Surgical Care for School-Aged Youth With Cleft: Results From a Multicenter, Prospective Observational Study.

OBJECTIVE: To explore factors related to completion of surgery recommendations among children with cleft lip and palate (CLP) or cleft palate only (CPO). DESIGN: Multicenter prospective longitudinal cohort study (2009-2015). SETTING: Six cleft centers in the United States. PATIENTS/PARTICIPANTS: 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.

Challenges in conducting multicentre, multidisciplinary, longitudinal studies in children with chronic 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.

Oral health-related quality of life in youth receiving cleft-related surgery: self-report and proxy ratings.

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- (ß = -2.18, p < 0.05) and proxy-rated (ß = -2.92, p < 0.02) OHRQoL when compared to non-surgical self- and proxy-rated OHRQoL at baseline. Both surgical and non-surgical youth (ß = 3.73, p < 0.001) and caregiver (ß = 1.91, p < 0.05) ratings of OHRQoL improved over time. There was significant incremental improvement (time × surgery interaction) in self-reported OHRQoL for youth postsurgery (ß = 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.

Surgeon's and Caregivers' Appraisals of Primary Cleft Lip Treatment with and without Nasoalveolar Molding: A Prospective Multicenter Pilot Study.

BACKGROUND: Despite the increasing use of nasoalveolar molding in early cleft treatment, questions remain about its effectiveness. This study examines clinician and caregiver appraisals of primary cleft lip and nasal reconstruction with and without nasoalveolar molding in a nonrandomized, prospective, multicenter study. METHODS: Participants were 110 infants with cleft lip/palate (62 treated with and 48 treated without nasoalveolar molding) and their caregivers seeking treatment at one of six high-volume cleft centers. Using the Extent of Difference Scale, standard photographs for a randomized subset of 54 infants were rated before treatment and after surgery by an expert clinician blinded to treatment group. Standard blocked and cropped photographs included frontal, basal, left, and right views of the infants. Using the same scale, caregivers rated their infants' lip, nose, and facial appearance compared with the general population of infants without clefts before treatment and after surgery. Multilevel modeling was used to model change in ratings of infants' appearance before treatment and after surgery. RESULTS: The expert clinician ratings indicated that nasoalveolar molding-treated infants had more severe clefts before treatment, yet both groups were rated equally after surgery. Nasoalveolar molding caregivers reported better postsurgery outcomes compared with no-nasoalveolar molding caregivers (p < 0.05), particularly in relation to the appearance of the nose. CONCLUSIONS: Despite having a more severe cleft before treatment, infants who underwent nasoalveolar molding were found by clinician ratings to have results comparable to those who underwent lip repair alone. Infants who underwent nasoalveolar molding were perceived by caregivers to have better treatment outcomes than those who underwent lip repair without nasoalveolar molding. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, II.

Caregiver responses to early cleft palate care: A mixed method approach.

OBJECTIVE: This study sought to understand caregivers' (CGs') responses to early cleft lip/palate care for their infants. METHOD: A prospective, mixed methods multicenter longitudinal study was conducted among CGs (N = 118) seeking treatment for their infants' cleft lip and palate or cleft lip only at 1 of 6 cleft treatment centers in the United States. Participants were in 1 of 2 treatment groups: traditional care only or nasoalveolar molding (NAM) plus traditional care. The CGs completed semistructured interviews and standardized questionnaires assessing psychosocial well-being and family impact at 3 time points: the beginning of treatment (∼1 month of age), prelip surgery (∼3-5 months of age), and postpalate surgery (∼12-13 months of age). Multilevel modeling was used to longitudinally assess CGs' psychosocial outcomes. RESULTS: Although 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. Qualitative and quantitative results indicated CGs of NAM-treated infants experienced more rapid declines in anxiety and depressive symptoms and better coping skills over time than CGs whose infants had traditional care. CONCLUSION: CGs of NAM-treated infants experienced more positive psychosocial outcomes than CGs whose infants had traditional care. Results from the mixed model support the family adjustment and adaptation response model as used in pediatric chronic condition research.

Assessment of presurgical clefts and predicted surgical outcome in patients treated with and without nasoalveolar molding.

