Correlation of Orthognathic Surgical Movements to Perception of Facial Appearance in Patients With Cleft Lip and Palate.

STUDY DESIGN: Cephalometric scans were compared before and after surgery to assess the degree of correction. Correlations between skeletal movements and survey outcomes were determined using multivariate regression analysis. OBJECTIVE: This study aims to identify relationships between subjective observer-reported improvements in esthetics and emotional appearance with specific surgical movements. METHODS: Ten patients at a single tertiary institution (average age: 18.1 ± 0.8), 9 males and 1 female, underwent orthognathic repair and had comprehensive cephalometric records. Standardized anterior posterior and lateral pre and postoperative photographs of patients were included in a survey to clinicians to assess noncognitive domains on a Likert Scale (1-10). CLEFT-Q was administered to gauge patient satisfaction in categories of appearance, speech, and quality of life. RESULTS: Per clinicians, multiple domains increased including facial attractiveness (4.1 ± 0.7 versus 7.3 ± 0.7, P < 0.001), friendliness (4.5 ± 0.4 versus 7.3 ± 0.5, P < 0.001), confidence (4.1 ± 0.4 versus 7.1 ± 0.4, P < 0.001), and recommendation for surgery decreased (8.9 ± 0.1 versus 3.6 ± 0.5, P < 0.001). Speech distress decreased with increased SNA and convexity, whereas Psychological and Social scores decreased with an increased ANB. Functional eating and drinking scores increased with maxillary depth. CONCLUSIONS: Orthognathic surgery improves many noncognitive domains in patients with cleft lip and palate as assessed by both patients and clinicians on all aspects of facial attractiveness and perception. These findings demonstrate objective bases of skeletal adjustments for perceived improvements in facial appearance and emotion.

Prevalence and Factors Associated with Behavioral Problems in 5-Year-Old Children Born with Cleft Lip and/or Palate from the Cleft Collective.

OBJECTIVES: To determine the UK prevalence of behavioral problems in 5-year-old children born with isolated or syndromic cleft lip and/or palate (CL/P) compared to the general population and identify potentially associated factors. DESIGN: Observational study using questionnaire data from the Cleft Collective 5-Year-Old Cohort study and three general population samples. MAIN OUTCOME MEASURE: The Strengths and Difficulties Questionnaire (SDQ). PARTICIPANTS: Mothers of children (age: 4.9-6.8 years) born with CL/P (n = 325). UK general population cohorts for SDQ scores were: Millennium Cohort Study (MCS) (n = 12 511), Office of National Statistics (ONS) normative school-age SDQ data (n = 5855), and Avon Longitudinal Study of Parents and Children (ALSPAC) (n = 9386). RESULTS: By maternal report, 14.2% of children born with CL/P were above clinical cut-off for behavioral problems, which was more likely than in general population samples: 7.5% of MCS (OR = 2.05 [1.49-2.82], P < 0.001), 9.8% of ONS (OR = 1.52 [1.10-2.09], P = 0.008), and 6.6% of ALSPAC (OR = 2.34 [1.70-3.24], P < 0.001). Children in the Cleft Collective had higher odds for hyperactivity, emotional and peer problems, and less prosocial behaviors. Maternal stress, lower maternal health-related quality of life and family functioning, receiving government income support, and maternal smoking showed evidence of association (OR range: 4.41-10.13) with behavioral problems, along with maternal relationship status, younger age, and lower education (OR range: 2.34-3.73). CONCLUSIONS: Findings suggest elevated levels of behavioral problems in children born with CL/P compared to the general population with several associated maternal factors similar to the general population.

The burden of psychiatric disorders associated with orofacial cleft pathology among children in Ontario, Canada.

