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.

Core outcomes for orofacial clefts: reconciling traditional and ICHOM minimum datasets.

OBJECTIVE/DESIGN/SETTING: This retrospective study sought voluntary participation from leading cleft centres from Europe and Brazil regarding core outcome measures. The results of this study would inform the debate on core outcome consensus pertaining to the European Reference Network for rare diseases (ERN CRANIO) and achieve a core outcome set for cleft care providers worldwide. INTERVENTION/METHOD: Five orofacial cleft (OFC) disciplines were identified, within which all of the International Consortium of Health Outcomes Measurement (ICHOM) outcomes fall. One questionnaire was designed for each discipline and comprised 1. the relevant ICHOM's outcomes within that discipline, and 2. a series of questions targeted to clinicians. What core outcomes are currently measured and when, did these align with the ICHOM minimum, if not how did they differ, and would they recommend modified or additional outcomes?. RESULTS: For some disciplines participants agreed with the ICHOM minimums but urged for earlier and more frequent intervention. Some clinicians felt that some of the ICHOM standards were compatible but that different ages were preferred and for others the ICHOM standards were acceptable but developmental stages should be preferred to absolute time points. CONCLUSION/IMPLICATIONS: Core outcomes for OFC were supported in principle but there are differences between the ICHOM recommendations and the 2002 WHO global consensus. The latter are established in many centres with historical archives of OFC outcome data, and it was concluded that with some modifications ICHOM could be moulded into useful core outcomes data for inter-centre comparisons worldwide.

Differences in analysis and treatment of upper airway obstruction in Robin sequence across different countries in Europe.

The goal of this study was to explore the availability of diagnostic and treatment options for managing upper airway obstruction (UAO) in infants with Robin Sequence (RS) in Europe. Countries were divided in lower- (LHECs, i.e., PPP per capita < $4000) and higher-health expenditure countries (HHECs, i.e., PPP per capita ≥ $4000). An online survey was sent to European healthcare professionals who treat RS. The survey was designed to determine the availability of diagnostic tools such as arterial blood gas analysis (ABG), pulse oximetry, CO2 analysis, polysomnography (PSG), and sleep questionnaires, as well as to identify the used treatment options in a specific center. Responses were received from professionals of 85 centers, originating from 31 different countries. It was equally challenging to provide care for infants with RS in both LHECs and HHECs (3.67/10 versus 2.65/10, p = 0.45). Furthermore, in the LHECs, there was less access to ABG (85% versus 98%, p = 0.03), CO2 analysis (45% versus 70%, p = 0.03), and PSG (54% versus 93%, p < 0.01). There were no significant differences in the accessibility concerning pulse oximetry, sleep questionnaires, home saturation monitoring, nasopharyngeal tubes, Tuebingen plates, and mandibular distraction.    Conclusion: This study demonstrates a large difference in available care for infants with RS throughout Europe. LHECs have less access to diagnostic tools in RS when compared to HHECs. There is, however, no difference in the availability of treatment modalities between LHECs and HHECs. What is Known: • Patients with Robin sequence (RS) require complex and multidisciplinary care. They can present with moderate to severe upper airway obstruction (UAO). There exists a large variety in the use of diagnostics for both UAO treatment indications and evaluations. In most cases, conservative management of UAO in RS is sufficient. Patients with UAO that persist despite conservative management ultimately need surgical intervention. To determine which intervention is best suitable for the individual RS patient, the level of UAO needs to be determined through diagnostic testing. • There is a substantial variation among institutions across Europe for both diagnostics and treatment options in UAO. A standardized, internationally accepted protocol for the assessment and management of UAO in RS could guide healthcare professionals in the timing of assessment and indications to prevent escalation of UAO. Creating such a protocol might be a challenge, as there are large financial differences between countries in Europe (e.g., health expenditure per capita in purchasing power parity in international dollars ranges from $600 to over $8500). What is New: • There is a substantial variation in the availability of objective diagnostic tools between European countries. Arterial blood gas analysis, CO2 analysis and polysomnography are not equally accessible for lower-healthcare expenditure countries (LHECs) compared to higher-healthcare expenditure countries (HHECs). These differences are not only limited to availability; there is also a difference in quality of these diagnostic tools. Surprisingly, there is no difference in access to treatment tools between LHECs and HHECs. • There is national heterogeneity in access to tools for diagnosis and treatment of RS, which suggests centralization of health care, showing that specialized care is only available in tertiary centers. By centralization of care for RS infants, diagnostics and treatment can be optimized in the best possible way to create a uniform European protocol and ultimately equal care across Europe. Learning what is necessary for adequate monitoring could lead to better allocation of resources, which is especially important in a low-resource setting.