Obtaining an esthetic and functional primary surgical repair in patients with complete cleft lip and palate (CLP) can be challenging because of tissue deficiencies and alveolar ridge displacement. This study aimed to describe surgeons' assessments of presurgical deformity and predicted surgical outcomes in patients with complete unilateral and bilateral CLP (UCLP and BCLP, respectively) treated with and without nasoalveolar molding (NAM). Cleft surgeon members of the American Cleft Palate-Craniofacial Association completed online surveys to evaluate 20 presurgical photograph sets (frontal and basal views) of patients with UCLP (n = 10) and BCLP (n = 10) for severity of cleft deformity, quality of predicted surgical outcome, and likelihood of early surgical revision. Five patients in each group (UCLP and BCLP) received NAM, and 5 patients did not receive NAM. Surgeons were masked to patient group. Twenty-four percent (176/731) of surgeons with valid e-mail addresses responded to the survey. For patients with UCLP, surgeons reported that, for NAM-prepared patients, 53.3% had minimum severity clefts, 58.9% were anticipated to be among their best surgical outcomes, and 82.9% were unlikely to need revision surgery. For patients with BCLP, these percentages were 29.8%, 38.6%, and 59.9%, respectively. Comparing NAM-prepared with non-NAM-prepared patients showed statistically significant differences (P < 0.001), favoring NAM-prepared patients. This study suggests that cleft surgeons assess NAM-prepared patients as more likely to have less severe clefts, to be among the best of their surgical outcomes, and to be less likely to need revision surgery when compared with patients not prepared with NAM.

Parent-Reported Family Functioning Among Children With Cleft Lip/Palate.

OBJECTIVE: To examine family functioning related to sociodemographic and clinical characteristics in youth with cleft lip and/or palate (CL/P). DESIGN: Cross-sectional, multi-site investigation. SETTING: Six U.S. cleft centers. PATIENTS/PARTICIPANTS: A diverse sample of 1200 children with CL/P and their parents. MAIN OUTCOME MEASURE: Parents completed the Family Environment Scale (FES), which assesses three domains of family functioning: cohesion (or closeness), expressiveness (open expression of feelings), and conflict. Demographic and clinical characteristics were also assessed including race, ethnicity, type of insurance, and surgical recommendations. RESULTS: The FES scores for families seeking team evaluations for their youth with CL/P (mean age = 11.6 years) fall within the average range compared with normative samples. Families receiving surgical recommendations for their youth also had FES scores in the average range, yet families of children recommended for functional surgery reported greater cohesion, expressiveness, and less conflict compared with those recommended for aesthetic surgery (P < .05). For cohesion and expressiveness, significant main effects for race (P = .012, P < .0001, respectively) and ethnicity (P = .004, P < .0001, respectively) were found but not for their interaction. No significant differences were found on the conflict domain. Families with private insurance reported significantly greater cohesion (P < .001) and expressiveness (P < .001) than did families with public insurance. CONCLUSIONS: Family functioning across domains was in the average range. However, observed differences by race, ethnicity, type of insurance, and surgical recommendation may warrant consideration in clinical management for patients and families.

Examining factors associated with oral health-related quality of life for youth with cleft.

BACKGROUND: Patient-reported quality-of-life outcomes in cleft lip-cleft palate treatment are critical for evidence-based care. Scant data exist analyzing treatment from the patient's perspective. The authors examined the interrelationship among variables associated with oral health-related quality of life among youth with cleft. METHODS: As part of an ongoing longitudinal study, clinical evaluations and research questionnaire packets were completed before surgical recommendations were made (baseline). Participants completed the Child Oral Health Impact Profile, a validated oral health-related quality-of-life measure for children with cleft. During the baseline clinical evaluations, plastic surgeons determined whether surgical interventions were recommended within the year (expert determination represents a greater degree of current clinical need). General linear models incorporating surgical recommendation, gender, and age were fit for each subscale of and for the total Child Oral Health Impact Profile. Significant interaction terms were evaluated for their effect on the subscale. RESULTS: Baseline assessments were obtained from 1200 participants (mean, 11.8 years; 57 percent male). Participants with a surgical recommendation had lower quality of life on all but the self-esteem subscale compared with those without a surgical recommendation (p < 0.002). Two subscales had statistically significant age-sex interactions (p < 0.003), whereas another subscale had a statistically significant surgery by sex interaction term (p = 0.027). CONCLUSIONS: Overall, youth for whom surgery is currently recommended had lower oral health-related quality-of-life scores on the Child Oral Health Impact Profile Total scale than those with no surgical recommendation; older female subjects had lower quality-of-life scores than male subjects. CLINICAL QUESTION/LEVEL OF EVIDENCE: Risk, II.

Examination of a theoretical model for oral health-related quality of life among youths with cleft.