BACKGROUND: Individuals with orofacial cleft (OFC) may be at a higher risk of developing psychiatric disorders (PD) than the general population. We determined the risk of psychiatric diagnoses in children with OFC in Canada. METHODS: This population-based retrospective cohort study used health administrative data from the province of Ontario, Canada. Children with OFC who were born between April 1, 1994, and March 31, 2017, in Ontario were matched to five non-OFC children based on sex, date of birth, and mother's age. We determined the rate of events and time-to-event for first diagnosis of PD in children aged ≥ 3 years (y), and for intellectual developmental delay (IDD) from birth. Risk factors for PD and IDD were assessed using 1-way ANOVA for means, Kruskal-Wallis for medians, and the χ2 test for categorical variables. OUTCOMES: There were 3051 children with OFC (matched to 15,255 controls), of whom 2515 patients with OFC (12,575 controls) had a complete follow-up to the third birthday. Children with OFC were more likely to have PD than controls (54.90 vs. 43.28 per 1000 patient-years, P < .001), with a mean age to first diagnosis of 8.6 ± 4.2 y. The cleft palate group had the highest risk (HR 1.33, 95% CI 1.18-1.49). Children with OFC also had a higher risk of IDD than non-OFC children (27.78 vs. 3.46 per 1000 patient-years, p < .001). INTERPRETATION: Children born with OFC in Ontario had a higher risk of psychiatric diagnosis and IDD compared to controls. Further research is also required to better understand the predictors of variation in risk, including geographic location and the presence of congenital abnormalities, and identify potential areas for intervention. EVIDENCE RATING SCALE FOR PROGNOSTIC/RISK STUDIES: Level II.

Optimizing the Psychosocial Function Measures in the International Consortium for Health Outcomes Measurement Standard Set for Cleft.

BACKGROUND: To ensure the feasibility of implementing PROMs in clinical practice, they must be continually appraised for undue burden placed on patients and clinicians and their usefulness for decision-making. This study assesses correlations between the CLEFT-Q psychosocial scales in the International Consortium for Health Outcomes Measurement Standard Set for cleft and explores their associations with patient characteristics and psychosocial care referral. METHODS: Spearman correlation coefficients were calculated for CLEFT-Q psychological function, social function, school function, face, speech function, and speech-related distress scales. Logistic regressions were used to assess the association of cleft phenotype, syndrome, sex, and adoption status on scale scores and clinical referral to psychosocial care for further evaluation and management. RESULTS: Data were obtained from 3067 patients with cleft lip and/or palate at three centers. Strong correlations were observed between social function and psychological function (r > 0.69) and school function (r > 0.78) scales. Correlation between school function and psychological function scales was lower (r = 0.59 to 0.68). Genetic syndrome (OR, 2.37; 95% CI, 1.04 to 5.41), psychological function (OR, 0.92; 95% CI, 0.88 to 0.97), school function (OR, 0.94; 95% CI, 0.90 to 0.98), and face (OR, 0.96; 95% CI, 0.94 to 0.98) were significant predictors for referral to psychosocial care. CONCLUSIONS: Because social function as measured by the CLEFT-Q showed strong correlations with both school and psychological function, its additional value for measuring psychosocial function within the Standard Set is limited, and it is reasonable to consider removing this scale from the International Consortium for Health Outcomes Measurement Standard Set for cleft.

Thematic Analysis of Coping Strategies Used by Parents of Children with Cleft Lip and Palate in El Salvador.

OBJECTIVE: The aim of this study was to describe coping strategies used by parents of children with cleft palate with or without a cleft (CP ± L) during the early development of their children in El Salvador. DESIGN: Qualitative interviews were completed with 16 parents of children born with CP ± L who were 6 months to 6 years old. Parents were questioned about their emotions and coping during eight time periods: prenatal, birth, social interaction before the first surgery, the beginning of surgeries, social interaction after the first surgery, early childhood education (ECE), speech-language therapy, and formal education. Thematic analysis (TA) was used to identify coping strategies as conceptualized by Lazarus and Folkman (1984). RESULTS: Four major themes emerged: (a) experienced emotions related to diagnosis, (b) interpretations related to the birth of a child with a cleft, (c) seeking and experiencing cleft treatment, and (d) social interaction of the children. During prenatal and birth stages, parents used emotion-focused strategies. A few hours to a week after birth, they used problem-focused strategies, which led them in search of treatment. Some parents used avoidance strategies during periods of social interaction before surgery, ECE, and formal education. Socioeconomic challenges impacted access to speech-language therapy. Sociocultural factors, such as discrimination, religion, and folk beliefs, appeared to influence some of the coping strategies used by parents. CONCLUSIONS: Problem-focused strategies appear to be helpful in seeking surgical treatments. The emotion-focused strategy of avoidance seemed to have adverse effects in minimizing opportunities for social interaction prior to surgery and early education.

