Introduction: A two-stage surgical approach, starting with the soft palate, has been practiced in the Gothenburg cleft team for unilateral cleft lip and palate (UCLP) since decades. To improve the velopharyngeal function, a modified soft palate closure (SPC) technique was introduced. Clinical observations did however indicate that the novel technique resulted in a deviant residual cleft shape, potentially contributing to future compromised dental arch form. Aim: This study aimed to compare residual cleft dimensions (area, width and length) between different SPC techniques and at various age, in individuals born with UCLP. Material and methods: The sample consisted of three groups of children with non-syndromic UCLP: 1. 27 individuals who had original SPC and hard palate closure (HPC) at 3 years (GBG3) 2. 37 individuals who had a modified SPC and HPC at 2 years (GBGmod) 3. 29 individuals who had original SPC and HPC at 2 years (GBG2). Residual cleft area, length and width were measured on digitised cast models using Blender software. Multiple linear regression analysis was used to compare the groups. Results: Larger residual cleft dimensions (area and length) were found for GBGmod compared to both GBG3 (p < 0.001, p = 0.009) and GBG2 (p = 0.009, p = 0.009). In addition, GBGmod had increased cleft width in comparison with GBG3 and GBG2 in quartile 1. Conclusion: The modified SPC technique resulted in larger residual clefts in comparison with the groups operated with the original SPC method. Timing differences between GBG3 and GBG2 are likely one explanation of cleft width variations anteriorly.

Children with cleft lip and/or palate (CL/P) often experience developmental issues, including language delay, learning difficulties, and attention issues. The aim of the study was to assess potential difficulties in children with CL/P and to determine the necessity of ongoing psychological support. Eighty-two children treated for CL/P, 29 5-year-olds and 53 10-year-olds, participated together with their parents in the study. The Strengths and Difficulties Questionnaire (SDQ) was used to assess difficulties and strengths. Proxy reports were used for the 5-year-olds, while self-reports and proxy reports were utilized for the 10-year-olds. Patients who were identified as requiring further support were offered psychological interventions. Parents of 5-year-olds and 10-year-olds with CL/P estimated significantly more difficulties compared with parents of children in the normal population. Thirty-nine percent of the parents estimated that their child had minor, moderate or severe difficulties. However, 10-year-olds with CL/P didn't perceive any difference in difficulties compared with preadolescents in the normal population. The 10-year-olds estimated significantly less emotional difficulties than children in the normal population. Nevertheless, 41% of the 10-year-olds reported that they overall had difficulties, either minor or severe, in one or more areas. There were significantly moderate correlations between parental and child reports on SDQ. According to the results of the standardized psychological screenings, 25.6% of children with CL/P required additional psychological support. One in 4 children with CL/P needs psychological support. Standardized psychological interview-based follow-ups for patients with CL/P can contribute to more information regarding the patient's needs rather than only using a questionnaire.

AIM:To longitudinally investigate speech production (consonant proficiency, consonant errors and perceived velopharyngeal competence) in 17 internationally adopted (IA) children with unilateral cleft lip and palate (UCLP) at three time points.METHOD:Consonant proficiency (percent consonants correct, consonant inventory) and number and type of consonant errors were calculated based on blind phonetic transcriptions of words from the Swedish Test of Articulation and Nasality (SVANTE). Velopharyngeal competence was perceptually rated by three blinded experienced speech-language pathologists at the ages of 3, 5 and 7-8 years.RESULTS:A significant positive development of speech production was found, although most children still scored very low for consonant proficiency at the age of 7-8 compared with normative values: the median for percent consonants correct was 79.7 and many children still had persisting cleft-related and developmental consonant errors. At the age of 7-8, almost half of the children were rated as having a competent velopharyngeal function and only three as having an incompetent velopharyngeal function.CONCLUSION:Persisting speech difficulties at school age in IA children with UCLP were found in the present study, which is one of the very few longitudinal studies. Our results highlight the need for detailed follow-up of speech production in clinical settings. Speech disorders may have a severe impact on a child's intelligibility and participation with peers, and there is a need for more studies investigating the actual everyday effect of the difficulties found.

Objective: In 2005, the number of internationally adopted children (IAC) with cleft lip and palate (CLP) from China increased globally, followed by an increase in the number of surgical complications. The aim was therefore to evaluate the craniofacial growth of children with unilateral CLP adopted from China and compare with non-adopted peers (NA). Methods: Cephalograms taken at 10 and 13 were analysed for the SNA, SNB, ANB, NAPg and NSBa angles. Surgical data were extracted from the medical records. Results: The rate of secondary cleft-related surgery including secondary alveolar bone grafting (SABG) was higher in the IAC group than in the native groups. At age 10, the IAC exhibited higher SNB and NAPg and lower ANB values than the NA-Mod group and a similar pattern was observed at age 13. The IAC group demonstrated significantly lower SNA and ANB and higher NAPg values than the NA-Orig group at age 10. At age 13, lower SNA and ANB and higher SNB, NAPg, and NSBa values were observed in the IAC group compared with the NA-Orig group. Limitations: Craniofacial growth is not finalized at age 13 years, why additional studies are needed to determine long-term outcomes. Conclusions: Results from this novel study present unfavorable craniofacial growth at age 10 and 13 years of age among IAC with UCLP and indicate an increased need for future orthognathic surgery compared with NA peers.

