Adolescents with orofacial clefts: understanding their experiences.

OBJECTIVE: To understand the experience of young people with orofacial clefts regarding life as an adolescent. METHODS: Descriptive, qualitative study, developed in a Brazilian public and tertiary hospital, a reference center in the care of patients with craniofacial anomalies and related syndromes, between February and April 2019. The sample was defined by theoretical saturation. The following inclusion criteria were established: age between ten and 19 years old and having previously operated on orofacial cleft (lip and/or palate). Individuals with fissure associated with syndromes or other malformations were excluded. Data collection was performed through semi-structured interviews, which were audio recorded and transcribed in full. The trigger element was: how has it been for you to experience your adolescence? For the construction of the results, content analysis was used in the thematic modality. RESULTS: Seventeen adolescents participated. From the speeches, three categories were revealed: interacting socially, feeling supported, and experiencing and facing prejudice. CONCLUSIONS: The biopsychosocial and conflicting complexity that adolescents with orofacial clefts experience was noticed, as well as the importance of receiving support and establishing modalities of situational coping.

The emotional and social concerns of mothers of children with cleft palate: A cross-sectional study.

PROBLEM: Among birth defects, a cleft palate is one of the most common defects globally, with a prevalence rate of 1 in 700 live-born children per annum. Caring for a child with cleft palate is considered a challenge for mothers who provide around clock care for these children. Despite this, there is limited knowledge of their experiences and needs particularly in low resource settings. BACKGROUND: Previous literature emphasized that mothers of children with cleft palate face many challenges involving emotional distress, social isolation, and financial burden. QUESTION: To investigate the emotional and social concerns of mothers of children with Cleft Palate compared to mothers of children without cleft palate. METHODS: A comparative study design was conducted at the Jordanian Royal Medical Services using convenience sample of 312 mothers of children with and without cleft palate in Jordan. FINDINGS: There was a significant difference in the presence of anxious feelings between mothers of children with cleft palate and those mothers without cleft palate children. Within group comparison for mothers of children with cleft palate has shown a significant difference in their social concerns with regards to social support and child's future. DISCUSSION: Mothers of children with cleft palate reported significantly higher levels of sadness feelings. These emotions could be attributed to several factors, including the financial burden associated with medical treatment and interventions, the burden of caring for a child with special needs, the social stigma related to the child's appearance, the lack of public awareness, and the insufficient social support services. CONCLUSION: Mothers of children with Cleft Palate experience a considerable amount of emotional and social concerns which require urgent interventions.

Language abilities and associated risk factors of school-aged children with cleft lip and palate.

Previous research on children with cleft lip and palate (CLP) reported unequivocal findings with regard to language skills, with the majority suggesting persistent difficulties in early childhood. While expressive language deficits improved with age, receptive language skills were consistently lower than peers. Further study investigating the long term and persistent impact of language deficits amongst school-aged children with CLP is warranted. This was a cross-sectional study, aimed to determine the language abilities and explore the associated risk factors in Malay speaking children with CLP in Malaysia. Fifty-two children with CLP aged 7- to 12-year-old participated in this study. Language skills were assessed using the Malay Preschool Language Assessment Tool and the adapted Subway-School-age Language & Assessment Measures. Findings revealed that 14 (26.92%) school-aged children with CLP demonstrated language deficits. Children with CLP performed significantly poorer in reading comprehension (p = 0.031) and narrative (p = 0.026) skills. It was found that the age significantly influenced total receptive language score (ß = 0.421, p = 0.003) and total expressive language score (ß = 0.477, p = 0.000). Findings suggested that children with CLP may continue to have persistent language deficits into their school-age years. Recommendations for regular monitoring of language performance especially for those from younger age groups is warranted to help maximize school attainment.

Evaluation of Anxiety in Turkish Parents of Newborns with Cleft Palate with or Without Cleft Lip.