OBJECTIVES: We evaluated relationships among physical and psychological indicators and oral health-related quality of life (OHRQOL) in youths with cleft. METHODS: We recruited youths aged 7 to 18 years with cleft palate (n = 282) or cleft lip and palate (n = 918) and their caregivers into a 5-year observational investigation at 6 US treatment centers from 2009 to 2011. At baseline, youths completed surveys to assess psychological status. After clinical evaluation, 433 youths received a recommendation for surgery. We developed structural models from the baseline data incorporating age, gender, race, and cleft diagnosis to examine direct relationships between depressive symptoms and, in separate models, self-concept and sense of mastery and OHRQOL. Subsequent models examined the possible mediating effect of surgical recommendation. RESULTS: Depressive symptoms were negatively and self-concept and self-efficacy were positively associated with OHRQOL. Surgical recommendation appeared to mediate the effect of psychological well-being, but not depression, on OHRQOL, even after adjustment for cleft diagnosis. In some models, individual-level characteristics were significant. CONCLUSIONS: Path analyses support the theoretical model and underscore the importance of examining positive psychosocial characteristics such as resiliency and self-concept in this patient population.

Evaluation of the similarities and differences in response patterns to the Pediatric Quality of Life Inventory and the Child Oral Health Impact Scores among youth with cleft.

PURPOSE: To examine (a) the pattern of responses to a generic health-related quality of life (HRQL) measure (Pediatric Quality of Life Inventory--PedsQL) and an oral health-related quality of life (OHRQoL) measure (Child Oral Health Impact Profile--COHIP), and (b) the associations of these scores with surgical recommendation status among youth with cleft. METHODS: Cross-sectional data (baseline) regarding clinicians' surgical recommendations and quality of life (QoL) measures were examined from an ongoing observational study on treatment outcomes. Approximately one-third of the racially and geographically diverse sample (N = 1,200; mean = 11.6 years) received surgical recommendations to correct either visible (aesthetic) or invisible (functional) defects. Effect sizes were used to quantify differences in QoL based on surgical recommendation and to compare the sensitivity of the PedsQL and COHIP subscales. Using Pearson coefficients, the scores of those recommended for surgery were compared with those without a surgical recommendation. RESULTS: A moderate correlation (0.52) was found between the total scores on the PedsQL and COHIP (p < 0.0001). Subscale correlations between the QoL measures ranged from 0.19 to 0.48 with the strongest correlation between the PedsQL Emotional (r = 0.47) and COHIP Socioemotional Well-being subscale. The effect size for the COHIP Socioemotional Well-being (0.39) was larger than the PedsQL Social/Emotional (0.07/0.11) subscale (Z = 5.30/Z = 4.64, p < 0.0001, respectively), and the total COHIP (0.31) was significantly greater than the total PedsQL scale (0.15, z = 2.65, p = 0.008). CONCLUSIONS: A significant relationship was found between generic HRQL, OHRQoL, and surgical needs among youth with cleft with the COHIP having larger effect sizes than the PedsQL among surgical groups.

Nasoalveolar molding: prevalence of cleft centers offering NAM and who seeks it.

INTRODUCTION: Nasoalveolar molding (NAM) is a treatment option available for early cleft care. Despite the growing debate about the efficacy of nasoalveolar molding, questions remain regarding its prevalence and the demographic characteristics of families undergoing this technique prior to traditional cleft surgery. OBJECTIVES: To determine the number of teams currently offering nasoalveolar molding and to identify salient clinical and sociodemographic variables in infants and families who choose nasoalveolar molding compared with those who choose traditional cleft care across three well-established cleft centers. RESULTS: Via phone surveys, 89% of the U.S. cleft teams contacted revealed that nasoalveolar molding is available at 37% of these centers. Chart reviews and phone correspondence with caregivers indicate that the average distance to the cleft center was 65.5 miles and caregiver age averaged 30.9 ± 5.7 years. Of families who chose nasoalveolar molding, 85% received total or partial insurance coverage. No difference in caregiver education, income, or distance to the clinic between treatment groups was found. On average, infants receiving nasoalveolar molding and cleft surgery had larger clefts and had more clinic visits than infants receiving traditional cleft surgery. Infants who were firstborn and those who did not have other siblings were more likely to receive nasoalveolar molding than were infants who were residing with other siblings. CONCLUSIONS: Currently more than one-third of U.S. cleft centers offer nasoalveolar molding. Although the cleft size was larger in the nasoalveolar molding group, no treatment group differences in education, income, and distance to the clinic were found.

Oral health, related behaviors and oral health impacts among homeless adults.

