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Objective: To identify the impact of sociodemographic and health variables on the age at which patients undergo cleft lip repair, cleft palate repair, and primary speech evaluation., Design: A retrospective, noninterventional quality assessment, and quality improvement study was designed., Setting: This institutional study was performed at Michigan Medicine in Ann Arbor, MI., Patients: All patients born between 2011 and 2014 who received surgical cleft repair, excluded those who were adopted (n = 165)., Main Outcome Measure: The age at which patients undergo cleft lip repair, cleft palate repair, and primary speech evaluation., Results: Cleft lip repair was performed significantly later for patients identifying as Asian (18 weeks, P = .01), patients with Child Protective Services contact (19 weeks, P = .01), patients with a significant comorbidity (14 weeks, P = .02), and patients who underwent preliminary lip adhesion surgery (19 weeks, P < .01). Cleft palate repair was performed significantly later for patients identifying racially as Asian (19 weeks, P = .03) and other (22 weeks, P = .03). Preliminary speech and language evaluation were performed significantly later for patients identifying as black (55 weeks, P = .03) and patients diagnosed with an isolated cleft lip (71 weeks, P < .01)., Conclusions: Timing of cleft lip, cleft palate repair, and primary speech and language evaluation are subject to variation which may be predicted by clinically accessible factors. By identifying race, Child Protective Services contact, and care variables as significant predictors of increased patient age at time of intervention, multidisciplinary cleft care teams can proactively allocate patient support resources.

Objective: Preference-sensitive surgical decisions merit shared decision-making, as decision engagement can reduce decisional conflict and regret. Elective cleft-related procedures are often preference sensitive, and therefore, we sought to better understand decision-making in this population., Design: Semistructured interviews were conducted to elicit qualitative data. A hierarchical codebook was developed through an iterative process in preparation for thematic analysis. Thematic analysis was performed to examine differences between patients and caregivers., Setting: Multidisciplinary cleft clinic at a tertiary care center., Participants: Patients with cleft lip aged 8 and older (n = 31) and their caregivers (n = 31) were purposively sampled. Inability to converse in English, intellectual disability, or syndromic diagnoses resulted in exclusion., Main Outcome Measures: Preferences surrounding surgical decision-making identified during thematic analysis., Results: Mean patient age was 12.7 (standard deviation: 3.1). Most had unilateral cleft lip and palate (43.8%). Three themes emerged: Insufficient Understanding of Facial Difference and Treatment, Diversity of Surgical Indications, and Barriers to Patient Autonomy. Almost half of caregivers believed their children understood their clefts, but most of these children failed to provide information about their cleft. Although many patients and caregivers acknowledged that surgery addressed function and/or appearance, patients and caregivers exhibited differences regarding the necessity of surgery. Furthermore, a large proportion of patients believed their opinions mattered in decisions, but less than half of caregivers agreed., Conclusions: Patients with clefts desire to participate in surgical decisions but have limited understanding of their facial difference and surgical indications. Cleft surgeons must educate patients and facilitate shared decision-making.

Background: Although revision surgery is part of the reconstructive process for children with cleft lip and/or cleft palate, the indications for revision vary, and the extent to which surgeons and families agree on appearance is unclear. The authors sought to understand the extent to which children with cleft lip and/or cleft palate, surgeons, caregivers, and control observers agree on satisfaction with appearance and the desire for revision., Methods: Children with cleft lip and/or cleft palate (n = 100) and their caregivers (n = 100) were surveyed regarding satisfaction with appearance using the Cleft Evaluation Profile. Surgeons (n = 10) and control observers (n = 10) rated photographs of these children using questions analogous to the Cleft Evaluation Profile. General linear model repeated measures analysis of variance were used to detect significant differences between raters, with an alpha of 0.05., Results: The children reported greater satisfaction with appearance across all domains compared with surgeons (nose, 7.77 versus 5.51, p < 0.001; lip, 7.94 versus 5.90, p < 0.001; maxilla, 8.16 versus 6.56, p < 0.001) and general observers (nose, 7.78 versus 6.00, p < 0.001; lip, 7.80 versus 6.12, p < 0.001; maxilla, 8.16 versus 7.40, p < 0.001). Children and caregivers expressed similar degrees of satisfaction with appearance of the lip (5.48 ± 1.69 versus 5.6 ± 1.49, p > 0.5) and maxilla (6.08 ± 1.1 versus 5.8 ± 1.2, p = 0.07). There was no significant relationship between children and surgeons in terms of the desire for revision surgery (p = 0.44)., Conclusions: All groups expressed differing levels of satisfaction with cleft-specific aspects of appearance. Importantly, children were more satisfied than all other groups. Care must be taken to evaluate perceptions of all stakeholders before moving forward with cleft revision surgery.

