individuals with mosaic monosomy X with Y chromosome material stratified by genital phenotype.

Individuals mosaic for monosomy X and a cell line with Y chromosome material can have genitalia that appear phenotypical female, male, or ambiguous. Those with this karyotype and typical female genitalia are diagnosed with Turner syndrome; however, this definition specifically excludes those with genitalia other than typical female. There is limited information on whether medical and neurodevelopmental risks are similar among individuals with monosomy X and Y chromosome material across genital phenotypes. This multicenter retrospective study compared comorbidities and clinical management in individuals with monosomy X and Y material and male/ambiguous genitalia to those with typical female genitalia. Electronic medical records for all patients with monosomy X and Y material (n = 76) at two large U.S. pediatric centers were abstracted for predetermined data and outcomes. Logistic regression was used to compare the two phenotypic groups adjusting for site and duration of follow-up. The male/ambiguous genitalia group was just as likely to have congenital heart disease (RR 1.0, 95%CI [0.5-1.9]), autoimmune disease (RR 0.6 [0.2-1.3]), and neurodevelopmental disorders (RR 1.4 [0.8-1.2]) as those with female genitalia. Despite similar risks, they were less likely to receive screening and counseling. In conclusion, individuals with monosomy X and Y chromosome material have similar medical and neurodevelopmental risks relative to individuals with Turner syndrome regardless of genitalia, but there are notable differences in clinical management.

Psychosocial Functioning Among Children With and Without Differences of Sex Development.

OBJECTIVE: To assess health-related quality of life (HRQoL), psychosocial adjustment, and family functioning of children with differences of sex development (DSD) or cleft lip and/or palate (CL/P). METHODS: In this cross-sectional study, parents of children with DSD (n = 67), CL/P (n = 121), and a comparison group of unaffected youth (n = 126) completed standardized measures assessing family functioning and their children's HRQoL and psychosocial adjustment. Medical charts were abstracted for youth with either congenital condition. RESULTS: Children with DSD were rated as having significantly lower HRQoL and greater internalizing problems compared to youth with CL/P and unaffected youth. Children in the DSD group were also significantly more likely to fall into the clinical risk categories for total and internalizing problems relative to the CL/P and unaffected groups. Caregivers of children with DSD were significantly more likely to endorse items about child suicidality compared with caregivers in the CL/P and unaffected groups. No significant differences were found between groups for externalizing problems or the expressiveness domain of family functioning; parents of children with DSD reported significantly less family conflict relative to the other groups and greater cohesion relative to the unaffected group. Conclusions Youth with DSD appear to be at greater risk for psychosocial problems relative to children with CL/P and unaffected peers. Results underscore the need for integrated interdisciplinary care and ongoing psychosocial risk monitoring in youth with DSD.

Perceptions About Lactation Consultant Support, Breastfeeding Experiences and Postpartum Psychosocial Outcomes.

INTRODUCTION: Many women seek lactation consultant support in the postpartum period. Lactation consultant support in community or clinical settings is often assumed to extend breastfeeding duration, improve breastfeeding experiences, and be well-received. Few studies have assessed women's perceptions of the support they received, nor have perceptions been examined in relationship to breastfeeding outcomes and maternal well-being. Our objective was to characterize the lactation consultant support women received and examine how women's perceptions about the support related to their breastfeeding outcomes, anxiety and depressive symptoms, and parenting stress. METHODS: This observational, cross-sectional study examined receipt of postpartum lactation consultant support among 210 US women. Perceptions of lactation consultant support were examined in relation to breastfeeding outcomes, anxiety and depressive symptoms, and parenting stress to explore outcomes of negative versus positive lactation consultant support experiences, using linear and proportional hazards regression. RESULTS: While overall perceptions of lactation consultant support were positive for most recipients (71%, n = 98), 29% (n = 40) reported negative perceptions of lactation consultant support. Negative perceptions were associated with lower breastfeeding self-efficacy (ß = - 11.7, 95% CI - 17.3, - 6.0), a less successful breastfeeding experience (ß = - 19.5, CI - 27.8, - 11.3), greater general anxiety (ß = 6.5, CI 2.1, 10.9), and shorter total duration of milk production (HR = 0.39, 95% CI 0.18, 0.84). Perceptions were not associated with depressive symptoms or parenting stress. DISCUSSION: Findings highlight the importance of ensuring that postpartum breastfeeding support provided by lactation consultants is perceived as positive by women.