Psychometric Validation of the CLEFT-Q Patient Reported Outcome Measure: A Prospective Study to Examine Cross-Sectional Construct Validity.

OBJECTIVE: CLEFT-Q is a condition-specific patient-reported outcome measure (PROM) for patients with cleft lip and/or palate (CL/P). The aim of this study was to examine the cross-sectional construct validity of the CLEFT-Q scales. DESIGN: Construct validity was assessed through a prospective study that tested hypotheses regarding correlations of scores with other PROMs that measure related constructs. SETTING: Seven cleft centres in Canada, the USA, and UK were involved. PATIENTS/PARTICIPANTS: Patients were aged eight to 29 years with CL/P. INTERVENTIONS: Before undergoing rhinoplasty, orthognathic, cleft lip scar revision, and alveolar bone graft, participants were asked to complete the following PROMs: CLEFT-Q (9 scales), Child Oral Health Impact Profile (socio-emotional subscale) and Cleft Hearing Appearance and Speech Questionnaire (features 1 subscale). MAIN OUTCOME MEASURE(S): The correlation coefficients examining the relationship between the scales were the main outcome measures. Correlations (Spearman) were calculated and interpreted as follows: <0.3 weak, 0.30 to 0.50 moderate, ≥0.50 strong. RESULTS: Participants (n = 177) were mostly male (61%) and aged between eight and 11 years (42%). Overall, 38 of 52 (73%) hypotheses tested were supported. More specifically, 20 of 26 (77%) hypotheses about correlations between the appearance scales were supported, two of three (67%) hypotheses about correlations between the health-related quality of life scales were supported, and 16 of 23 (70%) hypotheses about correlations between the appearance and health-related quality of life scales were supported. CONCLUSIONS: Cross-sectional construct validity of the CLEFT-Q scales adds further evidence of the psychometric properties of this instrument.

EFFECT OF BIOPSYCHOSOCIAL INTERVENTION ON BEAUTY SATISFACTION AFTER STAGED SURGERY AMONG ADOLESCENTS WITH ORAL FACIAL CLEFTS.

Oral facial clefts (OFC) among adolescents can pose several significant challenges. Assessing one's contentment with one's appearance following surgery may be useful in identifying young people who may have adjustment issues. Aim - to find the efficacy of the biopsychosocial intervention on beauty satisfaction among adolescents with oral facial clefts surgery. A descriptive survey was conducted among 86 adolescents, selected by simple random technique, who underwent staged surgical procedures for OFC at Charles Pinto Centre for Cleft Lip & Palate in India. Beauty satisfaction was measured by a Four-point rating scale. During the second phase, a Quasi-experimental design was adopted among 10 samples & Seven-day biopsychosocial intervention package was administered after the pretest assessment. Most of the samples have only an average level of beauty satisfaction of 48 (55.8%), whereas a high level of beauty satisfaction was observed only among 24 (27.9%) samples & 14 (16.3%) had a low level. The study also revealed that both genders equally experience dissatisfaction with beauty (P=0.381). F- value for comparing beauty satisfaction among different age groups of adolescents was found to be non-significant at 0.05 level (p=0.253). This depicts that as age increases adolescents are more conscious of their beauty and appearance. The Level of beauty satisfaction has increased after the implementation of the biopsychosocial intervention (p=0.001) at 0.01 level in the second phase, which is in tune with the study findings of Kapp Simon. Counselling and looking at actual facial features can help people accept their looks, according to study results on beauty satisfaction. Thus, a productive line of inquiry into cleft psychosocial research that ascertains the alterations in facial growth and the surgical experience must be assembled through observational, longitudinal, and cross-sectional investigations.

The impact of orthognathic surgery on quality of life in individuals with oral clefts.