Background: For children with cleft lip and/or palate, access to care is vital for optimizing speech, appearance, and psychosocial outcomes. The authors posited that inadequate access to care negatively impacts outcomes in this population., Methods: Sixty caregivers of children with cleft lip and palate were surveyed to assess perceived barriers using the validated Barriers to Care questionnaire. The questionnaire includes 39 items divided into five subscales, with higher scores indicating fewer barriers. Caregiver-reported outcomes were assessed using the Cleft Evaluation Profile, which captures cleft-specific appearance- and speech-related outcomes. Higher scores correspond to less satisfactory outcomes. Desire for revision surgery was assessed as a binary outcome among caregivers. Multivariable regression was used to evaluate the relationship of barriers to care, caregiver-reported outcomes, and desire for revision, adjusting for clinical and demographic covariates., Results: Sixty percent of caregivers perceived barriers to care, and caregivers who reported poorer access to care described poorer cleft-related outcomes (r = 0.19, p = 0.024). Caregivers with poorer skills (r = 0.17, p = 0.037), expectations (r = 0.17, p = 0.045), and pragmatics (r = 0.18, p = 0.026) subscale scores were associated with worse Cleft Evaluation Profile scores. Barriers were also negatively associated with aesthetic item scores (r = 0.11, p = 0.025). Finally, caregivers reporting fewer barriers were 21.2 percent less likely to express interest in revision surgery., Conclusions: Barriers to care were associated with poorer appearance-related outcomes and increased interest in revision among caregivers of cleft patients. Enhancing access to care is critical in order to effectively meet goals of care for these families.

Objective: To identify disparities in compliance of and care for patients with cleft lip and/or palate (CL/P) by determining the impact of sociodemographic variables on the rate of missed appointments and Child Protective Services (CPS) involvement., Design: A retrospective, noninterventional quality assessment and quality improvement study were designed., Setting: This institutional study was performed at the University of Michigan in Ann Arbor, Michigan., Patients: All patients born between January 1, 2011, and December 31, 2014, who underwent surgical CL/P repair, excluding those with fewer than 5 appointments (n = 178)., Main Outcome Measure: The rate of missed appointments, calculated as the total number of no-show appointments divided by the total number of scheduled appointments. All appointments from CL/P diagnosis to data collection were considered, including those outside of plastic surgery., Results: The average patient was 4.5 years of age and had 49 total scheduled appointments. The overall rate of missed appointments was 9.6%, with 66.8% of patients missing at least 1 scheduled visit. Patients who were black ( P = .04), not affiliated with a religion ( P = .01), Medicaid users ( P = .01), from an unstable social background ( P = .01), or received need-based financial assistance ( P = .00) were significantly more likely to miss appointments. Child Protective Services was involved with 3.9% of patients., Conclusions: Disparities exist in attendance rates among patients with CL/P, and at-risk patient populations have been identified. The allocation of cleft care resources must be efficiently planned in order to enhance the quality of care for at-risk individuals.

Objective: Gender values and beliefs are known to influence family functioning. The aim of the current study was to describe the association between views about gender and family functioning within families with a child with a craniofacial difference (CFD)., Design: Participants included 74 dyads composed of children (8-18 years old) with CFD (n = 36 female), including cleft lip/palate, and a parent (n = 56 female). Children and caregivers both completed the activities subscale of the Occupations, Activities, and Traits-Attitudes Measure (C/OAT-AM). Children completed selected items from the Parent Perception Inventory (PPI). Caregivers completed the nurturance subscale of the Parenting Dimensions Inventory-Short Form (PDI-S) and the Parenting Stress Scale (PSS)., Setting: Participants were recruited from an outpatient program at an academic medical center., Results: There were no significant relationships among the participant demographics and the study variables, with the exception that more complex CFD diagnoses were correlated with increased parenting stress. General linear modeling showed that parents with more flexible gender attitudes reported more nurturing parenting behaviors. There was also a positive association between parental flexibility in gender views and child-reported parent-child relationship quality. An interaction showed that the relationship between parental flexibility in gender views and child-reported relationship quality was stronger for females and nonsignificant for males., Conclusions: Caregivers with more flexible gender attitudes perceived themselves as more nurturing and were seen more positively by their daughters with a CFD. This pattern may inform parenting interventions for CFD populations.

