The Association of Speech/Language Risk With Phonological Awareness, Rapid Naming, and Reading Ability in Children With Cleft Lip and/or Palate.

Children with cleft lip and/or palate were assessed for speech, language, phonological awareness (PA), rapid naming (RN) and reading ability using standardized instruments at baseline (T1; N = 142, Mage = 6.14 years, 51% males) and 2-year follow-up (T2; 89% retention, Mage = 8.38). Children with no speech or language risk scored higher for T1 and T2 PA, RN, and reading than children with both speech and language risk [Adjusted Mean Difference (AMD) ranged from 11.79 to 21.25]; language risk (AMD 8.37 to 13.58); and speech risk (0.51 to 6.87). No significant differences by cleft type or child sex.

Developmental Risk for Infants with Cleft Lip with or Without Cleft Palate Based on Caregiver-Proxy Reports.

OBJECTIVES: (1) Assess caregiver-reported development in infants born with cleft lip ± alveolus (CL ± A) and cleft lip and palate (CLP); (2) determine factors associated with increased developmental risk; and (3) determine consistency of developmental risk before and after surgery for cleft lip. DESIGN: Prospective, longitudinal assessment of development. Time (T) 1, prior to lip closure; T2, 2 months post lip closure. SETTING: Three US craniofacial teams and online parent support groups. PARTICIPANTS: 123 total caregivers (96% mothers); 100 at T1, 92 at T2, and 69 at both T1 and T2. MEASURE: Ages and Stages Questionnaire-3 (ASQ-3): Communication, Gross Motor, Fine Motor, Problem Solving, Personal Social Domains. RESULTS: At T1 47%; at T2 42% passed all 5 Domains; 36% of infants pass all 5 Domains at both T1 and T2. Infants with CLP were at greatest risk on Communication [B = 1.449 (CI = .149-20.079), p = .038; Odds Ratio (OR) = 4.3 (CI = .923-19.650)] and Gross Motor Domains [B = 1.753 (CI = .316-20.605), p = .034; OR = 5.8 (CI = 1.162-28.671)]. Male infants were at greatest risk on Fine Motor [B = 1.542 (CI = .495-20.005), p = .009; OR = 4.7 (CI = 1.278-17.101)] and Problem Solving Domains [B = 1.200 (CI = .118-19.708), p = .044; OR = 3.3 (CI = .896-12.285)]. CONCLUSIONS: Based on caregiver report, infants with CL ± A and CLP meet referral criteria at a high rate. Infants with CLP and male infants were at greatest risk. Regular developmental screening is recommended.

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 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.

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.

Parent Observations of the Health Status of Infants With Clefts of the Lip: Results From Qualitative Interviews.

OBJECTIVE: To explore, using semistructured qualitative interviews, parent observations of their infant's health as they relate to having a cleft lip or cleft lip and cleft palate (CL±P) and/or associated treatments. DESIGN: Cross-sectional, qualitative study across 3 sites. SETTING: Parents were recruited from 3 academic craniofacial centers. Most interviews were conducted over the telephone, audio-recorded, and transcribed. PARTICIPANTS: Forty-one parents (31 English-, 10 Spanish-speaking) were interviewed. Parents had a child ages 1 to 35 months diagnosed with CL±P. MAIN OUTCOME MEASURE(S): Twelve domains reflecting infant health and well-being, likely affected by a CL±P and/or associated treatments, were identified from clinical experience and literature review. Study investigators conducted semistructured interviews based on 12 identified domains. After transcripts of the interviews were reviewed, our multidisciplinary team selected illustrative quotes from each domain that reflected consistent observations made by parents. RESULTS: Parents' responses covered all 12 domains. Specifically, parents discussed the domain of feeding most frequently, followed by observations about sleep, development, comfort, breathing, and vocalization. CONCLUSIONS: We found that parents of infants with clefts provided caregiver-centered language useful in describing their daily observations. No additional domains of infant health were noted by parents. We will use these observations and language to develop and validate a parent-reported observation diary that can augment assessments of the impacts of interventions on infants who are receiving care for CL±P.