Objective The aim was to evaluate dental arch relation and craniofacial growth for individuals born with unilateral cleft lip and palate (UCLP), who had two-stage palatal surgery, with hard palate closure (HPC) at the ages of 3 or 8 years. Design Longitudinal cohort study. Setting Ceft lip and palate team in Gothenburg, Sweden. Patients The inclusion criteria were nonsyndromic individuals born with complete UCLP who were consecutively operated according to two different surgical protocols; soft palate closure at 6 months followed by hard palate closure at the age of 3 years (HPC3) or at the age of 8 years (HPC8). In this evaluation, 28 individuals had HPC3 and 59 individuals had HPC8. Internventions The main outcome, longitudinal series of dental casts and lateral radiographs from the ages of 5, 10, 16, and 19 years, were evaluated using GOSLON Yardstick and cephalometric analysis. Results At the age of 10 years, 78% of the individuals with HPC3 demonstrated GOSLON scores of 1 and 2 and 86% in HPC8. At the age of 19 years, 54% of the individuals exhibited GOSLON scores of 1or 2 when compared with 74% in HPC8. A statistical significant difference for SNA was observed at the age of 5 years (P  =  .004), with a lower SNA in HPC3, but not at the ages of 10, 16 and 19 years. At the final age, SNA was 75.2° for HPC3 and 76.8° for HPC8. Conclusions The decrease in age for HPC did not have an adverse effect on long-term dental arch relationship or craniofacial growth.

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.

This national registry-based study compares surgical procedures, demography, and concurrent medical conditions, in internationally adopted and Swedish-born children with cleft lip and/or palate until the age of five years. Data on the cleft type and gender for 331 internationally adopted children and 2064 Swedish-born children born from 2007 to 2018, were extracted from the registry and analyzed. Data on surgical procedures performed in Sweden and concurrent medical conditions and were collected for internationally adopted children and Swedish-born children with unilateral or bilateral cleft, born 2007-2013. A higher prevalence of unilateral and bilateral clefts (p < 0.0001), as well as a predominance of male patients with unilateral clefts (p = 0.0025), were identified among the internationally adopted children compared with children born in Sweden. Differences in the concurrence of other medical conditions in internationally adopted children versus Swedish-born infants were non-significant. Primary palatal surgeries performed in Sweden were significantly delayed for the adopted group. More secondary palatal surgeries such as speech improving surgery and palatal re-repair were needed for internationally adopted children (p < 0.0001) until age five. Conclusions: The Swedish CLP Registry provided national coverage of the CL/P cohort. Internationally adopted children exhibited a predominance of more severe cleft types, a predominance of males, delayed primary palatal surgery and increased need for secondary surgeries before age five.

Summary 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.

BACKGROUND:Delayed hard palate closure in unilateral cleft lip and palate (UCLP) patients show on a safe surgical method and good speech outcome, however, occurrence of orally retracted articulation before hard palate closure at 8 years. The aim of this study was to describe surgical and speech outcome in UCLP patients closing the hard palate at 3 years.METHODS:A consecutive of 28 participants were operated with Gothenburg two-stage method including soft palate closure at 6 months and hard palate at 3 years. Surgical and speech outcome were evaluated. Recordings of sentences and spontaneous speech at 5, 10, 16, and 19 years were analyzed blindly and independently by three speech-language pathologists. Compensatory articulation, hypernasality, hyponasality, weak pressure consonants, and nasal air leakage were evaluated on ordinal four-point and intelligibility and perceived velopharyngeal function on three-point scales.RESULTS:Long-term follow-up revealed a safe surgical method. Articulation disorders were present in 25-30% at 5-year but largely not later. About 20% had incompetent velopharyngeal function at 5 years but none at 19 years. Most participants were well intelligible after 5 years. Hard palate closure at 3 years indicated less occurrence of orally retracted articulation compared with a cohort who had hard palate closure at 8.2 years.CONCLUSIONS:Long-term, follow-up of individuals with UCLP after Gothenburg two-stage palate closure including closure of the soft palate closure at 6 months and hard palate at 3 years of age shows a safe surgical method and indicates less retracted oral articulation compared with hard palate closure at 8 years.

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.