BACKGROUND/OBJECTIVES: To evaluate the relationships between individual, environmental, clinical factors and oral health-related quality of life (OHRQoL) in patients with cleft lip and palate (CLP) following orthognathic surgery. MATERIALS AND METHODS: A follow-up study was conducted involving 69 adults with unilateral and bilateral CLP under orthodontic treatment. Interviews and oral examinations were conducted prior to orthognathic surgery (T0) to evaluate age, gender, psychological well-being, dental caries, malocclusion, social support, social networks, family income and education and OHRQoL. All participants were reviewed after 6 months (T1) to re-assess psychological well-being, malocclusion and OHRQoL. Structural equation modeling estimated the associations between the variables. RESULTS: OHRQoL total scores reduced following orthognathic surgery, from 11.7 to 6.9 (P < 0.01). Occlusal characteristics and psychological well-being improved between T0 and T1. In the structural equation modeling, reduction of malocclusion (ß = 0.02) between T0 and T1 directly predicted poor OHRQoL at T1. Improvement of psychological well-being between T0 and T1 was associated with better OHRQoL at T1 (ß = -0.07). Dental caries and malocclusion at T0 were indirectly linked to poor OHRQoL at T1 (ß = 0.02). LIMITATIONS: The short follow-up period of 6 months after orthognathic surgery. CONCLUSIONS/IMPLICATIONS: This represents the first prospective study examining the interrelationships of predictors of OHRQoL in patients with CLP after orthognathic surgery. OHRQoL and psychological well-being improved after orthognathic surgery. Clinical and psychological characteristics were important determinants of OHRQoL. These findings suggest the importance of the biopsychosocial model of health and the patient-centered approach in oral health care in individuals with CLP.

Quality of Life of Adopted Chinese Versus Nonadopted Dutch Children with Cleft Lip and/or Palate: A Propensity Score Matched Analysis.

OBJECTIVE: To examine quality of life in internationally adopted children with cleft lip and/or palate (CL/P) versus non-adopted children with CL/P. DESIGN: Cross sectional study. SETTING: Multidisciplinary cleft team of a secondary and tertiary hospital in the Netherlands. METHODS: Parents of children under the age of 8 treated by the multidisciplinary cleft team of our institutions were asked to fill out a questionnaire containing demographic and clinical data and a validated parent proxy measure of cleft-specific quality of life instrument for children aged 0-8: the CleftChild-8. Adopted children were matched to non-adopted children using propensity score matching based on sex, age, type of cleft, if palatal surgery was completed and the level of education of the parent. CleftChild-8 scores were then compared between the matched samples of adopted and non-adopted children with CL/P. MAIN OUTCOME MEASURE(S): Differences in (sub)domain scores of the CleftChild-8. RESULTS: Most median CleftChild-8 scores of the adopted children (n = 29) were slightly lower compared to the 29 matched non-adopted children. A significant difference was seen for the domain score 'satisfaction with (operative) treatment' and 3 of the 13 subdomain scores: 'post-operative results', 'acceptance by siblings' and 'acceptance by family/friends'. CONCLUSIONS: By parent report, adopted children with CL/P experienced some areas of lower quality of life when compared to non-adopted children. Members of cleft teams should be aware of the problems associated with adoption and offer additional guidance and counseling to adopted children and their parents.

Evaluating Psychosocial Problems in School-Age Children with Cleft Lip and Palate in Bandung, Indonesia Using CBCL/6-18.

BACKGROUND: Cleft lip with or without palate (CL ± P) may impact children's eating, drinking, speaking, breathing, and hearing. We aim to evaluate psychosocial problems in Indonesian cleft center school-age patients identified after one or more surgical interventions. METHODS: This is a cross-sectional study of parent report of patients with unilateral CL ± P who had cleft surgery from 2011 to 2016 in the Bandung Cleft Center using the Bahasa Indonesia version of CBCL/6-18 questionnaire. Descriptive statistics were completed based on measure norms and score ranges. RESULTS: There were 104 participants (56.7% male) with a median age of 8 years old and 73.0% had unilateral cleft of lip, gum, and palate. We found that speech and appearance problems were not perceived by parents for 34.6% of participants after undergoing surgery. The majority of parents reported normal range scores for the Social scale (93.3%) and the School scale (92.3%). In contrast, largely due to the restrictions in the covid-19 pandemic, 78.8% of the patients had below normal range scores for the Activities scale. Borderline or Clinical range scores were reported for 6.7% of children on the Problem Items section and 15.4% of parents endorsed one or more Critical Items about their children, which indicate significant behavioral concerns. CONCLUSION: In this study, we found 6.7% of the school-age children population with CL/P had psychosocial problems. The result of this study hopefully can shed some light in the long-term psychosocial conditions of the CL/P children post-operatively.