The First Hybrid International Educational Comprehensive Cleft Care Workshop.

OBJECTIVE: Describe the first hybrid global simulation-based comprehensive cleft care workshop, evaluate impact on participants, and compare experiences based on in-person versus virtual attendance. DESIGN: Cross-sectional survey-based evaluation. SETTING: International comprehensive cleft care workshop. PARTICIPANTS: Total of 489 participants. INTERVENTIONS: Three-day simulation-based hybrid comprehensive cleft care workshop. MAIN OUTCOME MEASURES: Participant demographic data, perceived barriers and interventions needed for global comprehensive cleft care delivery, participant workshop satisfaction, and perceived short-term impact on practice stratified by in-person versus virtual attendance. RESULTS: The workshop included 489 participants from 5 continents. The response rate was 39.9%. Participants perceived financial factors (30.3%) the most significant barrier and improvement in training (39.8%) as the most important intervention to overcome barriers facing cleft care delivery in low to middle-income countries. All participants reported a high level of satisfaction with the workshop and a strong positive perceived short-term impact on their practice. Importantly, while this was true for both in-person and virtual attendees, in-person attendees reported a significantly higher satisfaction with the workshop (28.63 ± 3.08 vs 27.63 ± 3.93; P = .04) and perceived impact on their clinical practice (22.37 ± 3.42 vs 21.02 ± 3.45 P = .01). CONCLUSION: Hybrid simulation-based educational comprehensive cleft care workshops are overall well received by participants and have a positive perceived impact on their clinical practices. In-person attendance is associated with significantly higher satisfaction and perceived impact on practice. Considering that financial and health constraints may limit live meeting attendance, future efforts will focus on making in-person and virtual attendance more comparable.

Classification Systems of Cleft Lip, Alveolus and Palate: Results of an International Survey.

OBJECTIVE: This study aimed to identify commonly used classification systems by cleft providers around the world, including the perceived indications and limitations of each system. DESIGN: A cross-sectional survey. PARTICIPANTS: A total of 197 registrants from three international cleft/craniofacial meetings. INTERVENTIONS: Participants were sent a web-based questionnaire concerning cleft classification systems. MAIN OUTCOME MEASURES: Frequency of commonly used classification systems, their perceived indications and limitations. RESULTS: A total of 197 respondents from 166 different centers completed the questionnaire. Healthcare professionals from all disciplines responded, with the most frequent respondents being plastic surgeons (38.1%), maxillofacial surgeons (28.4%) and orthodontists (23.9%). Eighteen different classification systems were in use. The most frequently used systems were the International Statistical Classification of Diseases and Related Health Problems (ICD-10) (35.5%), LAHSHAL (34.0%), and Veau (32.5%) classification systems. Most respondents (32.5%) indicated that anatomical and morphological characteristics are essential components of a classification system. However, respondents indicated that their current classification systems lacked sufficient description of cleft extension and severity. CONCLUSIONS: Great variety in the use of classification systems exists among craniofacial specialists internationally. The results recommend the usage of the LAHSHAL classification of OFCs, due to its comprehensiveness, relatively high implementation rate globally, convenience of usage and complementarity with the ICD-10 system. Moreover, it can overcome deficiencies inextricably linked to ICD-10, such as incapacity to describe laterality and clefts of the alveolus. More international exposure to the merits of using the LAHSHAL classification system would be highly recommended.

Influence of Gender, Dispositional Optimism, and Coping Strategies on Appearance-Related Distress Among Swedish Adults With Cleft Lip and Palate.

OBJECTIVE: To investigate the influence of gender, dispositional optimism, and coping strategies on appearance-related distress among individuals with unilateral cleft lip and palate (UCLP). DESIGN: Cross-sectional design with self-report questionnaires analyzed primarily with Spearman correlations (rs) and multivariate regression analyses. SETTING: A tertiary cleft center in Sweden. PARTICIPANTS: Eighty individuals with UCLP born 1966 to 1986. The mean age for men (n = 50) and women (n = 30) was 38.8 and 37.4 years, respectively. MAIN OUTCOME MEASURES: The Derriford Appearance Scale 24 measured appearance-related distress, the Life Orientation Test-Revised, short version measured dispositional optimism and pessimism, and the Coping Orientation to Problems Experienced, short version included 14 coping strategies. RESULTS: Women had higher appearance-related distress than men, which was significantly (P < .05) related to self-blame (rs = 0.59), pessimism (rs = 0.59), and low optimism (rs = -0.56). Men's appearance-related distress was significantly associated with low active coping (rs = 0.35), low use of emotional support (rs = 0.29), denial (rs = 0.39), behavioral disengagement (rs = 0.41), and pessimism (rs = 0.28). The only significant gender interaction reflected greater impact of optimism in reducing appearance-related distress for women (ß = -0.06). CONCLUSIONS: This study showed that high levels of dispositional optimism decrease appearance-related distress, particularly for women. The coping strategies used differed between men and women, and the results suggest that both gender and psychosocial facto rs need to be considered in regard to appearance-related distress among individuals with UCLP in both clinical and research settings. A possible way to decrease distress is to strengthen positive coping strategies and dispositional optimism.

