Faculty Bibliography

Background/Purpose: Quality of life (QoL) is an integral parameter in understanding, assessing, and improving children's well-being across health conditions. Assessing oral health-related quality of life (OHRQoL) is crucial to evaluating patient well-being and measuring treatment efficacy in those with oral-facial conditions. The Child Oral Health Impact Profile (COHIP) is a validated instrument created to measure the OHRQoL of school-aged children (ages 8-18), yet no measure of OHRQoL exists for preschool children. The purpose of this study was to develop and validate a preschool version of the COHIP (COHIP-PS) for children aged 2 to 5. Methods/Description: A standard multistage process consisting of item selection, face validity testing, item impact testing, reliability and validity testing, and confirmatory factor analysis was used to develop the COHIP-PS. A cross-sectional convenience sample of caregivers with children 2 to 5 years old from 4 groups at varying locations completed the item clarity and impact forms. Groups were recruited from pediatric health clinics or preschools/day care centers, speech clinics, dental clinics, or cleft/craniofacial centers. Participants' children had a variety of oral health-related conditions, including caries, cleft lip and/or palate, or speech/language disorders.
Result(s): A total of 327 caregivers were included in the sample. Participants included caregivers from the cleft group (n = 81), speech group (n = 69), community group (n = 75), and dental group (n = 102). Approximately 84% of the overall caregiver sample were female, whereas 48% of the children were female. For children, 43% were white, followed by 33% Latino, 11% black, 7% Pacific Islander, 4.7% Other, and 1.5% Indian. For the cleft sample, children had cleft lip and palate (62%), cleft palate only (29%), or cleft lip only (9%). The COHIP-PS was found to have acceptable internal validity (= 0.71) and high test-retest reliability (0.87). Although discriminant validity results indicated significant differences across study groups, the overall magnitude of differences was modest. Results from confirmatory factor analyses support the use of a 4-factor model consisting of 12 items across oral health, functional well-being, socialemotional well-being, and self-image domains.
Conclusion(s): QoL is integral factor in understanding and assessing children's well-being. The COHIP-PS is a validated OHRQoL measure for preschool children with cleft or other oral conditions

Background/Purpose: Quality of life (QoL) data provide an assessment of treatment outcomes complementary to standard clinical measures. Yet, it is critical to consider time restraints when measuring QoL, especially for patients requiring multidisciplinary assessments. QoL instrument short forms are a preferred alternative to lengthy measures if they provide comparable, valid results. This study examined longitudinal oral health-related quality of life (OHRQoL) data among participants followed for cleft care using the long and short form of the Child Oral Health Impact Profile (COHIP). The objectives include: (1) reporting OHRQoL data over time; and (2) comparing the results from the validated, original long form of the COHIP to the validated COHIP-SF (short form). Methods/Description: Data were systematically and prospectively collected by 6 cleft teams representing geographically and ethnically diverse patients and their caregivers (CGs). CGs and their children independently completed the COHIP in conjunction with their team visits. The long form of the validated COHIP includes 34 items across 5 domains; the validated COHIP-SF includes 19 items from the original COHIP across 3 domains (Oral Health, Functional Well-being, and Socioemotional Well-being). The sample is composed of 1196 youth who averaged 12 years old at baseline. Team recommendations for surgery included: secondary palate or fistula, bone graft, lip and/or nose revisions, and orthognathic surgery. Forty-three percent (N= 514) had surgery over the course of the 6-year grant period. Individuals were followed at baseline, 1 and 2 years postsurgery, or 1 and 2 years postbaseline for those who did not have surgery.
Result(s): Children who had cleft-related surgery and their CGs had significantly lower self- and CG-rated COHIP scores (P < .01) at baseline and follow-up compared to children who did not have surgery and their CGs. The COHIP-SF provides comparable OHRQoL scores to the original COHIP for children with cleft (Pearson correlation = 0.96) and their CGs (Pearson correlation = 0.97) at each data collection point. Analyses also demonstrate that the original COHIP and COHIP-SF scores significantly improved (P < .01) among the surgical patients and their caregivers over time. In sum, the correlation between the short and long forms is uniformly high; the short form takes ~5 minutes to complete.
Conclusion(s): The results indicate that youth with a surgical recommendation and their CGs reported lower COHIP scores at baseline and postsurgery than individuals without a surgical recommendation. Significant improvement in OHRQoL was observed over time among those who had surgery. The COHIP-SF provides comparable OHRQoL scores to the original COHIP for children with cleft and their CGs