Quality of Life and Temporomandibular Disorders in Patients With Skeletal Class III Malocclusion With Cleft Lip and Palate.

OBJECTIVE: The aim of the study was to assess the quality of life (QOL), oral health-related QOL (OHRQOL), temporomandibular disorders (TMDs), and psychological factors in patients with skeletal Class III malocclusion with cleft lip and palate (CLP) and without CLP. DESIGN: Case-control. SETTING: Primary care, institutional practice. PATIENTS: One hundred thirty-six patients with skeletal Class III malocclusion with CLP (n = 68) and without CLP (n = 68). MAIN OUTCOME MEASURES: QOL and OHRQOL were assessed using the World Health Organization Quality of Life-BREF (WHOQOL-BREF) questionnaire and the Oral Health Impact Profile-14 questionnaire, respectively. TMDs and psychological factors were assessed using the Research Diagnostic Criteria for TMD (RDC/TMD). RESULTS: No differences in QOL were found between the groups (P > 0.05). Patients with CLP reported a better OHRQOL (P = 0.025) in the physical pain, physical disability, and psychological disability domains (P < 0.05). Patients with CLP presented with less myofascial pain (OR, 0.28; 95% CI, 0.11-0.71] and other articular conditions (OR 0.24; 95% CI 0.06-0.90]. More patients with CLP reported no chronic pain (P = 0.012). The QOL of patients with CLP with no depression or with no nonspecific physical symptoms including pain (NSPSIP) was better than that of patients without CLP. The OHRQOL of patients with CLP without TMDs or no psychological factors was better than that of patients without CLP. CONCLUSIONS: Patients with skeletal Class III malocclusion who require orthognathic surgery with CLP have better OHRQOL and present with fewer TMDs than those patients without CLP.

Factors Associated With Psychological Adjustment in Adults With Cleft Lip and/or Palate: Findings From a National Survey in the United Kingdom.

OBJECTIVES: Research has identified adults born with cleft lip and/or palate (CL/P) to be at risk of poorer psychological outcomes compared to the general population. This study investigated factors that may contribute to positive and negative adjustment in adults born with CL/P. DESIGN: A survey was designed and distributed by the Cleft Lip and Palate Association in collaboration with the Centre for Appearance Research CAR at the University of the West of England (UWE). There were 207 eligible responses (95% completed online) received between July and October 2018. Dependent variables included the Body Esteem Scale for Adolescents and Adults, Harter's Self Perception Profile for Adults (Global Self-Worth, Social Competence, and Intimacy subscales), the Fear of Negative Appearance Evaluation Scale, and the Revised Adult Attachment Scale. Independent variables were the Revised Life Orientation Test, biodemographic data, and self-reported single-item questions. RESULTS: Factors associated with positive adjustment included reports of a happy childhood, talking about CL/P with family, close friendships, comfort in public spaces, satisfaction with appearance, and a positive life orientation. Psychological distress was associated with a desire for further surgery to improve appearance and/or function. CONCLUSIONS: Several factors were identified that may influence psychological adjustment in adults with CL/P. Throughout childhood, family-centered practice to support family cohesion and an open dialogue about CL/P is indicated, as is support for young people to develop social confidence. For adults returning to the cleft service, treatment options for appearance and/or functional concerns should be explored, with access to psychological support when indicated. Interventions to increase optimism, resilience, and self-acceptance may also be warranted throughout the life span.

Emotional and Behavioral Problems in Children With a Cleft Lip With or Without Palate or an Infantile Hemangioma.