Scores of the Cleft Hearing, Appearance and Speech Questionnaire (CHASQ) in Swedish Participants With Cleft lip and/or Cleft Palate and a Control Population.

OBJECTIVE: The primary aim of this study was to investigate whether there was any difference in scores of the Cleft Hearing, Appearance and Speech Questionnaire (CHASQ) between patients with cleft lip and/or cleft palate (CL/P) and a control population. The second aim was to compare CL/P and control population scores in this study with a British norm CL/P population. DESIGN: Single-site, cross-sectional study with an age-matched control population. SETTING: Participants were recruited from a hospital, a school, and a sports club. They answered the CHASQ in the hospital or at home. PARTICIPANTS: Sixty-four participants with CL/P (7-19 years of age) and a control population of 56 participants without CL/P (9-20 years of age). MAIN OUTCOME MEASURE: CHASQ. RESULTS: There was no statistically significant difference in satisfaction with cleft-related features between the CL/P and the control population. Participants with CL/P were significantly more satisfied with non-cleft-related features than the control population. Cleft Hearing, Appearance and Speech Questionnaire scores were also similar to earlier established British normative data of a CL/P population. CONCLUSION: The results indicated that children and young people with CL/P were as satisfied with their appearance, hearing, and speech as children and young people without CL/P. Swedish CHASQ scores were also similar to British scores.

Comparison of Corresponding Scores From the Cleft Hearing Appearance and Speech Questionnaire (CHASQ) and CLEFT-Q in Swedish Patients With Cleft Lip and/or Palate.

OBJECTIVE: The primary aim of this study was to compare corresponding scores between 2 existing cleft-specific patient-reported outcome measures (PROMs)-Cleft Hearing Appearance and Speech Questionnaire (CHASQ) and CLEFT-Q. The second aim of the study was to investigate patient opinion on the 2 PROMs. DESIGN: Cross-sectional questionnaire study. SETTING: Participants were recruited from a University Hospital. They answered CHASQ and CLEFT-Q either in the hospital or at home. PARTICIPANTS: Thirty-three participants with cleft lip and/or palate, aged 10 to 19 years. MAIN OUTCOME MEASURE: CHASQ and CLEFT-Q. RESULTS: The CHASQ scores and the corresponding CLEFT-Q scores on appearance correlated significantly. Corresponding scores regarding speech did not correlate significantly. A majority, 15 (58%) participants, answered that they liked CLEFT-Q more than CHASQ, 18 participants (69%) thought CHASQ was easier to complete, and 19 (76%) thought CLEFT-Q would better inform health care professionals. CONCLUSION: Both instruments showed strengths and limitations. Clinicians will have to consider each instrument's respective qualities when choosing to implement either PROM.

A gender perspective on appearance-related concerns and its manifestations among persons born with unilateral cleft lip and palate.

Cleft lip and palate (CLP) affects a person's facial appearance and can cause appearance-related distress. Appearance-related distress and its manifestations are not fully understood. Women with CLP tend to experience a higher degree of distress as well as undergo more aesthetic revisions than men. The aim of this study was to investigate if self-reported appearance-related gender differences occur among adults born with unilateral CLP using the Derriford Appearance Scale 24 (DAS24). One hundred sixty two consecutive patients, 107 men and 55 women, with unilateral CLP and no associated syndromes treated at the same hospital were asked to answer the DAS24. A mixed methods approach was used to analyse the questionnaires both quantitively and qualitatively. Fifty men and 30 women participated, the results showed that the aspects of appearance considered most disturbing was cleft-related among both genders, most common  in regards to the nose. Three themes were found: acceptance, cleft features, and general appearance issues. The quantitative part showed that women reported higher appearance-related social anxiety and avoidance than men. This study demonstrated that appearance-related distress is present in both genders; however, women expressed more distress than men. Moreover, non-cleft as well as cleft features are of importance for self-reported dissatisfaction.

