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.

Hormonal support in women with Asherman syndrome does not lead to better outcomes: A randomized trial.

Purpose: The purpose of the study was to investigate if adjuvant hormones after successful adhesiolysis lead to a reduction in spontaneous recurrence of adhesions and influence reproductive outcomes. Methods: A single-blind randomized controlled trial comparing administration of oral estrogen (the usual care group) with not giving estrogen (no estrogen) in women after successful adhesiolysis for Asherman syndrome. Women were included between September 2013 and February 2017, with a follow-up of 3 years to monitor recurrences and reproductive outcomes. Analyses were based on an intention to treat analyses. This study was registered under NL9655. Results: A total of 114 women were included. At 1 year, virtually all patients (except 3) were either having a recurrence or were pregnant. Women who did not receive estrogen did not have more recurrences of adhesions in the first year prior to pregnancy (66.1% in the usual care group, 52.7% in the no-estrogen group, p = 0.15). Of the women in usual care, 89.8% got pregnant within 3 years, and 67.8% got a living child; this was 83.6% and 60.0%, respectively, in the no-estrogen group (p = 0.33 and p = 0.39, respectively). Conclusion: Usual care does not lead to better outcomes as compared with not giving exogenous estrogen but is associated with side effects.

Long-term longitudinal follow-up of individuals with UCLP after Gothenburg twostage palate closure: surgical and speech outcomes.

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.

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.

Efficacy of Oral Nifedipine, Naproxen, or Placebo for Pain Relief During Diagnostic Hysteroscopy in an Office Setting: A Randomized Pilot Study.

STUDY OBJECTIVE: To compare nifedipine, naproxen, or placebo for pain relief during diagnostic hysteroscopy. DESIGN: Double-blind, randomized controlled pilot study. SETTING: University hospital. PATIENTS: Women scheduled for office diagnostic hysteroscopy (n = 60). INTERVENTIONS: Women received nifedipine (2 tablets of 10 mg), naproxen (2 tablets of 250 mg), or placebo (2 tablets of 500 mg lactose) 30 to 60 minutes prior to hysteroscopy. MEASUREMENTS AND MAIN RESULTS: Sixty patients were enrolled in the study (21 in the nifedipine group, 19 in the naproxen group, and 20 in the placebo group). The median pain scores during hysteroscope insertion, measured on a Visual Analog Scale (VAS), were 1 (interquartile range (IQR) 0-0), 2 (0-4) and 1 (0-1) in the nifedipine, naproxen and placebo group, respectively (P,14). The median VAS scores during hysteroscopy were 5 (IQR 2-7), 5 (4-8) and 5 (3-7) in the nifedipine, naproxen and placebo group, respectively (P,73). The median VAS scores immediately after hysteroscopy were 2 (IQR 0-4), 3 (0-6) and 3 (1-5) in the nifedipine, naproxen and placebo group, respectively (P,40). The median VAS scores 30 minutes after hysteroscopy were 1 (IQR 0-2), 1 (0-1) and 1 (0-2) in the nifedipine, naproxen and placebo group, respectively (P,63). Hysteroscope insertion failed in 1 case (naproxen group) because of cervica`l stenosis (P,32). Flushes, fatigue and vertigo, 30 minutes after the procedure, were significantly more prevalent in the nifedipine group compared to the naproxen (p < .001, p,03, p,03, respectively) and the placebo group (p < .001, p,01, p,01, respectively). Palpitations occurred only in the nifedipine group (p < .001). The day after the procedure, the headache was most prevalent in the nifedipine group compared to the naproxen group (p,001) and the placebo group (p,001). CONCLUSION: In our pilot study, pain relief and success rates for office diagnostic hysteroscopy were not significantly different between nifedipine, naproxen, and placebo. Nifedipine was associated with more, albeit tolerable, side-effects.

Long-term Outcome for Two-Stage Palatal Closure With Different Timings for Hard Palate Surgery: Craniofacial Growth and Dental Arch Relation.

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.

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.