OBJECTIVE: (1) To compare anxiety between parents of newborns with cleft lip and palate (CLP), isolated cleft palate (CP), and healthy newborns and (2) to evaluate anxiety between parental dyads within these groups. DESIGN: A cross-sectional study. SETTING: University Hospital. PARTICIPANTS: Surveys were completed by 20 mothers and 20 fathers of newborns with CLP, 21 mothers and 21 fathers of newborns with CP, and 23 mothers and 23 fathers of healthy newborns (controls). MAIN OUTCOME MEASURE: The State-Trait Anxiety Inventory (STAI) assessed parental anxiety. Mothers of newborns with a cleft reported on concerns regarding cleft-related issues and facial appearance. RESULTS: State and trait anxiety were generally in the moderate range for parents of newborns with a cleft, while control parents had low state anxiety and moderate trait anxiety. Mothers of newborns with CP and CLP had significantly higher state and trait anxiety levels than control mothers (p < .05). Fathers of newborns with CLP had a higher state anxiety level than control fathers. When maternal and paternal anxiety was compared within the groups, only trait anxiety scores were significantly higher in mothers of newborns with CLP than that of fathers (p < .05). More than half of mothers of newborns with a cleft were concerned about their newborn's feeding, speech, and palate. CONCLUSIONS: Parents of children with a cleft may need psychological support in the early postnatal period. It is important for neonatal cleft team providers to help reduce parental anxiety and educate families about cleft care, with a focus on feeding.

Being Normal yet Different: A Qualitative Study on the Dualistic Experience of Living With Unilateral Cleft Lip and Palate.

The aim of the present study was to describe the experiences of young adults living with cleft lip and palate (CLP) and to explore potential gender differences.A descriptive qualitative study was designed involving semi-structured interviews. The interviews were analyzed using qualitative content analysis, as described by Graneheim and Lundman.A total of 9 women and 8 men, aged 22 to 26 years with UCLP.The main theme identified was: the duality of living with a cleft-being normal yet different, and 2 subcategories: "My cleft and me" and "My cleft and the World." The participants described themselves as normal yet different, both in relation to themselves and in relation to others. They also stated that gender norms regarding appearance affected their lives and how they saw the cleft.This study adds to the growing body of qualitative research on CLP. It highlights the dualistic experiences of feeling normal and different at the same time. The interviews indicated that this dualism was based on context and gender, showing the psychological complexity of an individual. The clinical implications of this study emphasizes the need of a person-centered care approach in the cleft care setting where the clinicians are aware of the potential dualistic experience that also may differ over time that individuals with cleft can experience. This can also help clinicians better understand and help patients reduce distress and strengthen positive coping mechanisms.

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.

"Challenges and concerns faced by parents of a group of Egyptian children with cleft lip/palate: a qualitative study".

BACKGROUND: Cleft lip and palate are the most common developmental anomalies that affect the mouth and related structures. They can both affect children physiologically, socially, and functionally and lead to psychological distress in their parents. The present study aims to understand the challenges parents of cleft lip and palate patients face in Egypt, elucidate how they cope with these challenges, and assess their concerns for the future. METHODS: For the present phenomenological qualitative exploration, the parents of cleft lip and palate patients attending the cleft care clinic were invited to participate in the study through face-to-face recruitment at the clinic. An interview guide about the research question was developed to include standardized open-ended questions providing a framework for structured discussions. The interviews were audio-recorded after obtaining written informed consent from participants then collected data were transcribed for data analysis. RESULTS: Of the 12 participants, there were nine mothers and three fathers. Their children's ages ranged from 1.5 years to 19 years and had different presentations of cleft lip and palate from unilateral cleft lip to complete bilateral cleft lip and palate. Feeding difficulty was one of the main challenges encountered by the parents. At the same time, fear of being subjected to bullying was the main concern for the future of their children. Six themes were noted that were continually reported: Health & Wellbeing; Parental emotions; Parental attitudes & behaviors; Financial aspects; Relationship aspects; and Career/Education. CONCLUSIONS: There were 4 factors that directly impacted the themes, namely: the type of cleft, gender of the child, gender role of the parent, and the age of the child impacted the parental concerns and the challenges faced under the influence of sociocultural beliefs and existing support systems.

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.

A protocol for a randomized-controlled trial to investigate the effect of infant sign training on the speech-language development in young children born with cleft palate.