Prioritizing Cleft/Craniofacial Surgical Care after the COVID-19 Pandemic.

BACKGROUND: It is anticipated that in due course the burden of emergency care due to COVID-19 infected patients will reduce sufficiently to permit elective surgical procedures to recommence. Prioritizing cleft/craniofacial surgery in the already overloaded medical system will then become an issue. The European Cleft Palate Craniofacial Association, together with the European Cleft and Craniofacial Initiative for Equality in Care, performed a brief survey to capture a current snapshot during a rapidly evolving pandemic. METHODS: A questionnaire was sent to the 2242 participants who attended 1 of 3 recent international cleft/craniofacial meetings. RESULTS: The respondents indicated that children with Robin sequence who were not responding to nonsurgical options should be treated as emergency cases. Over 70% of the respondents indicated that palate repair should be performed before the age of 15 months, an additional 22% stating the same be performed by 18 months. Placement of middle ear tubes, primary cleft lip surgery, alveolar bone grafting, and velopharyngeal insufficiency surgery also need prioritization. Children with craniofacial conditions such as craniosynostosis and increased intracranial pressure need immediate care, whilst children with craniosynostosis and associated obstructive sleep apnea syndrome or proptosis need surgical care within 3 months of the typical timing. Craniosynostosis without signs of increased intracranial pressure needs correction before the age of 18 months. CONCLUSIONS: This survey indicates several areas of cleft and craniofacial conditions that need prioritization, but also certain areas where intervention is less urgent. We acknowledge that there will be differences in the post COVID-19 response according to circumstances and policies in individual countries.

Chronic obstructive pulmonary disease and comorbidities' influence on mortality in non-small cell lung cancer patients.

Background: In Denmark, lung cancer is the most common cause of cancer-related death and chronic obstructive pulmonary disease (COPD) is the most common comorbidity in patients with non-small cell lung cancer (NSCLC). The aim of this study was to investigate the influence of COPD and other common comorbidities on NSCLC mortality. Methods: Patients (n = 534) diagnosed with NSCLC at Aalborg University Hospital from 2008-2010 were included retrospectively in this study. Patient records were assessed and the population was dichotomized in COPD and non-COPD subgroups. Comorbidities i.e., ischemic heart disease, hypertension, diabetes mellitus, apoplexia, former malignancy, interstitial lung disease and psychiatric comorbidity were registered and a comorbidity count were calculated. Survival was assessed with log-rank test and uni- and multivariate regression analysis were performed for COPD-status and comorbidity count adjusting for age, gender, BMI, smoking exposure, cancer stage, method of treatment and eastern cooperative cancer group (ECOG) performance status score. Results: Of 534 NSCLC patients included, 470 were divided into COPD and non-COPD subgroups, 70% with COPD (329/470) and 30% without COPD (141/470). Only 32.5% of the patients in the COPD-group had previously diagnosed COPD. Log-rank test did not show statistically significant difference in survival between the COPD and non-COPD groups (p = .215). Multivariate Cox regression analysis did not show statistically significant association between overall 5-year mortality and the presence of COPD (HR-adj = 0.808, 95% CI = 0.612; 1.068) or other comorbidities (HR-adj = 1.101, 95% CI = 0.979; 1.237) when adjusted for age, BMI, gender, smoking exposure, ECOG performance status, treatment and TNM-stage. Conclusion: Our findings do not suggest that COPD nor other common comorbidities are significantly associated with higher mortality in NSCLC patients.

The Psychosocial Impact of Cleft in a Western Australian Cohort Across 3 Age Groups.

BACKGROUND: Management of a cleft of the lip and/or palate (CL/P) involves a multidisciplinary team approach lasting from birth to potentially postskeletal maturity. This condition is complex, with both medical and psychosocial implications that may place individuals with a cleft at higher risk of developing psychosocial problems. METHODOLOGY: A self-administered questionnaire was completed by a sample from the Western Australian cleft population comprising 3 age groups: child (n = 100), adolescent (n = 101), and adult (n = 158). RESULTS: Public speaking, being photographed, special relationships, and participation in school were identified as the areas most impacted by having a cleft. Hearing and speech were reported to have a higher importance than facial and dental appearance. Participants rated support given to them by their parents as the most important, with high ratings for treatment providers. For teasing, the impact of cleft was significantly higher among participants with cleft lip and palate for both the adolescent and adult age groups. There was little significant difference by gender across the variables, which suggests that males are just as likely to require support as females. CONCLUSION: The impact of a cleft across multiple psychosocial domains needs to be recognized and addressed as part of craniofacial team care across age groups.