OBJECTIVE: Life can be challenging for children with a visible difference due to a medical condition, and they might be at risk for emotional and behavioral problems. This study examines emotional and behavioral problems in children with a cleft lip with or without palate (CL ± P) or an infantile hemangioma (IH) in relation to the visibility of the condition, the presence of additional condition-related problems, and parental affect. SETTING: This cross-sectional study took place in an academic medical hospital in the Erasmus MC Sophia Children's Hospital, the Netherlands. PARTICIPANTS: A total of 309 parents (mean age = 40.34, 44.00% male) of 182 children with CL ± P and 48 parents (mean age = 39.21, 37.50% male) of 33 children with an IH completed questionnaires. Children were 1.5 to 12 years old. RESULTS: Parents reported fewer child emotional and behavioral problems compared to normative data. Problems reported were mainly related to learning difficulties and parent gender, while visibility of the condition had no significant influence. Parental negative affect was related to child internalizing problems. Parental positive affect was not related to any of the outcome measures. CONCLUSIONS: Parents reported fewer problems for their children compared to normative data. This is inconsistent with previous research, showing similar or worse scores for these children compared to peers. Our findings may be explained by a protective parenting style, a response shift in parents, or problems developing at a later point in life.

Early Alveolar Bone Grafting Is Associated with Lower Regraft Rates and Improvements in Long-Term Psychosocial Outcomes.

BACKGROUND: Late childhood (8 to 10 years of age) has emerged as a vulnerable period in children with cleft and craniofacial anomalies such that increased interventions during this period are associated with worse long-term patient-reported anxiety and depressive symptoms. These findings suggest that one possible practice change may be to consider changes in timing for surgical treatment algorithms. In this work, the authors investigated outcomes in altering the timing of the most common operation in late childhood for cleft lip and palate patients, alveolar bone grafting. METHODS: A two-part, multi-institutional cohort study was conducted. To understand the feasibility of changing alveolar bone graft timing with respect to surgical success, reoperation rates were retrospectively compared among patients grafted at different ages (4 to 7, 8 to 10, and 11 to 13 years of age). To understand the long-term effect of changing alveolar bone graft timing on psychosocial outcomes, the psychosocial suite of the Patient-Reported Outcomes Measurement Information System was prospectively administered to teenagers and adults with cleft lip and palate. RESULTS: Among the three age groups, early grafting (4 to 7 years of age) demonstrated the lowest regraft rates compared with the other groups. As these results suggested that early grafting is a viable alternative to standard timing, we next compared the differences in long-term psychosocial outcomes. Patients who were grafted early reported lower levels of anxiety and depressive symptoms as teenagers and adults. CONCLUSION: Altering timing of one stage in cleft lip and palate reconstruction to an earlier age decreases regraft rates and improves long-term patient-reported anxiety and depressive symptoms. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, III.

FACE-Q craniofacial module: Part 2 Psychometric properties of newly developed scales for children and young adults with facial conditions.

BACKGROUND: The FACE-Q Craniofacial Module is a patient-reported outcome measure designed for patients aged 8 to 29 years with conditions associated with a facial difference. In part 1, we describe the psychometric findings for the original CLEFT-Q scales tested in patients with cleft and noncleft facial conditions. The aim of this study was to examine psychometric performance of new FACE-Q Craniofacial Module scales. METHODS: Data were collected between December 2016 and December 2019 from patients aged 8 to 29 years with conditions associated with a visible or functional facial difference. Rasch measurement theory (RMT) analysis was used to examine psychometric properties of each scale. Scores were transformed from 0 (worst) to 100 (best) for tests of construct validity. RESULTS: 1495 participants were recruited with a broad range of conditions (e.g., birthmarks, facial paralysis, craniosynostosis, craniofacial microsomia, etc.) RMT analysis resulted in the refinement of 7 appearance scales (Birthmark, Cheeks, Chin, Eyes, Forehead, Head Shape, Smile), two function scales (Breathing, Facial), and an Appearance Distress scale. Person separation index and Cronbach alpha values met criteria. Three checklists were also formed (Eye Function, and Eye and Face Adverse Effects). Significantly lower scores on eight of nine scales were reported by participants whose appearance or functional difference was rated as a major rather than minor or no difference. Higher appearance distress correlated with lower appearance scale scores. CONCLUSION: The FACE-Q Craniofacial Module scales can be used to collect and compare patient reported outcomes data in children and young adults with a facial condition.

Optimising the computerised adaptive test to reliably reduce the burden of administering the CLEFT-Q: A Monte Carlo simulation study.