OBJECTIVE:To explore factors related to completion of surgery recommendations among children with cleft lip and palate (CLP) or cleft palate only (CPO). DESIGN/METHODS:Multicenter prospective longitudinal cohort study (2009-2015). SETTING/METHODS:Six cleft centers in the United States. PATIENTS/PARTICIPANTS/METHODS:A diverse sample of 1186 youth aged 7.5 to 18.5 years with CLP or CPO and a caregiver. RESULTS:Data were collected from child-caregiver dyads at baseline and up to 3 follow-up visits. Of the 765 surgeries recommended during the study period, 83 were postponed and 597 were completed; this represents a completion rate of 78%. There were multiple reasons identified by patients for why the remaining 85 recommended surgeries were not completed. Children with nonprivate insurance were more likely to postpone recommended surgeries. Hispanic/Latino and Asian children had fewer surgeries compared to whites/Caucasians. Greater severity in rated speech differences was associated with more surgeries. Among participants who had completed all recommended surgeries, providers rated over 86% as having normal or mildly different facial appearance and 83% as having a normal or mild speech intelligibility rating. Similarly, caregivers rated 80% of facial appearance and 78% of speech positively. Approximately 25% of children had an additional surgical recommendation at the end of the study. CONCLUSIONS:The majority of surgical recommendations were completed with positive outcomes in facial appearance and speech intelligibility by provider and caregiver ratings. Several factors contributed to surgical recommendations not being completed and the results underscore the importance of examining socioeconomic and demographic disparities in surgical care.

Quality of life is a valid patient-reported parameter that provides an assessment of treatment need or outcomes complementary to standard clinical measures. Such patient-reported assessments are particularly salient when examining chronic conditions with prolonged treatment trajectories, such as cleft lip and palate. This critical review identifies key questions related to ongoing research on the oral health-related quality of life (OHRQoL) in children with cleft and caregiver well-being. Details of the design and results from 2 longitudinal multicenter studies are presented. This article also provides an update on recent published reports regarding OHRQoL in individuals with cleft. Methodological issues in OHRQoL research are discussed, including condition-specific versus generic instruments, incorporating positive items in OHRQoL instruments, calculating minimally important differences in OHRQoL, implementing mixed methods design, and utilizing validated short assessment forms in OHRQoL research. Finally, new directions for research in cleft as a chronic condition are identified and discussed.

OBJECTIVE: The Child Oral Health Impact Profile (COHIP) is a validated instrument created to measure the oral health-related quality of life of school-aged children. The purpose of this study was to develop and validate a preschool version of the COHIP (COHIP-PS) for children aged 2-5. BASIC RESEARCH DESIGN: The COHIP-PS was developed and validated using a multi-stage process consisting of item selection, face validity testing, item impact testing, reliability and validity testing, and factor analysis. PARTICIPANTS: A cross-sectional convenience sample of caregivers having children 2-5 years old from four groups completed item clarity and impact forms. Groups were recruited from pediatric health clinics or preschools/daycare centers, speech clinics, dental clinics, or cleft/craniofacial centers. Participants had a variety of oral health-related conditions, including caries, congenital orofacial anomalies, and speech/language deficiencies such as articulation and language disorders. MAIN OUTCOME MEASURE: COHIP-PS. RESULTS: The COHIP-PS was found to have acceptable internal validity (a = 0.71) and high test-retest reliability (0.87), though internal validity was below the accepted threshold for the community sample. While discriminant validity results indicated significant differences across study groups, the overall magnitude of differences was modest. Results from confirmatory factor analyses support the use of a four-factor model consisting of 11 items across oral health, functional well-being, social-emotional well-being, and self-image domains. CONCLUSIONS: Quality of life is an integral factor in understanding and assessing children's well-being. The COHIP-PS is a validated oral health-related quality of life measure for preschool children with cleft or other oral conditions.