Background: The association between perception of speech and health-related quality of life (HRQOL) among patients with cleft palate is not well understood. The purpose of this study was to determine: the agreement between patient and parent perception of speech, the correlation between patient/parent speech perception and objective analysis by a speech-language pathologist (SLP), and the relationship between objective speech analysis and HRQOL among children with cleft lip with or without palate (CLCP)., Methods: The authors surveyed 108 CLCP patients who received treatment at a large tertiary medical center from 2013 to 2014. Patients and parents were queried regarding their difficulty with speech, and an SLP performed perceptual speech analysis with each patient. Patient-reported survey instruments were used to assess anxiety, depression, anger, peer relationships, stigma, and overall psychosocial health. The authors assessed the agreement between patients and SLP analysis as well as association between speech and HRQOL., Results: Patient and parent-reported speech quality demonstrated moderate agreement regarding the quality of the child's speech (r = 0.46-0.64). Parent and patient speech perception was not well associated with SLP analysis (V = 0.06-0.30). Patient speech perception was correlated with depression (P = 0.03), while SLP analysis was correlated with anger (P = 0.03, P = 0.004), depression (P = 0.007), and difficulty with peer relationships (P = 0.02)., Conclusions: Patients and parents have different perceptions of the quality of the child's speech, and their ratings differ from SLP perceptual speech analysis. Both patient speech perception and SLP analysis are correlated with important aspects of quality of life, and should be considered when evaluating children with CLCP.

Background: Health-related quality of life is inconsistently captured among children with cleft lip and palate. The Patient-Reported Outcomes Measurement Information System (PROMIS) captures health-related quality of life, with the added benefit of comparability across clinical conditions. In this study, the authors define the validity and feasibility of PROMIS among children with clefts., Methods: Children with cleft lip and palate who were at least 5 years old and able to complete instruments independently were eligible for inclusion (n = 93). Children completed PROMIS anxiety, depression, and peer relationship item banks as short forms or computerized adaptive tests. Participants also completed the Pediatric Quality of Life Inventory. Construct validity was measured by Spearman correlations between PROMIS and the Pediatric Quality of Life Inventory controlling for race, sex, age, and income. Feasibility was measured using instrument completion time, reading level, and floor/ceiling effects., Results: PROMIS computerized adaptive tests (peer relationship, r = 0.49; depression, r = -0.56; and anxiety, r = -0.36) and short forms (peer relationship, r = 0.65; depression, r = -0.54; and anxiety, r = -0.56) demonstrated moderate correlation with the Pediatric Quality of Life Inventory. Computerized adaptive tests had fewer floor (0 percent versus 0 percent) and ceiling (8.6 to 19.3 percent versus 21.8 to 41.9 percent) effects than short forms, and demonstrated better readability. Computerized adaptive tests required more time than short forms (peer relationship, 0.84 ± 0.67 versus 1.3 ± 0.92; depression, 0.52 ± 0.38 versus 1.1 ± 0.73; and anxiety, 0.53 ± 0.23 versus 1.1 ± 0.62; p = 0.001), as each computerized adaptive test included on average four more questions., Conclusions: PROMIS correlates well with the Pediatric Quality of Life Inventory and demonstrates similar accuracy, with better readability and efficiency. Use of PROMIS will improve our ability to compare children with cleft lip and palate to diverse populations and clinical conditions., Clinical Question/level of Evidence: Diagnostic, II.