Abstract Background The objective of the Swedish cleft lip and palate registry (CLP registry) is to promote quality control, research and improvement of treatment, by the comparison of long-term results. The aim was to compare data from the CLP registry among the six treatment centres, regarding data on surgery and speech outcomes at 5 years of age. Methods The participants were 430 children born in Sweden from 2009 to 2014, with cleft palate with or without cleft lip and without known syndromes and/or additional malformations. The number of primary and secondary palatal surgeries up to 5 years of age, timing of the last primary palatal surgery, percentage consonants correct, percentage non-oral speech errors and perceived velopharyngeal competence at 5 years were assessed. Multivariable binary logistic regression adjusted for sex and cleft type was used to compare results between the six centres. Results At one centre (centre 4), the palate was closed in one to three stages, and at the remaining centres in one or two stages. At centre 4, more children underwent a higher number of palatal surgeries, and the last primary palatal surgery was performed at a higher age. Children in centre 4 were also less likely to achieve ≥86% correct consonants (OR = 0.169, P = < 0.001), have no non-oral speech errors (OR = 0.347, P = < 0.001), or have competent or marginally incompetent velopharyngeal competence (OR = 0.244, P = < 0.001), compared to the average results of the other centres. No clear association between patient volume and speech outcome was observed. Conclusions The results indicated the risk of a negative speech result if the last primary palatal surgery was performed after 25 months of age. Whether the cleft in the palate was closed in one or two stages did not affect speech outcome. The Swedish CLP registry can be used for open comparisons of treatment results to provide the basis for improvements of treatment methods. If deviating negative results are seen consistently at one centre, this information should be acted upon by further investigation and analysis, making changes to the treatment protocol as needed.

Objective This single-centre retrospective cohort study aimed to evaluate secondary alveolar bone grafting (SABG) and identify the factors associated with failure. Methods Individuals born with alveolar cleft with or without cleft palate who had SABG consecutively between 2004-2006 and 2011-2013 were enrolled in this study. After the exclusion of 20 cases due to poor quality radiographs, 115 patients (50 girls and 65 boys) who had undergone 131 SABG procedures were included. According to a modification of the Bergland Scale (mBS), the alveolar bone level was assessed using occlusal films obtained 3 months after SABG. Data on factors plausible for SABG failure were collected from patient records, photographs, cast models, and presurgical occlusal radiographs. The Mantel Haenszel Chi-Square test was used to test the possible impact of these factors on the mBS scores. Results A total failure (mBS score of 4) was observed in 9% of the cases. The alveolar bone level correlated with cleft laterality (P = 0.039), alveolar cleft extension (P = 0.033), age at SABG (P = 0.007), root developmental stage (P = 0.021), and oral hygiene (P = 0.007). Conclusion Secondary alveolar bone grafting failure was correlated with a bilateral alveolar cleft, absence of an initial partial alveolar bridge, higher age, increased root developmental stage, and poor oral hygiene. Efforts to achieve optimal oral hygiene are recommended to improve outcomes, particularly when SABG is performed in individuals with a bilateral cleft, increased alveolar cleft extension, or at higher ages.

Objective: To compare speech outcome and self-reported speech and communicative ability (SOK) in young adults treated with one-stage (OS) or two-stage (TS) palatal repair. Furthermore, to compare with normative data on individuals without cleft lip and palate and to study the relationship between patients' and experts' judgments. Design: A cross-sectional group comparison study with long-term follow-up. Participants: Patients born with unilateral cleft lip and palate treated at 2 cleft centers; 17 with OS at 14 months and 25 with TS, soft palate repair at 7 months and hard palate repair at 6.2 years. Pharyngeal flap surgery was performed in 53% (OS) and 24% (TS) of patients. Main Outcome Measure(s): Speech characteristics were blindly assessed by speech and language pathologist from audio recordings, SOK at 19 years of age. Results: No group differences were found. Although the occurrence of nasality symptoms was low in both groups, only 60% (OS)/65% (TS) were assessed with competent velopharyngeal function (VPC). Articulation proficiency (percentage of consonants correct [PCC]) was 91%/97%, the /s/-sound specifically 87%/91%. Good intelligibility was found in 91%/87%. Patient opinion was in agreement with norms and significantly associated with intelligibility (rs = 0.436, P < .01), PCC (rs = -0.534, P < .01), and correct /s/ (rs = -0.354, P < .05). Conclusions: No differences in speech outcome were related to operation method. The low prevalence of VPC was not clearly reflected in nasality symptoms. Patient opinion was related to articulation and intelligibility. A higher burden of care in terms of pharyngeal flap surgery was seen after the OS technique.