A national registry-based study of surgery and demography comparing internationally adopted and children born in Sweden with cleft lip and/or palate.

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.

The link between intrauterine adhesions and impaired reproductive performance: a systematic review of the literature.

BACKGROUND: Intrauterine adhesions (IUAs) are one of the main reproductive system diseases in women worldwide. Fusion between the injured opposing walls leads to partial-to-complete obliteration of the cavity and/or cervical canal. The main clinical manifestations in case of IUAs are menstrual disturbances, cyclic pain and reproductive disorders. The reproductive outcomes of women with IUAs remain limited and inefficient compared to women without IUAs, even after adhesiolysis. An exact understanding of the underlying mechanisms and processes to explain the compromised reproductive performance and outcomes in case of IUAs are lacking. METHODS: A systematic literature review of MEDLINE-PubMed (1966 to January 2022) and EMBASE (1974 to January 2022) was performed following Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. Studies were included if they reported underlying causes, related mechanisms and processes to explain the association between IUAs and impaired reproductive performance, pregnancy and obstetric complications. RESULTS: After an extensive review of the literature, 58 articles were identified reporting underlying mechanisms to explain the association between IUAs and impaired fertility. Intrauterine scarring influences the process of fertilization, reproductive performance and ultimately reproductive outcome. IUAs can disturb the cervico-utero-tubal sperm transport and result in an avascular and unresponsive endometrium with decreased receptivity and thickness. Abnormal decidualization and abnormal trophoblastic infiltration leads to placental attachment disorders. Moreover, the risk for premature delivery, intrauterine fetal growth restriction and fetal anomalies is increased in case of IUAs. CONCLUSION: The impact of IUAs on reproductive performance, even after adhesiolysis, is becoming more apparent. The postulated mechanisms to explain the association are related to sperm transport, embryo implantation and placentation. Prevention, by preserving the basal layer of the endometrium is essential. Effective and evidence-based strategies for the prevention of endometrial injury and formation of IUAs, are urgently needed.

Persisting speech difficulties at 7-8 years of age - a longitudinal study of speech production in internationally adopted children with cleft lip and palate.

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.

Inter-centre comparison of data on surgery and speech outcomes at 5 years of age based on the Swedish quality registry for patients born with cleft palate with or without cleft lip.

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.

Predictive factors for secondary alveolar bone graft failure in patients with cleft alveolus.

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.

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.

Speech Outcome and Self-Reported Communicative Ability in Young Adults Born With Unilateral Cleft Lip and Palate: Comparing Long-Term Results After 2 Different Surgical Methods for Palatal Repair.

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.

Live births after Asherman syndrome treatment.

OBJECTIVE: To study the reproductive outcomes after surgical management of women with Asherman syndrome (AS). DESIGN: Cohort study. SETTING: International referral hospital for women with AS. PATIENT(S): A total of 500 women who were diagnosed with and treated for AS between January 2003 and December 2016 and followed for a minimum of 2 years. INTERVENTION(S): Hysteroscopic adhesiolysis using conventional instruments with concomitant fluoroscopy as a guidance method. MAIN OUTCOME MEASURE(S): Live birth rate. RESULT(S): Of the 500 women included in the cohort, 569 pregnancies were achieved within 3 years after surgery. The miscarriage rate was 33.0%, and the live birth rate was 67.4%. Age, the causal procedure, and at least one miscarriage after adhesiolysis strongly predicted the outcome of a live birth. CONCLUSION(S): The overall take-home newborn rate was 67.4% after adhesiolysis in women with AS. Women with AS who are relatively young, with a first-trimester procedure preceding AS, and with low grades of adhesions and no miscarriage after adhesiolysis have the best chance of a newborn delivery.

Predictive factors for canine position in patients with unilateral cleft lip and palate.

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.

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.

Reliability and Predictive Validity of Dental Arch Relationships Using the 5-Year-Olds' Index and the GOSLON Yardstick to Determine Facial Growth.

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.

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.

Coverage, reporting degree and design of the Swedish quality registry for patients born with cleft lip and/or palate.

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.