Adult Narratives of the Psychosocial Impact of Cleft in a Western Australian Cohort.

BACKGROUND: Living with a cleft condition involves many years of multidisciplinary therapy, treatment, and surgical intervention. This complex process may have both physical and psychosocial long-term impacts. There is limited evidence of the psychosocial impact of cleft on individuals in Australia. AIM: To obtain an understanding of the adult patient perception of having a cleft and explore the impact of the condition on their lives. DESIGN: Qualitative methodologies at one case study site in Western Australia. POPULATION OF INTEREST: Adults with a cleft lip and/or palate (CL/P) who had received treatment at Princess Margaret Hospital, the only treatment center for cleft in Western Australia. METHODOLOGY: Individual in-depth semi-structured qualitative interviews were conducted and recorded and transcribed for analysis. RESULTS: Fifteen adults with a CL/P. Two main themes were identified: lived experience (with 3 subthemes: normality, support networks, and impacts in later life) and advice to others. The majority of participants reported social rejection and isolation, which occurred mostly at school and in some instances at home, but did not often last into adulthood. The greatest protective factor was the support of family and friends, which if missing during their childhood, was desired and appreciated in adulthood. CONCLUSION: Adults with CL/P may require psychosocial support from their cleft team including referral to clinicians in adult services. There is also the need for earlier psychological interventions and social programs to support those with appearance-related social difficulties.

Together I Can! Joint Attention Boosts 3- to 4-Year-Olds' Performance in a Verbal False-Belief Test.

Effects of joint attention were addressed on 3- to 4-year-olds' performance in a verbal false-Belief Test (FBT), featuring the experimenter as co-watcher rather than narrator. In two experiments, children (N = 183) watched a filmed-FBT jointly with a test leader, disjointed from a test leader, or alone. Children attending jointly with a test leader were more likely to pass the FBT compared with normative data and to spontaneously recall information indicating false-belief understanding, suggesting that joint attention strengthens the plausibility of the FBT and renders plot-critical information more salient. In a third experiment (N = 59), results were replicated using a typical, image-based FBT. Overall findings highlight the profound impact of experimenter as social context in verbal FBTs, and link recall of specific story features to false-belief understanding.

Application of a new patient-reported outcome measure in orofacial clefts: An exploratory study in two countries.

Patients with repaired cleft lip and/or palate may vary in satisfaction with their treatment. The exploratory study investigated the satisfaction of patients with orofacial clefts, and their parents with cleft treatment outcomes using the Cleft Hearing, Appearance and Speech Questionnaire (CHASQ). The study included 29 Vietnamese and 27 Estonian patients aged ≥ 7 years with repaired cleft lip and/or palate. The CHASQ was translated into Vietnamese and Estonian. The questionnaire was completed independently by patients and their parents. There were nine items in the CHASQ associated with the cleft (Factor 1) and six items less associated with the cleft (Factor 2). Significant moderate positive correlations were related to Factor 1. Vietnamese patients self-rated lower than Estonian patients in most of the items except speech. The agreement between patients and parents varied from low to moderate positive correlations in the features associated with the cleft, and mainly in the Vietnam sample. Vietnamese patients were less satisfied than Estonian patients. CHASQ is an easy tool to evaluate patients' satisfaction with hearing, appearance, and speech.

Adult-Specific Life Outcomes of Cleft Lip and Palate in a Western Australian Cohort.

BACKGROUND: People with a cleft of the lip and/or palate (CL/P) differ from their peers due to their facial appearance, hearing and speech difficulties, and the significant time spent attending appointments and recovering from surgical interventions. These differences may impact life outcomes including occupation, income, education, relationships, psychosocial health issues, and lifestyle choices. METHODOLOGY: A self-administered questionnaire was posted to 338 former and current patients of the Cleft Lip and Palate Unit of Princess Margaret Hospital (PMH), Perth, Western Australia. RESULTS: Completed questionnaires were returned by 158 former and current patients. In comparison to the Australian Bureau of Statistics, study participants attained equivalent highest education levels, full-time annual income levels, occupational categories, employment rates, and home ownership levels. They did not marry later and demonstrated positive health-related lifestyle behaviors. However independent living was significantly delayed, and the number of romantic relationships, marriages, and children was lower, with separation/divorce rates also being lower. A key finding was that 78% of participants self-reported that they experienced at least 1 psychosocial health issue and more than half experienced anxiety and/or depression. CONCLUSION: When comparing the sociological outcomes for the study participants, the psychosocial outcomes were the areas of most concern. Further investigation is required to determine the causes for the high self-reported rates of anxiety and/or depression found in this study.