Feeding and Growth in Infants With Cleft Lip and/or Palate: Relationships With Maternal Distress.

OBJECTIVE: To examine relationships between early feeding and growth and maternal distress in infants with and without cleft lip and/or palate (CL/P). DESIGN: Cross-sectional. SETTING: Pediatric academic medical center in the Midwestern United States. PARTICIPANTS: Mothers of infants 1 to 12 weeks old with CL/P (n = 30) and without CL/P (control group, n = 30) were recruited at craniofacial clinic or pediatrician appointments. MAIN OUTCOME MEASURE(S): Maternal responses on the Feeding/Swallowing Impact Survey (FS-IS), Parenting Stress Index, Fourth Edition, Short Form (PSI-4-SF), and Edinburgh Postnatal Depression Scale. Infant feeding history and growth measurements were obtained. RESULTS: Having an infant with CL/P revealed greater impact on maternal health-related quality of life due to feeding problems (F = 4.83, P = .03). Mothers of infants with CL/P reported average range Total Stress scores on the PSI-4-SF, which were higher than controls (F = 4.12, P = .05). Edinburgh Postnatal Depression Scale scores did not differ between groups. Compared to controls, infants with cleft palate had lower percentiles for weight (t = 4.13, P = .04) and length (t = 2.93, P = .01). Higher FS-IS scores were associated with longer feeding duration (r = 0.32, P = .01) and lower weight (r = -0.31, P = .02) and length (r = -0.32, P = .02). CONCLUSIONS: Despite receiving early team care and feeding interventions, mothers of infants with CL/P reported higher stress and more challenges with feeding and growth. Future studies should examine targeted psychosocial interventions to improve feeding and growth outcomes in infants with CL/P.

Social Experiences of Turkish Parents Raising a Child With Apert Syndrome: A Qualitative Study.

OBJECTIVE: Apert syndrome is a rare and understudied craniofacial condition with regard to its psychosocial impact on children and their parents. Due to the lack of studies focusing solely on the social experiences of children and families with Apert syndrome, it is difficult to develop interventions and strategies to support well-being and positive adjustment for this particular population. This study addressed this gap by assessing the unique social experiences of parents who are raising their children with Apert syndrome including difficulties they face and strategies they use to cope with challenges. DESIGN: Descriptive qualitative study using thematic analysis. PARTICIPANTS: Participants included 21 parents of 12 children (aged 1-12 years) with Apert syndrome (9 couples, 2 fathers, and 1 mother) who were recruited from a pediatric neurosurgery unit in Turkey. RESULTS: The qualitative analysis yielded four main themes describing the experiences of parents including (1) social challenges; (2) coping with negative reactions; (3) promoting socialization and independence; and (4) sources of strength. CONCLUSIONS: Parents reported several challenges, including social stigmatization, and utilized a range of strategies to support both their own and their child's positive adaptation and resiliency, including the use of religion and reliance on their spouses for support. Results offer clinically relevant insights about the strengths and challenges of families coping with Apert syndrome.

Retrospective Evaluation of Number of Surgeries and Parent Ratings of Academic and Behavioral Functioning Among Children With Isolated Oral Clefts.