OBJECTIVES: Conducting longitudinal, multicentre, multidisciplinary research for individuals with chronic conditions can be challenging. Despite careful planning, investigative teams must adapt to foreseen and unforeseen problems. Our objective is to identify challenges encountered and solutions sought in a recently completed observational, longitudinal study of youth with cleft lip and palate as well as their caregivers. METHODS: Data for analysis were derived from a 6-year, multicentre, prospective, longitudinal study of youth with cleft conducted from 2009 to 2015 that examined oral health-related quality of life and other related clinical observations over time in youth who had cleft-related surgery compared to those who did not. Youth and their caregivers participating in this study were followed at one of six geographically diverse, multidisciplinary cleft treatment centres in the USA. RESULTS: Establishing effective communication, ensuring protocol adherence, safeguarding data quality, recognizing and managing differences across sites, maximizing participant retention, dealing with study personnel turnover, and balancing/addressing clinical and research tasks were particularly exigent issues that arose over the course of the study. Attending to process, ongoing communication within and across sites, and investigator and clinician commitment and flexibility were required to achieve the stated aims of the research. CONCLUSION: Studying children with cleft and their caregivers over time created both foreseen and unforeseen challenges. Solutions to these challenges are presented to aid in the design of future longitudinal research in individuals with chronic conditions.

PURPOSE: This paper evaluated the impact of cleft-related surgery on the oral health-related quality of life (OHRQoL) of youth with cleft over time. METHODS: Data were derived from a 5-year, multi-center, prospective, longitudinal study of 1196 youth with cleft lip and/or palate and their caregivers. Eligible youth were between 7.5 and 18.5 years old, spoke English or Spanish, and were non-syndromic. During each observational period, which included baseline, and 1- and 2-year post-baseline follow-up visits, youths and their caregivers completed the Child Oral Health Impact Profile, a validated measure of OHRQoL. Multilevel mixed-effects models were used to analyze the effects of receipt of craniofacial surgery on OHRQoL over time. RESULTS: During the course of this study a total of 516 patients (43 %) received at least one surgery. Youth in the surgery recommendation group had lower self- (beta = -2.18, p < 0.05) and proxy-rated (beta = -2.92, p < 0.02) OHRQoL when compared to non-surgical self- and proxy-rated OHRQoL at baseline. Both surgical and non-surgical youth (beta = 3.73, p < 0.001) and caregiver (beta = 1.91, p < 0.05) ratings of OHRQoL improved over time. There was significant incremental improvement (time x surgery interaction) in self-reported OHRQoL for youth postsurgery (beta = 1.04, p < 0.05), but this postsurgery increment was not seen in the caregiver proxy ratings. CONCLUSIONS: Surgical intervention impacts OHRQoL among youth with cleft. Youth who were surgical candidates had lower baseline self- and caregiver-rated OHRQoL when compared to non-surgical youth. Youth who underwent cleft-related surgery had significant incremental improvements in self-rated but not caregiver (proxy)-rated OHRQoL after surgery.

BACKGROUND: The Child Oral Health Impact Profile (COHIP) is an instrument designed to measure the self-reported oral health-related quality of life of children between the ages of 8 and 15, including domains for oral health, functional well-being, social-emotional well-being, school environment and self-image. The purpose of this study was to estimate the minimally important difference (MID) of the COHIP for patients with cleft lip/palate. METHODS: Data from a 6-year, prospective, longitudinal cohort study of children with cleft lip/palate were analyzed to estimate the MID. Analysis was restricted to patients with data at baseline and first follow-up and not receiving a surgical intervention in the intervening years (N = 281). MIDs were estimated via the anchor-based method, using the Global Assessment of Change, and the effect size distribution method. RESULTS: Based on the distributional method, the minimally important differences were 0.16 (oral health), 0.12 (functional), 0.22 (social-emotional), 0.21 (school environment) and 0.19 (self-image). MID anchor estimates for COHIP domains ranged from -0.32 to 0.84. The anchor-based and effect size MID estimates for the overall COHIP score were 2.95 and 0.25, respectively. CONCLUSION: The minimally important difference of the Child Oral Health Impact Profile is recommended for interpreting clinically meaningful change in patients with cleft lip/palate.