BACKGROUND: Computerised adaptive testing (CAT) has the potential to transform plastic surgery outcome measurement by making patient-reported outcome measures (PROMs) shorter, individualised and more accurate than pen-and-paper questionnaires. OBJECTIVES: This paper reports the results of two optimisation studies for the CLEFT-Q CAT, a CAT intended for use in the field of cleft lip and/or palate. Specifically, we aimed to identify the optimal score estimation and item selection methods for using this CAT in clinical practice. These represent two major components of any CAT algorithm. METHOD: Monte Carlo simulations were performed using simulated data in the R statistical computing environment and incorporated a range of score estimation and item selection techniques. The performance and accuracy of the CAT was assessed by mean items administered, correlation between CAT scores and paired linear assessment scores, and the root mean squared deviation (RMSD) of these score pairs. RESULTS: The accuracy of the CLEFT-Q CAT was not significantly affected by the choice of score estimation or item selection method. Sub-scales which originally contain more items were amenable to greater item reduction with CAT. CONCLUSION: This study shows that score estimation and item selection methods that need minimal processing power can be used in the CLEFT-Q CAT without compromising accuracy. This means that the CLEFT-Q CAT could be administered quickly and efficiently with basic hardware demands. We recommend the use of less computationally intensive techniques in future CLEFT-Q CAT studies.

Preliminary evaluation of pre-speech and neurodevelopmental measures in 7-11-week-old infants with isolated oral clefts.

BACKGROUND: The purpose of this research study was to evaluate the earliest markers of vocal functioning and neurological development in infants with isolated oral cleft of the lip and/or palate (iCL/P). METHODS: Participants were recruited through advertisements and clinic visits at a local mid-western university. A total of eight participants (four unaffected and four with iCL/P), ranging in age from 7.29 to 11.57 weeks, were enrolled and completed demographic and pre-speech measures. A subset of six males (four unaffected and two with iCL/P) successfully completed a structural magnetic resonance imaging scan. RESULTS: Patterns of disrupted vocal control and reduced myelinated white matter were found in participants with iCL/P. CONCLUSIONS: The findings of this study provide a foundation from which to build further research on the neuronal development of infants with oral clefts: the need to evaluate measures of cortical development, inclusion of information on anesthesia exposure and airway obstruction, and suggestions for avoiding identified pitfalls/blocks to obtaining data are discussed. IMPACT: Research in children with isolated oral clefts has demonstrated higher rates of learning disorders connected to subtle differences in brain structure. There is no work evaluating the potential impact of exposure to anesthesia on development. This is the first known attempt to evaluate brain structure and function in infants with isolated oral clefts before exposure to anesthesia. Potential trends of early vocal issues and structural brain differences (less myelinated white matter) were identified in infants with isolated oral clefts compared to unaffected controls. Differences in brain structure and function in infants with isolated oral clefts may be present before surgery.

Long-Term Effect of Multiple Operations on Psychosocial Function in Teenage Cleft Lip and Palate Patients.

BACKGROUND: Cleft lip and palate patients undergo a significant number of interventions during their childhood and adolescence. Although the intention of such interventions is to improve psychosocial functioning, there exists a paucity of data on the psychosocial outcomes of the burden of care on cleft children. In this work, the long-term effects of quantity and timing of childhood operations on teenagers with cleft lip and palate were evaluated. METHODS: Cleft lip and palate patients (aged 14 to 17 years; n = 55) and an age-matched unaffected cohort (n = 14) prospectively enrolled from two institutions were administered the anger, anxiety, and depressive symptoms instruments from the Pediatric Patient-Reported Outcomes Measurement Information System. Total number of operations and operations stratified by age groups (0 to 7, 8 to 10, 11 to 13, and 14 to 17 years) were evaluated in relationship to instrument scores. Descriptive statistics, independent t tests, Pearson correlations, and multiple linear regression analyses were conducted. RESULTS: No differences in overall psychosocial functioning were found between the cleft lip and palate and comparison groups. Total quantity of childhood operations did not correlate to psychosocial functioning of cleft lip and palate teenagers. However, multiple linear regression analyses demonstrated that increased number of operations in the 8- to 10-year-old age range predicted increased anxiety and depressive symptoms in teenagers (ß = 0.38, p = 0.009; and ß = 0.29, p = 0.03, respectively). CONCLUSIONS: It was previously reported by the authors' group that the 8- to 10-year-old age range is an at-risk period for psychosocial distress in children with craniofacial anomalies. Their current work demonstrates that increased number of operations during this time frame may result in long-term consequences in anxiety and depression in cleft lip and palate patients. CLINICAL QUESTION/LEVEL OF EVIDENCE: Risk, II.