BACKGROUND: Children born with a cleft palate with or without cleft lip (CP ± L) are known to be at risk for speech-language disorders that impact educational and social-emotional growth. It is hypothesized that speech-language intervention delivered before the age of 3 years could decrease the impact of CP ± L on speech-language development. Infant sign training in combination with verbal input expands the natural communication of young children including multimodal speech-language input (i.e., verbal and manual input) via caregivers who act as co-therapists. AIMS: To determine the effectiveness of infant sign training in 1-year-old children with CP ± L by comparing different interventions. METHODS & PROCEDURES: This is a two-centre, randomized, parallel-group, longitudinal, controlled trial. Children are randomized to either an infant sign training group (IST group), a verbal training group (VT group) or no intervention control group (C group). Caregivers of children who are assigned to the IST group or VT group will participate in three caregiver training meetings to practise knowledge and skills to stimulate speech-language development. Outcome measures include a combination of questionnaires, language tests and observational analyses of communicative acts. OUTCOMES & RESULTS: It is hypothesized that speech-language development of children with CP ± L will benefit more from IST compared with VT and no intervention. Additionally, the number and quality of communicative acts of both children and caregivers are expected to be higher after IST. CONCLUSIONS & IMPLICATIONS: This project will contribute to the development of evidence-based clinical practice guidelines regarding early speech-language intervention in children with CP ± L under the age of 3 years. WHAT THIS PAPER ADDS: What is already known on the subject Children with CP ± L are known to be at risk for speech-language delays that impact educational and social emotional growth. Given the limited scientific prove of the impact of early speech-language intervention, no standardized clinical practice guidelines are available yet for children with CP ± L under the age of 3 years. Early intervention in this population mostly focuses on improving verbal input via caregivers or professionals without including a multimodal language input. A growing scientific interest has been seen in the use of infant signs to support speech-language development and caregiver-child interaction in typically developing children and children with developmental delays. What this study adds to existing knowledge No evidence is yet available for the effectiveness and feasibility of early intervention based on infant sign training in combination with verbal input to improve speech-language skills in young children with CP ± L. The current project will investigate the effect of infant sign training on the speech-language development in this population. Outcome measures are compared with those of two control groups: verbal training only and no intervention. It is hypothesized that infant signs may support the intelligibility of verbal utterances produced by children with CP ± L. Improving children's intelligibility may increase the opportunities for these children to engage in early, frequent and high-quality interactions with their caregivers resulting in a richer social and linguistic environment. As a result, infant sign training may result in better speech-language skills compared with the control interventions. What are the potential or actual clinical implications of this work? If providing early intervention based on infant sign training is effective, there is the potential for improved speech-language outcomes in early childhood, resulting in increased speech intelligibility, increased well-being of the child and family and less need for speech-language therapy on the long-term. This project will contribute to the development of evidence-based clinical practice guidelines regarding early speech-language intervention in children with CP ± L under the age of 3 years.

'Sometimes I feel sad': A qualitative study on children's perceptions with cleft palate speech and language therapy.