Purpose: Children with cleft lip with or without palate (CLCP) require multiple reconstructive procedures, however, little is known about their desire for surgical revision. The purpose of this study was to examine the relationship between health-related quality of life (HRQOL) and the desire for revision., Methods: The authors surveyed children with CLCP (n = 71) and their caregivers regarding general and cleft-specific HRQOL and the desire for revision surgery. The authors used logistic regression models to evaluate the relationship between HRQOL and the desire for revision stratified by age, and determined the level of agreement between caregivers and children., Results: In this cohort, 54.9% of children desired revision, primarily of the nose (n = 23), lip (n = 20), and dentoalveolar structures (n = 19). Children 11 years or older were more likely to desire revision than younger children (OR 3.39, 95% CI [1.19, 9.67], P <0.05). Children who reported poorer HRQOL with respect to appearance (OR 2.31, 95% CI [1.25-4.29], P = 0.008), social development (OR 0.91, 95% CI [0.84-0.99], P = 0.02), and communication (OR 0.94, 95% CI [0.89-0.99], P = 0.02) were significantly more likely to desire revision than children who reported more positive HRQOL. Caregivers' and children's desires for revision were only modestly correlated (r = 0.41)., Conclusions: Children with CLCP who report poorer HRQOL are more likely to desire revision than children with higher HRQOL; these differences are further magnified among older children. Given the modest correlation between patient and caregiver goals for revision, it is important to evaluate both perspectives when considering revision surgery.

Background: Health care policy makers are increasingly encouraging comparative effectiveness research. Little is known regarding comparative studies among children with cleft lip and/or palate. Cleft lip and/or palate profoundly influences self-perception and social functioning, and patient-reported outcomes provide a unique perspective on the success of reconstruction. The purpose of this study was to systematically review the literature regarding patient-reported outcomes among patients with cleft lip and/or palate., Methods: The authors reviewed articles from MEDLINE, Embase, and PsycInfo that examined the use of patient-reported outcome instruments for cleft lip and/or palate. Studies of patients with cleft lip and/or palate across any age that described the use of patient-completed measures in patient and control populations were included. A research librarian confirmed the search, and two independent, blinded reviewers performed full-text review., Results: The authors identified 1979 articles and selected 30 for inclusion. Forty-two different assessment tools were used to analyze factors such as self-esteem, behavior, and social support. The Strengths and Difficulties Questionnaire was most commonly used (n = 7), followed by the Childhood Experience Questionnaire (n = 5), and the Satisfaction with Appearance survey (n = 4). Barriers to analysis included lack of standardization of survey administration, effect of publication bias, and variations in patient populations between individual studies., Conclusions: Comparative studies of patient-reported outcomes among patients with cleft lip and/or palate are infrequent. Many instruments exist to measure patient-reported outcomes in this population, but no specific standard exists. Identifying efficient and targeted forms of instrument selection and administration will enhance comparative studies among children with cleft lip and/or palate., Clinical Question/level of Evidence: Diagnostic, III.

Objective: To identify the impact of sociodemographic and health variables on the age at which patients undergo cleft lip repair, cleft palate repair, and primary speech evaluation. Design: A retrospective, noninterventional quality assessment, and quality improvement study was designed. Setting: This institutional study was performed at Michigan Medicine in Ann Arbor, MI. Patients: All patients born between 2011 and 2014 who received surgical cleft repair, excluded those who were adopted (n = 165). Main Outcome Measure: The age at which patients undergo cleft lip repair, cleft palate repair, and primary speech evaluation. Results: Cleft lip repair was performed significantly later for patients identifying as Asian (18 weeks, P =.01), patients with Child Protective Services contact (19 weeks, P =.01), patients with a significant comorbidity (14 weeks, P =.02), and patients who underwent preliminary lip adhesion surgery (19 weeks, P <.01). Cleft palate repair was performed significantly later for patients identifying racially as Asian (19 weeks, P =.03) and other (22 weeks, P =.03). Preliminary speech and language evaluation were performed significantly later for patients identifying as black (55 weeks, P =.03) and patients diagnosed with an isolated cleft lip (71 weeks, P <.01). Conclusions: Timing of cleft lip, cleft palate repair, and primary speech and language evaluation are subject to variation which may be predicted by clinically accessible factors. By identifying race, Child Protective Services contact, and care variables as significant predictors of increased patient age at time of intervention, multidisciplinary cleft care teams can proactively allocate patient support resources. [ABSTRACT FROM AUTHOR]