OBJECTIVE: The purpose of this study was to evaluate possible relationships between number of surgeries and parent ratings of academic functioning among children with isolated oral clefts. DESIGN: Multicenter, retrospective cohort study including structured interviews, questionnaires, and chart reviews. SETTING: Completion of questionnaires occurred during clinical visits at 6 different cleft centers across North America. PARTICIPANTS: Parents of 285 children with isolated clefts of the lip and/or palate, aged 8 to 10 years old, participated in structured interviews and completed questionnaires regarding the academic and behavioral functioning of their children. MAIN OUTCOME MEASURES: Parent interview and medical chart review of number of surgeries to date and parent ratings on the Adaptive Behavior Assessment System, Third Edition-Functional Academics Scale (ABAS-FA) and Child Behavior Checklist (CBCL) Total Competency Scale. RESULTS: Parent ratings of ABAS-FA were at or above normative expectations, while ratings across CBCL Competency Scales were lower than normative expectations. Socioeconomic status (SES), age, and race were consistent predictors of parent ratings (higher SES, older age, and Caucasian race were associated with better functioning). Number of surgeries did not add significantly to academic ratings but did significantly contribute to ratings of social and activity participation. Patients with more surgeries were rated with lower functioning in these domains. CONCLUSIONS: Findings do not support a connection between number of surgeries and later ratings of academic functioning but do support a connection to social and activity involvement. Recommendations for conducting direct studies of the connection between surgeries and academic functioning as well as clinical considerations for surgeries and impact on social and activity involvement are discussed.

Breast Milk Feeding Practices and Barriers and Supports Experienced by Mother-Infant Dyads With Cleft Lip and/or Palate.

OBJECTIVE: To examine trends in breast milk provision and to characterize the breast milk feeding practices experienced by mother-infant dyads with cleft lip and/or palate (CL/P) in a large US sample. DESIGN: Cross-sectional study. METHODS: An online survey was distributed through cleft-related social media sites in the United States and in a single cleft lip and palate clinic. Statistical analyses included Kruskal-Wallis H tests and post hoc Mann-Whitney tests to examine group differences based on cleft type and prenatal versus postnatal cleft diagnosis. Linear regression was used to estimate associations between obtained variables. PARTICIPANTS: One hundred fifty biological mothers of infants (8-14 months of age) with CL/P (15% cleft lip, 29% cleft palate, 56% cleft lip and palate). RESULTS: Forty-six percent of mothers of infants with CL/P provided breast milk to their infant for at least 6 months. Five percent of infants ever fed at breast, and 43% received pumped breast milk via bottle. The most commonly reported supports included lactation consultants, nurses, feeding therapists, and online support groups. Feeding therapy was received by 48% of infants. CONCLUSIONS: A lower percentage of mothers of infants with CL/P reported providing breast milk compared to national estimates of the general population of infants without clefting. Results suggested there are multiple barriers, as well as numerous medical and psychosocial supports that facilitated breast milk feeding success. Implications for care are discussed.

Adherence to Orthodontic Treatment in Youth With Cleft Lip and/or Palate.

OBJECTIVE: To identify rates of nonadherence to orthodontic treatment among youth with cleft lip and/or palate (CL/P) and to compare demographic and clinical characteristics of patients categorized as adherent or nonadherent. DESIGN: Retrospective chart review. SETTING: Orthodontic treatment program affiliated with a US pediatric hospital-based craniofacial team. PARTICIPANTS: Medical charts of 54 patients with CL/P beginning phase I or phase II orthodontics between 2011 and 2014 (54% male; mean age: 11.7 ± 3.2 years) were reviewed. MAIN OUTCOME MEASURE(S): Data abstracted included demographic and clinical characteristics. Patients were classified as nonadherent based upon treatment termination due to nonadherence, treatment courses extending beyond 3 years, and information about missed appointments, poor oral hygiene, and broken appliances. RESULTS: In all, 24% had treatment terminated due to nonadherence and were significantly more likely to have behavioral health diagnoses (P = .01) or visits (P = .02) and social work consults (P = .01) than patients without termination. Thirty-seven percent had treatment courses beyond 3 years; youth with longer courses were significantly more likely to have cleft lip and palate versus cleft palate only or cleft lip (P = .03). Patients who missed more than 4 appointments were less likely to have behavioral health diagnoses (P < .01) compared to those with fewer missed appointments. Poor hygiene and broken appliance notations were common. Youth with poor hygiene notations were significantly older (P < .01) at treatment initiation than those without notations. CONCLUSIONS: Nonadherence was associated with diagnosis, age, and history of behavioral health or social work involvement. An understanding of adherence and relationships with clinical and demographic factors can inform clinical care and support intervention development to improve outcomes.