BACKGROUND: Evidence-based speech therapy involves the integration of (1) the scientific evidence for therapy with (2) the perspectives of clinicians concerning therapy and (3) the perspectives of patients about therapy. The cleft literature has already paid attention to the first two cornerstones of evidence-based speech therapy. Much less is known about how children perceive cleft speech therapy. AIMS: The purpose of the current qualitative study was to investigate the perceptions, emotions and expectations of Flemish-speaking Dutch children with a cleft (lip and) palate (CP ± L), aged 5-12 years, with regard to the speech therapy they receive. In this study, a focus was made on speech therapy to eliminate compensatory cleft speech errors. METHODS & PROCEDURES: Six children with a CP ± L, aged between 5 and 12 years, were included in this study. Child-friendly semi-structured interviews were conducted using a participatory, art-based qualitative approach. This means that the 'play and puppets technique' and 'draw-write and photo-elicitation technique' were used to guide the children through the interviews. Data derived from these interviews were analysed using an inductive thematic approach. Trustworthiness of the data was achieved by applying researcher triangulation, negative case analysis and an audit trail. OUTCOMES & RESULTS: Analyses of the interviews revealed three major themes of importance to the children: (1) treatment values, (2) treatment practices and (3) treatment outcomes. Each theme was divided into different subthemes. The theme 'treatment values' consisted of the subthemes expectations and emotions around therapy and interference with daily living. Information flow, therapy content, confirmation and rewards, parents' attendance, therapy intensity, and homework were subthemes of the major theme 'treatment practices'. The theme 'treatment outcomes' was divided into two subthemes, namely speech improvement and peers' reactions. CONCLUSIONS & IMPLICATIONS: Most children had positive attitudes towards speech therapy: it was 'something they liked' and 'something fun'. If children had negative attitudes they were related to having a fear of making mistakes during therapy. Children had clear expectations of the purpose of speech therapy. Speech therapy should 'help' improve their speech and make it more understandable to others. The children in this sample made some suggestions to decrease the experienced burden related to speech therapy. The results of this study will help to better tailor speech therapy programmes to the needs and experiences of children with a CP ± L. WHAT THIS PAPER ADDS: What is already known on the subject Evidence-based speech therapy involves the integration of (1) the scientific evidence for therapy with (2) the perspectives of clinicians concerning therapy and (3) the perspectives of patients and their families about therapy. The cleft literature has already paid attention to the first two cornerstones of evidence-based speech therapy. Different studies investigated the perspectives of SLPs and parents with regard to cleft palate speech therapy. However, much less is known about the children's own experiences with and perceptions around this speech therapy. What this study adds to existing knowledge This study used a qualitative research design to investigate the perceptions, emotions and expectations of children with a cleft (lip and) palate, aged 5-12 years, with regard to the speech therapy they receive. Speech therapy needed to focus on the elimination of compensatory speech errors. This study provides knowledge on the speech therapy-related experiences of children with a cleft palate. What are the potential or actual clinical implications of this work? Children in this sample made some concrete suggestions to decrease the experienced burden related to cleft speech therapy, for example, integration of school work during therapy sessions and practising on the level of spontaneous speech. The results of this study help us to better tailor speech therapy programmes to the needs and experiences of children with a CP ± L.

The impact of cleft lip and palate on the quality of life of young children: A scoping review.

INTRODUCTION: Cleft lip and palate (CL/P) are among the most common congenital abnormalities. The purpose of the present study was to review the literature relating to the quality of life (QoL) in young patients with cleft lip and/or palate (CL/P) and to identify the specific aspect of QoL in young patients with CL/P that is mostly affected. Other associated variables within studies that may have an impact on QoL were also identified. MATERIALS AND METHODS: Systematic searches of PubMed, Scopus and Web of Science databases were conducted. Independent reviewers screened the title, abstract and full texts according to predetermined inclusion and exclusion criteria. Articles published in English from January 2012 to March 2022 reporting the QoL of non-syndromic young patients aged 7-18 years with CL/P were included. Review articles and articles reporting the psychological adjustment of parents or other family members with CL/P were excluded. RESULTS: 975 publications were identified, of which 20 studies met our inclusion criteria. The majority of studies reported that the CL/P condition has a negative impact on the QoL. Psychological health, functional well-being, socialemotional well-being and school environment are domains that are affected. Compared with typically developing young patients, those with CL/P had lower QoL scores even though QoL was assessed using different instruments across studies. The impact of CL/P on overall QoL scores varied by age but not gender or cleft type. CONCLUSION: Our reviews had shown the presence of CL/P negatively affects the QoL of young patients. Psychological health is the most affected QoL domain. Understanding the impacted domain will help in planning and delivering better health care for individuals with CL/P and reducing the stigma commonly associated with CL/P. Future studies should target intervention on psychological health and consider resilience factors towards positive adjustment.

Peer attitudes towards adolescents with speech disorders due to cleft lip and palate.