Objective: Preference-sensitive surgical decisions merit shared decision-making, as decision engagement can reduce decisional conflict and regret. Elective cleft-related procedures are often preference sensitive, and therefore, we sought to better understand decision-making in this population. Design: Semistructured interviews were conducted to elicit qualitative data. A hierarchical codebook was developed through an iterative process in preparation for thematic analysis. Thematic analysis was performed to examine differences between patients and caregivers. Setting: Multidisciplinary cleft clinic at a tertiary care center. Participants: Patients with cleft lip aged 8 and older (n = 31) and their caregivers (n = 31) were purposively sampled. Inability to converse in English, intellectual disability, or syndromic diagnoses resulted in exclusion. Main Outcome Measures: Preferences surrounding surgical decision-making identified during thematic analysis. Results: Mean patient age was 12.7 (standard deviation: 3.1). Most had unilateral cleft lip and palate (43.8%). Three themes emerged: Insufficient Understanding of Facial Difference and Treatment, Diversity of Surgical Indications, and Barriers to Patient Autonomy. Almost half of caregivers believed their children understood their clefts, but most of these children failed to provide information about their cleft. Although many patients and caregivers acknowledged that surgery addressed function and/or appearance, patients and caregivers exhibited differences regarding the necessity of surgery. Furthermore, a large proportion of patients believed their opinions mattered in decisions, but less than half of caregivers agreed. Conclusions: Patients with clefts desire to participate in surgical decisions but have limited understanding of their facial difference and surgical indications. Cleft surgeons must educate patients and facilitate shared decision-making. [ABSTRACT FROM AUTHOR]

Objective: To identify disparities in compliance of and care for patients with cleft lip and/or palate (CL/P) by determining the impact of sociodemographic variables on the rate of missed appointments and Child Protective Services (CPS) involvement. Design: A retrospective, noninterventional quality assessment and quality improvement study were designed. Setting: This institutional study was performed at the University of Michigan in Ann Arbor, Michigan. Patients: All patients born between January 1, 2011, and December 31, 2014, who underwent surgical CL/P repair, excluding those with fewer than 5 appointments (n = 178). Main Outcome Measure: The rate of missed appointments, calculated as the total number of no-show appointments divided by the total number of scheduled appointments. All appointments from CL/P diagnosis to data collection were considered, including those outside of plastic surgery. Results: The average patient was 4.5 years of age and had 49 total scheduled appointments. The overall rate of missed appointments was 9.6%, with 66.8% of patients missing at least 1 scheduled visit. Patients who were black (P = .04), not affiliated with a religion (P = .01), Medicaid users (P = .01), from an unstable social background (P = .01), or received need-based financial assistance (P = .00) were significantly more likely to miss appointments. Child Protective Services was involved with 3.9% of patients. Conclusions: Disparities exist in attendance rates among patients with CL/P, and at-risk patient populations have been identified. The allocation of cleft care resources must be efficiently planned in order to enhance the quality of care for at-risk individuals. [ABSTRACT FROM AUTHOR]

Objective: Gender values and beliefs are known to influence family functioning. The aim of the current study was to describe the association between views about gender and family functioning within families with a child with a craniofacial difference (CFD). Design: Participants included 74 dyads composed of children (8-18 years old) with CFD (n = 36 female), including cleft lip/palate, and a parent (n = 56 female). Children and caregivers both completed the activities subscale of the Occupations, Activities, and Traits–Attitudes Measure (C/OAT-AM). Children completed selected items from the Parent Perception Inventory (PPI). Caregivers completed the nurturance subscale of the Parenting Dimensions Inventory–Short Form (PDI-S) and the Parenting Stress Scale (PSS). Setting: Participants were recruited from an outpatient program at an academic medical center. Results: There were no significant relationships among the participant demographics and the study variables, with the exception that more complex CFD diagnoses were correlated with increased parenting stress. General linear modeling showed that parents with more flexible gender attitudes reported more nurturing parenting behaviors. There was also a positive association between parental flexibility in gender views and child-reported parent-child relationship quality. An interaction showed that the relationship between parental flexibility in gender views and child-reported relationship quality was stronger for females and nonsignificant for males. Conclusions: Caregivers with more flexible gender attitudes perceived themselves as more nurturing and were seen more positively by their daughters with a CFD. This pattern may inform parenting interventions for CFD populations. [ABSTRACT FROM AUTHOR]