OBJECTIVE: To assess the oral health needs, related behavior and oral health impact among homeless persons in Newark, New Jersey. METHODS: Participants represented a convenience sample of adults (n=46) participating in Homeless Services Day, an annual event sponsored by the Homeless Services Division of the Newark, NJ Department of Health and Human Services. Their mean age was 40.4 yr (SD=10.0), and 51.1% were female. The majority (76%) reported African-American ethnicity; remaining participants self-identified as Hispanic, White or Asian. Participants reported being homeless for a median of 11 months, with a range of 1 to 108 months. RESULTS: The 46 participants had 745 teeth (averaged 16.2 per person) that were either missing, had fillings or had untreated decay. Diseased teeth averaged 3.8 per person; missing teeth averaged 8.6 per person; and filled teeth averaged 3.7 per person. Only 28.3% had a dental visit in the past year. Approximately 87% reported negative oral health impacts impact: over half (55.6%) had current oral facial pain and two-thirds of our participants reported having dental-related face pain during the past year. Additional oral health impacts included: eating (42%), smiling (33%), concentrating (18%) and talking (16%). CONCLUSIONS: Consistent with other studies, this homeless sample presented with considerable oral health needs. Newark's homeless, like other homeless cohorts, face access to care and negative oral health impacts. This study informs the need for future research that can provide substantive evidence for care providers and policy makers.

Facial trauma: a recurrent disease? The potential role of disease prevention.

PURPOSE: The objectives of this investigation were to determine a profile of facial trauma patients presenting to the emergency department of University Hospital, University of Medicine and Dentistry of New Jersey, Newark, NJ, and to assess patient interest in violence or stress reduction programs. MATERIALS AND METHODS: A prospective study of the patients was conducted with the aid of a data collection form generated specifically for the purpose of this study. Data regarding patient age, race, gender, various aspects of social history, mechanism, and nature of injury were collected over a period of 1 year. In addition, all patients were asked to respond to 4 health promotion questions. All patients for whom the oral maxillofacial surgery service was consulted were included in the study. Descriptive statistical analysis of the data was used. RESULTS: A total of 92 patients were enrolled, of whom 80% were males. The mean age of patients enrolled was 30.5 years with the peak incidence of injury occurring in the 20- to 30-year-old age group (n = 30). The most frequent etiology was assault (75%), followed by motor vehicle accidents (18.5%). The most frequently occurring injury was mandible fracture (46.7%), followed by lacerations (42.4%). Within the study sample, 42.4% had previous injuries. Patients with facial injuries were 1.5 times more likely to have experienced previous interpersonal violence. This patient group also expressed an overwhelming willingness to change their behavior patterns (91.3%). CONCLUSIONS: The findings of this investigation indicated that most facial trauma patients are between the ages of 20 and 30 years and male. Assault is the most common etiologic agent resulting in facial trauma. Mandible fractures and lacerations are the most likely injuries in the facial trauma patient. Patients experiencing recurrent trauma due to assault are more responsive to violence reduction programs than those experiencing only 1 assault.

Perceived barriers and facilitators to dental treatment among female caregivers of children with and without HIV and their health care providers.

PURPOSE: This study sought to identify barriers and facilitators to dental care among families of predominately low socioeconomic status having children with and without HIV. METHODS: Twelve focus group sessions with African-American and Hispanic caregivers and 18 individual semistructured interviews with key informant health care providers were held at two sites: a hospital-based program (HBP) and a dental school-based program (DSBP), that provide pediatric dental services. SPSS Textsmart software was used to analyze qualitative data within and across group types and sites. RESULTS: Focus group participants (n = 72, averaging 6 women per group) included: HIV-seropositive biological mothers of HIV-seropositive children (4 groups); HIV-seronegative caregivers of HIV-seropositive children (4 groups); and Medicaid-eligible, HIV-seronegative caregivers of HIV-seronegative children (4 groups). The most commonly expressed barrier to dental care across groups was poor interpersonal communication between dental staff and caregiver/child. HIV-seronegative groups cited health care delivery system factors as barriers to receiving dental care more frequently than HIV-seropositive caregivers who cited shame/anger and family illness as being more important. Common facilitators were positive communication and transportation assistance. Unique facilitators for HIV-seropositive groups were coordination of the dental visits with medical appointments at the HBP. Key informants acknowledged high stress in families having children with HIV/AIDS, cited dental fear among caregivers as a barrier to dental treatment adherence and reported that dental care seemed to be a low priority among many of these families. CONCLUSIONS: Facilitators and barriers to care included factors in the family, dental care and health care delivery systems as well as interpersonal communication between the dental providers and the families.