Body Esteem in a Western Australian Cleft Lip and/or Palate Cohort Across 3 Age Groups.

OBJECTIVES: To determine if patients with cleft lip and/or palate (CL/P) from a Western Australian (WA) cohort were more dissatisfied with their body esteem than a normative non-cleft cohort, and identify demographic variables that may have significant associations with body esteem. DESIGN: Questionnaire study using the Body-Esteem Scale (BES) and Cleft Research Questionnaire (CRQ). PARTICIPANTS: Self-selected participants from a Western Australian CL/P population across 3 age groups (n=359). MAIN OUTCOME MEASURES: The BES is comprised of 3 factors: BE-Appearance, BE-Weight and BE-Attribution. Study mean BES factor scores were compared to normative non-cleft scores. Regression analysis was used to determine significant associations within each age group between BES factor scores and CRQ variables of: gender, self-reported body weight category, cleft type and importance of facial appearance rating. RESULTS: Study mean BE-Attribution scores were significantly lower than the normative scores and significantly lower than the mean BE-Appearance and BE-Weight factor scores within the same age groups of this study. Having a cleft type of lip and palate, being overweight, and placing a high importance on facial appearance had significant negative associations with BES scores. Maintaining a normal body weight and placing a lower level of importance on facial appearance had significant positive associations. Gender had no significant associations. CONCLUSION: In this study, the attribution aspect of body esteem had a greater negative impact on patients than their appearance and body weight. This has important implications for clinical treatment and support of patients.

Female and Male Differences in Academic Achievement in Individuals With Cleft: A Population-Based Register Study.

OBJECTIVE: The focus of this study was to determine if there is any significant difference in academic achievement for girls and boys with a cleft compared to the general population of Swedish students at graduation from compulsory school. DESIGN: A retrospective population-based study using data obtained from the Swedish Medical Birth Register that was linked to the Swedish School-Grade Register. PARTICIPANTS: Two hundred seventy girls and 241 boys with cleft palate (CP), 222 girls and 429 boys with cleft lip (CL), and 299 girls and 531 boys with cleft lip and palate (CLP) were compared with the compulsory school population comprising 609,397 girls and 640,007 boys. MAIN OUTCOME MEASURES: (1) Odds of receiving the lowest grade and reduced odds in receiving high grades in Mathematics, English, and Swedish. (2) grade point average (GPA). RESULTS: In all 3 subject grades, for boys with cleft there was no difference when compared to the male population. Girls with cleft were similar to their peers with a few exceptions. Girls with CLP had lower Math grades, and girls with CP had lower Math, English, and Swedish grades. Girls with CP and CLP achieved a significantly lower GPA in comparison to the female population and boys with CP and CL achieved lower GPAs in comparison to the male population. CONCLUSIONS: This study indicates that educational outcomes for girls with cleft are more negatively affected than for boys with cleft.

Opportunities and Challenges in Establishing a Cohort Study: An Example From Cleft Lip/Palate Research in the United Kingdom.

BACKGROUND: Cleft lip and/or palate (CL/P) is one of the most common birth conditions in the world, but little is known about its causes. Professional opinion remains divided as to which treatments may be the most beneficial for patients with CL/P, and the factors that contribute to psychological adjustment are poorly understood. The use of different methodological approaches and tools plays a key role in hampering efforts to address discrepancies within the evidence base. A new UK-wide program of research, The Cleft Collective, was established to combat many of these methodological challenges and to address some of the key research questions important to all CL/P stakeholders. OBJECTIVE: To describe the establishment of CL/P cohort studies in the United Kingdom and to consider the many opportunities this resource will generate. RESULTS: To date, protocols have been developed and implemented within most UK cleft teams. Biological samples, environmental information, and data pertaining to parental psychological well-being and child development are being collected successfully. Recruitment is currently on track to meet the ambitious target of approximately 9800 individuals from just more than 3000 families. CONCLUSIONS: The Cleft Collective cohort studies represent a significant step forward for research in the field of CL/P. The data collected will form a comprehensive resource of information about individuals with CL/P and their families. This resource will provide the basis for many future projects and collaborations, both in the United Kingdom and around the world.