Problemas de Salud Mental en Niños con Fisura Labiopalatina / Mental Health Problems in Children with a Lip-Palatal Fissure

Los niños que padecen enfermedades genéticas son una población vulnerable para desarrollar problemas de salud mental. Una de las anomalías congénitas más frecuentes es la fisura labiopalatina (FLAP). Objetivo: Estudiar el impacto a nivel familiar del diagnóstico de FLAP. Metodología: Alcance exploratorio, de diseño cuantitativo y corte transversal. Se construyó un cuestionario autoadministrado compuesto por un consentimiento informado, un cuestionario de datos sociodemográficos, el Child Behavior CheckList y preguntas abiertas. El muestreo fue intencional y la muestra fue de 42 casos. Resultados: El diagnóstico de una fisura labio palatina es una experiencia difícil en los padres, quienes viven este momento de manera negativa, con tristeza y miedo. Sin embargo, los niños pequeños con FLAP presentan niveles de salud mental similares a la población general.

Children with genetic diseases are a vulnerable population in the development of mental health problems. The cleft lip and palate (FLAP) is one of the most common congenital anomalies. Objective: To study the impact at family level of the FLAP diagnosis. Methodology: Exploratory and quantitative design and cross-sectional scope. A self-administered questionnaire consisting of an informed consent, a sociodemographic data questionnaire, the Child Behavior CheckList and open questions, was constructed. The sampling was intentional and composed by 42 cases. Results: The diagnosis of a cleft palatal lip is a difficult experience in parents, who experience this moment in a negative way, with sadness and fear. Nevertheless, children with FLAP present similar levels of mental health with general populatio.

Lay People Esthetic Evaluation of Primary Surgical Repair on Three-Dimensional Images of Cleft Lip and Palate Patients.

Background and Objectives: Previous literature has disclosed that facial attractiveness affects the esthetic evaluation of nose and lip deformity on frontal and lateral photographs. However, it has never been debated if the removal of the external facial features on three-dimensional (3D) models ("cropped assessment bias") could provide a considerable usefulness in the interpretation and comparison of the results. Additionally, it has been assumed on two-dimensional (2D) studies that esthetic assessment biases with respect to observer gender, and it is not acknowledged if and to the extent that "gender assessment bias" may be influenced by a three-dimensional layout. The aim of this study is to investigate if facial traits and observers' gender may affect the esthetic ratings of unilateral cleft lip and palate (UCLP) patients after soft tissue reconstruction. Materials and Methods: Three-dimensional images of ten UCLP patients' images were acquired before the intervention (T0), one-month (T1) and six-months (T2) postoperative. Geomagic® software (version 2014; 3D Systems, Rock Hill, SC, USA) was used to remove the external facial features of 3D surface models. Five-point scale developed by Asher-McDade et al. was used to rate both nasolabial attractiveness and impairment for full-face (FF) and cropped-face (CF) 3D images. Forty-three judges (21 males, 22 females) were enrolled for the esthetic evaluation. Intraclass correlation coefficient (ICC) was used to test intra- and inter-examiner reliability; a value of 0.7 was set as the minimum acceptable level of reliability. Results: When comparing the 2 sets of observations (FF and CF), the ICC ranged from 0.654 to 0.823. Concerning gender assessment bias, the ICC ranged from 0.438 to 0.686 and from 0.722 to 0.788 for males and females, respectively. Concerning inter-examiner reliability, ICC for questions 2-7 ranged from 0.448 to 0.644 and from 0.659 to 0.817 at T0 and T2, respectively. Conclusions: The removal of external facial features provides subtle differences on the esthetic assessment of UCLP patients. Moreover, based on our data, examiners' gender differences may affect esthetic assessment of UCLP patients. Despite the subjectivity of esthetic judgments, a reliable, validated and reproducible scoring protocol should consider the influence of gender differences on 3D esthetic assessment of UCLP patients.