BACKGROUND AND AIMS: Individuals with speech disorders are often judged more negatively than peers without speech disorders. A limited number of studies examined the attitudes of adolescents toward peers with speech disorders due to a cleft lip with or without a cleft of the palate (CL ± P). Therefore, the aim of the present study was to investigate the attitudes of peers toward the speech of adolescents with CL ± P. METHOD: Seventy-eight typically developing adolescents (15-18 years, 26 boys, 52 girls) judged audio and audiovisual samples of two adolescents with CL ± P based on three attitude components, i.e., cognitive, affective, and behavioral. The degree of speech intelligibility was also scored by their peers. The study investigated whether the three attitudes were determined by speech intelligibility or appearance of an individual with CL ± P. Furthermore, the influence of knowing someone with a cleft, the age, and gender of the listeners on their attitudes were explored. RESULTS: A significantly positive correlation was found between the speech intelligibility percentage and the three different attitude components: more positive attitudes were observed when the speech intelligibility of the speaker was higher. A different appearance due to a cleft lip does not lead to more negative attitudes. Furthermore, boys seem to have more negative attitudes toward individuals with CL ± P compared to girls. CONCLUSION: This study provided additional evidence that peers show more negative attitudes toward adolescents with less intelligible speech due to CL ± P. Intervention should focus on changing the cognitive, affective, and behavioral attitudes of peers in a more positive direction and remove the stigma of patients with a cleft. Further research is needed to verify these results.

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.

HOW PRO'S CAN CONTRIBUTE TO WHAT MATTERS MOST TO PATIENTS WITH OROFACIAL CLEFTS.

An orofacial cleft (OC) is a congenital cleft that may affect the lip, alveolus, hard and soft palate. An OC impacts the individuals' appearance, psychosocial well-being and causes functional problems including feeding, dentition, growth of jaws, hearing and speech. The treatment of an orofacial cleft usually reaches from birth until 22 years of age or later. To understand and listen in an objective manner, OC specific Patient reported Outcome (PRO) and experience (PRE) measures are imperative. In patients with OCs many shared decision making moments exist from birth until adulthood. The aim is to understand what knowledge exists on PROs and PREs in patients with OCs, to formulate challenges to improve care to OC patients and how research on OCs needs to adapt. PROs and PREs need to be tailored to the individual with an OC. A framework with specific OC key domains including appearance, facial function and Health related Quality of Life exists. The current framework does not include the social network around the OC patients yet. However attention should be put on including family and community support into the framework. Also at an individual level more attention should be paid to enhancing experiences compared to impeding experiences. To create a better understanding traditional indicators and outcomes are combined with PROMs in a structured way. Challenges were identified that seek to improve our complete set of (PRO and PRE) instruments to provide better care to the individual with an OC and provide a voice so that good shared decision making is enabled. Thereby the individual with an OC is further empowered.

Relationship Between Stigma Experience and Self-Perception Related to Facial Appearance in Young Japanese Patients with Cleft lip and/or Palate.

OBJECTIVE: To examine the relationship between stigma experience related to facial appearance in Japanese youths with cleft lip and/or palate (CL/P) and their self-perception. DESIGN: A cross-sectional study. PARTICIPANTS: Sixty-nine Japanese youths with CL/P (11-18 years old). OUTCOME MEASURES: The participants' stigma experience in relation to facial appearance (measured with 7 single contextual scale items) and their self-perception (measured with 5 domain scores based on 30 perceptual items) were assessed using the Japanese version of the Youth Quality of Life Instrument-Facial Differences Module. Participants were categorized into high and low self-perception subgroups with a threshold of 1 standard deviation for each domain. The frequency of stigma experiences was compared between the following 2 subgroups: age, sex, cleft palate only versus other cleft, and high versus low self-perception. Correlations between the responses regarding stigma and all domain scores were examined. RESULTS: Sixteen percent of the participants reported experiencing stigma. Hearing others say something about their face occurred significantly more frequently in youths 15 to 18 years of age than in youths 11 to 14 years of age. Stigma frequency was not found to differ by sex or cleft type. Stigma experiences were significantly more frequent for youth with higher scores across negative self-perception domains as well as higher coping skills. Significant correlations were identified between responses regarding stigma items and all domain scores (r = 0.27-0.63, p < .05). CONCLUSIONS: It was found that stigma experiences related to facial appearance may influence negative self-perceptions of facial differences as well as higher coping skills among Japanese youths with CL/P.

A Multisite Study Investigating Child and Parent Proxy Reported Quality of Life in Children With Cleft Lip and/or Palate.