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 (r s) 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 (r s = 0.59), pessimism (r s = 0.59), and low optimism (r s = −0.56). Men’s appearance-related distress was significantly associated with low active coping (r s = 0.35), low use of emotional support (r s = 0.29), denial (r s = 0.39), behavioral disengagement (r s = 0.41), and pessimism (r s = 0.28). The only significant gender interaction reflected greater impact of optimism in reducing appearance-related distress for women (β = −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 r s 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.

Summary Objectives The aims were to compare cleft and non-cleft canine position, to find predictive factors for canine position and to assess surgically exposed canines. Materials/Methods One hundred forty-eight individuals, born 1978–2005, with total unilateral cleft lip and palate (including Simonart’s band <5 mm) treated in Gothenburg, were included. Canine angulation as well as vertical and horizontal position were assessed on panoramic radiographs (PAN) taken at the age of 10. Plausible predictive factors were registered from PAN, cast models and medical records. Data on spontaneous eruption or surgical exposure were available for 88 patients. Cleft- and non-cleft side was compared using paired t-test and Fisher’s exact test. Multiple stepwise regression analysis and logistic regression analysis were used to detect possible predictors for cleft canine position. Results The cleft canine angulation was 29.3 ± 13.1 degrees (mean ± standard deviation) versus 7.6 ± 8.2 degrees on the non-cleft side (p < 0.001). Cleft canines were higher positioned and located closer to the midline compared to non-cleft canines (p < 0.001). Age for both hard palate closure and bone grafting, cleft lateral agenesis as well as transposition were associated with canine position. Cleft canines that required surgical exposure (28%) had an increased angulation and were higher positioned than spontaneously erupted canines (p < 0.001). Limitations The shortcomings were the retrospective design and incomplete assessment of the buccal–palatal canine position on 2D images. Conclusions/Implications With increased age for bone grafting, decreased age for hard palate closure and transposition, an association with abnormal canine position was found. A rigorous monitoring of cleft canine eruption is, therefore, advocated.

Aims: To determine reliability and predictive validity of the 5-year-olds’(5YO) Index and GOSLON Yardstick in 119 patients born with unilateral cleft lip and palate at 5, 7/8, 10, 15/16, and 19 years. Methods: Five hundred thirty-four dental study models were appraised by 2 teams in 2 centers, twice in each center. Intrateam and interteam reliability in scoring the models was calculated using κ. Dental arch prediction rates were calculated as the proportion of models remaining in the same category (good–scores 1 and 2; fair–score 3; poor–scores 4 and 5) over time. Results: Intrateam and interteam κ statistics ranged from 0.74 to 0.89 and from 0.74 to 0.81, respectively. The 5YO Index and GOSLON Yardstick at 5 years produced almost identical results. The prediction rate of 19-year-old (n = 106) outcome was >80% for those in groups 1 and 2 at 5 years, while for those in groups 4 and 5 prediction was poor (<40%). Prediction of groups 4 and 5 remained poor until 10 years when it increased to 77%. At 15/16 years prediction rate was 93% for those in groups 4 and 5. Prediction of cases in group 3 was very poor at all ages. Conclusions: These results question the predictive value of “poor” dental arch relationships before 10 years of age. However, the predictive value of “good” dental arch relationship scores over time is good in all age groups. This has implications for audit policies to predict facial growth outcomes.

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.

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.

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.

Background The objective of the Swedish cleft lip and palate (CLP) registry is to promote quality control, research and improvement of treatment, by comparison of the long-term results of surgery, orthodontics and speech from all six Swedish CLP centres. The purpose of the study was to investigate the coverage and reporting degree of the Swedish CLP registry, and to describe the design of the registry and discuss questions of reliability and validity of the data included. Methods All six Swedish CLP centres participate in the registry. All children in Sweden with cleft lip and/or cleft palate, born from 2009 onwards, are included in the registry. Baseline data such as cleft type (ICD-10 diagnosis), heredity, birth weight and additional deformities and/or syndromes, as well as pre-surgical treatment, are recorded at first visit. Data on surgical treatment are recorded continuously. Treatment outcome regarding dentofacial development and speech are recorded at follow-ups at 5, 10, 16 and 19 years of age. Data on dentofacial development are also recorded 1 year after orthognathic surgery. In addition, data on babbling and speech are recorded at 18 months of age. Coverage degree and reporting degree of surgery was assessed by comparison with registrations in the Swedish Central patient registry. Reporting degree of orthodontic and speech registrations at 5 years of age was assessed by comparison with registrations at baseline. Results The average coverage degree for children born 2009 to 2018 was 95.1%. For cleft-related surgeries, the average reporting degree was 92.4%. Average reporting degree of orthodontic registrations and speech registrations at age 5 years was 92 and 97.5% respectively. Conclusion In order to achieve valid and reliable data in a healthcare quality registry, the degree of coverage and reporting needs to be high, the variables included should be limited and checked for reliability, and the professionals must calibrate themselves regularly. The Swedish CLP registry fulfils these requirements.