OBJECTIVES: Restorative interventions for cleft lip and palate involve annual evaluations, adjunct treatment, and multiple surgeries. The purpose of this study was to investigate the longitudinal impact of cleft surgery on psychosocial functioning among youth with cleft. METHODS: Data were derived from a 5-year, multicenter, prospective longitudinal study of children with cleft (N = 1196). Children completed psychological inventories for self-concept, anxiety, depression, mastery, and relatedness. Multilevel mixed-effects models were used to analyze the effects of craniofacial surgery for cleft on psychosocial outcomes over time. RESULTS: There were 1196 participants at baseline, of whom 258 (27.5%) received a surgical intervention prior to their 1st follow-up visit. Approximately 78% of participants had cleft lip and palate, and 22% had cleft palate only. Surgery receipt was significantly associated with lower relatedness (beta = -1.48, 95% CI = -2.91, -0.05) and mastery (beta = -1.32, 95% CI = -2.49, -0.15) scores, although overall scores appeared to increase over time. Surgery was not related to anxiety (beta = -0.15, 95% CI = -1.08, 0.79), depression (beta = 0.18, 95% CI = -0.65, 1.01), and self-concept (beta = -0.84, 95% CI = -1.83, 0.15). The treatment-time interaction was not significant. Significant differences in psychosocial functioning were found across sex, race/ethnicity, and age groups. CONCLUSIONS: Surgery may have negative short-term impacts on psychosocial functioning, although effects may diminish over time. Given the limited postsurgical follow-up period, long-term change in psychological well-being and the moderating effects of surgery may not be fully realized. Further follow-up of children with cleft through adulthood to explore developmental trajectories of psychosocial functioning in more detail is recommended.

Background/Purpose: This study sought to understand caregivers' (CGs') responses to early cleft lip/palate care for their infants receiving traditional cleft surgery or Nasoalveolar Molding (NAM) and traditional cleft surgery. Given the controversy around caregiver burden, the research questions included: 1) Are there treatment group differences in CGs' psychosocial adjustment over time?, 2) How do CGs cope with their infant's cleft and cleft treatment during the first year of life?, and 3) How do resources and demands affect family adaptation among CGs of infants with cleft over time? Methods/Descriptions: A prospective, mixed methods multicenter longitudinal study was conducted among CGs (N=118) seeking treatment for their infants' cleft lip and palate at one of six cleft treatment centers in the U.S. Participants were in one of two treatment groups: traditional cleft surgery only or NAM plus traditional cleft surgery. The CGs completed semi-structured interviews and standardized questionnaires (Parent Stress Index, Family Environment Scale, Coping Health Inventory for Parents, Generalized Anxiety Disorder scale, and the Patient Health Questionnaire) assessing psychosocial well-being and family impact at three time points: the beginning of treatment (~1 month of age), pre-lip surgery (~3-5 months of age), and post-palate surgery (~12-13 months of age). Multi-level modeling was used to longitudinally assess CGs' psychosocial outcomes. Results: While the first year was demanding for all CGs, NAM onset and the child's lip surgery were particularly stressful times. CGs used optimism, problem-solving behavior, and social support to cope with this stress. Qualitatively, CGs' ability to balance cleft treatment demands with their psychosocial resources and coping strategies influenced family adaptation. Questionnaire results indicated that the NAM treatment group experienced more rapid declines in the likelihood of anxiety and depressive symptoms between the first and second visits coupled with more rapid declines in anxiety and depressive symptoms over time when compared to those in the traditional cleft surgery group (p<0.05). The groups did not differ in stress levels at baseline, and both groups' stress decreased over time, though the NAM group reported lower levels of stress over time (p<0.05). The NAM treatment group reported significantly improved coping at pre-lip surgery and at post palate visits compared to CGs in the traditional treatment group. Conclusions: CGs of NAM-treated infants experienced more positive psychosocial outcomes than CGs whose infants had traditional cleft surgery. Results from the mixed methods support the Family Adjustment and Adaptation Response Model as used in pediatric chronic condition research