This observational, multisite cohort study explored health-related quality of life (HRQoL) in children with cleft lip and/or palate (CL/P), including interrater agreement and ratings for this group relative to clinical cutoff scores and published means for healthy and chronically ill children.Participants (338 children ages 8-10 years, 45.9% male and their parents, 82.0% female) across 6 sites completed the Pediatric Quality of Life Inventory Generic Core Scales (PedsQL).Intraclass correlation revealed poor interrater agreement for most HRQoL domains. Although ratings were generally higher than those expected for children with a chronic illness, child ratings were below healthy means for school functioning, and parent proxy ratings were below healthy means for all domains except physical functioning. Lower ratings consistent with chronic illness means were found for self-reported emotional and psychosocial functioning in children with cleft lip and palate (CLP), as well as parent proxy-reported emotional, school, and psychosocial functioning for children with cleft palate (CP). Scores were most likely to be in the clinical range for children with CP for social, school, and total functioning.Although parent proxy report provides important information about observed functioning, poor interrater agreement indicates that both child and parent proxy reported HRQoL should be included in outcomes assessment for CL/P. HRQoL ratings may be higher for children with CL/P compared to youth with other chronic illnesses, but psychosocial functioning may be negatively impacted when compared with healthy youth, particularly for emotional, social, and school functioning in children with CLP or CP.

COMT rs4818 less common allele is associated with psychological and psychiatric worse indicators in a cohort of individuals born with cleft lip and palate.

OBJECTIVE: Individuals born with cleft lip and palate may face difficulties in speech function, nutrition, facial aesthetics, and long-term care. These difficulties may increase the risk of psychological and psychiatric diseases. This work aimed to test if the variant allele of COMT was carried more frequently among individuals that have psychological and psychiatric outcomes within a cohort of patients born with cleft lip and palate. METHOD: DNA extraction from saliva of two hundred and fifteen individuals born with cleft lip with and/or palate and genotyping was performed, and the frequency of COMT rs4818 alleles was determined. The domain 'Psychological Function' of Cleft-Q™ was used to generate scores for analysis. The scores were computed, and differences in genotype or allele frequencies between individuals with psychological function scores 60 or above and 59 or below were compared. The history of psychiatric illness (family history of psychiatric disease or self-reported psychiatric illness) was registered. RESULTS: Genotype and allele frequencies were compared between individuals with and without a family history of psychiatric illness. Individuals with lower Psychological Function (Cleft-Q™) scores were more likely to be GG (P = .04) or carriers of allele G (P < .001). The reported psychiatric illness and positive family history of psychiatric illness were compared to COMT rs4818 allele and genotype frequencies of individuals without these indicators, and individuals with psychiatric illness and positive family history of psychiatric illness were more likely to carry allele G (P = .03 and P = .008, respectively). CONCLUSION: The study confirms previously suggested role of COMT rs4818 in psychiatric and psychological outcomes in a distinct cohort of patients born with cleft lip and palate.

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.

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.

Childhood Experiences and Perspectives of Individuals With Orofacial Clefts: A Qualitative Systematic Review.

OBJECTIVE: Children and adolescents with orofacial clefts may experience ongoing psychosocial impacts due to the continuous nature of cleft treatments, facial and dental differences, and speech and hearing difficulties. The aim of this qualitative systematic review was to better understand the experiences of children and adolescents with orofacial clefts. DESIGN: A systematic search strategy using PubMed, Embase, Emcare, Scopus, and Web of Science databases was performed to identify relevant qualitative studies evaluating the lived experience of children and adolescents with orofacial clefts from inception through to June 2021. Eligible studies were critically appraised using the Joanna Briggs methodology and a meta-aggregative approach. RESULTS: The search identified 2466 studies, with 13 found to meet the inclusion criteria. Extraction of 155 findings resulted in 27 categories, which were meta-aggregated into 7 overarching synthesized findings. These 7 core findings included aspects of child experience and findings that enhanced or impeded child experience at the individual, family, and community levels. CONCLUSIONS: Factors that impeded child experience at the individual, family, and community levels were more pronounced than factors that enhanced their experience among children and adolescents with orofacial clefts. Further initiatives are needed to provide support to individuals, families, and school communities to enhance children's experience of orofacial cleft during the formative childhood and adolescent years.