Ethical Challenges for Plastic Surgery in Treating Internationally Adopted Children With Cleft Lip and Palate.

A large number of children with cleft lip and palate (CLP) were adopted to Sweden in the last decade, mainly from China. Most of the children arrived with unoperated palates and at later ages than earlier years. This article aims to present an overview of ethical challenges within the practice of international adoption of children with CLP from the perspective of plastic surgery in a welfare health care system. An overview of CLP treatment is presented, followed by a normative discussion and ethical analysis using the 4 principles of Beauchamp and Childress: non-maleficence, beneficence, autonomy, and justice. The following themes and subthemes were analyzed: the search for normality and the potential challenge of being adopted and having CLP-treatment autonomy of the child and future preferences, adoptive parents' expectations of plastic surgery, the journey of the adoptee and the adoptive parents; and general issues-reconstructive possibilities and consequences of CLP in the country of origin, information to the adoptive parents, health care needs, and reconstructive possibilities for children with CLP in the receiving country. Clinical implications are discussed, and suggestions for future research are provided.

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.

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.

Apical root resorptions in girls with Turner syndrome: a controlled longitudinal study.

OBJECTIVES: To study, longitudinally, the development of apical root resorptions (ARRs) in Turner syndrome (TS) and to correlate these to the karyotype and orthodontic treatment. MATERIALS AND METHODS: Thirty girls with TS participated in the study, mean age 10.8 years (6.6-23.4) at the first registration (T1), and 14.3 years (9.2-25.2) at the second registration (T2). Forty girls without TS, orthodontically untreated, served as controls. ARR was diagnosed in panoramic radiographs, and root/crown ratios of the lower permanent first molar were measured at T1 and T2 with a mean follow-up period of 42 (11-89) months. RESULTS: During the follow-up period, ARR was seen in 40% of all TS patients and in 2.5% in the control group. The majority displayed ARR on the distal root of the mandibular first permanent molars (30%). ARR at T2 was seen in 56% of 45,X and isochromosome karyotype and 21% of every other TS karyotype patients. AAR was seen in 5 out of 12 TS patients with orthodontic treatment. No statistically significant differences in root/crown ratios between T1 and T2 were found. LIMITATIONS: The number of patients studied is limited, thus possible differences might be disguised for this reason. Spatial projection errors in panoramic radiograph recordings might have influenced measurement, resulting in an underestimation of ARR. CONCLUSIONS: There is a higher risk for ARR in girls with TS and probably the risk is therefore probably also higher during orthodontic treatment. Thus, if treated, frequent radiographic follow-ups should be taken during the treatment. PROTOCOL: The protocol was not published before trial commencement.

Scandcleft randomized trials of primary surgery for unilateral cleft lip and palate: comparison of dental arch relationships and dental indices at 5, 8, and 10 years.

BACKGROUND AND TRIAL DESIGN: The Scandcleft intercentre study evaluates the outcomes of four surgical protocols (common method Arm A, and methods B, C, and D) for treatment of children with unilateral cleft lip and palate (UCLP) in a set of three randomized trials of primary surgery (Trials 1, 2, and 3). OBJECTIVES: To evaluate and compare dental arch relationships of 5-, 8-, and 10-year-old children with UCLP after four different protocols of primary surgery and to compare three dental indices. The results are secondary outcomes of the overall trial. METHODS: Study models taken at the ages of 5 (n = 418), 8 (n = 411), and 10 years (n = 410) were analysed by a blinded panel of orthodontists using the Eurocran index, the 5-year-olds' (5YO) index, and the GOSLON Yardstick. Student's t-test, Pearson's correlation, chi-square test, and kappa statistics were used in statistical analyses. RESULTS: The reliability of the dental indices varied between moderate and very good, and those of the Eurocran palatal index varied between fair and very good. Significant correlations existed between the dental indices at all ages. No differences were found in the mean 5-, 8-, and 10-year index scores or their distributions within surgical trials. Comparisons between trials detected significantly better mean index scores in Trial 2 Arm C (at all ages) and in Trial 1 Arm B (at 5 and 10 years of age) than in Trial 3 Arm D. The mean Eurocran dental index scores of the total material at 5, 8, and 10 years of age were 2.50, 2.60, and 2.26, and those of the 5YO index and GOSLON Yardstick were 2.77, 2.90, and 2.54, respectively. At age 10 years, 75.8% of the patients had had orthodontic treatment. CONCLUSIONS: The results of these three trials do not provide evidence that one surgical method is superior to the others. The reliabilities of the dental indices were acceptable, and significant correlations existed between the indices at all ages. The reliability of the Eurocran palatal index was questionable. TRIAL REGISTRATION: ISRCTN29932826.

Lateral and Frontal Cephalometric Measurements in a Cohort With Saethre-Chotzen Syndrome.

OBJECTIVE: Descriptions of the craniofacial morphology in Saethre-Chotzen syndrome (SCS) are primarily based on case reports or visual assessments of affected families. The aim of this study was to compare cephalometric measurements of the craniofacial skeleton in a cohort of individuals with SCS and age- and sex-matched individuals without craniofacial anomalies. DESIGN: Retrospective case series. PATIENTS: Eight girls and 4 boys with SCS (age range, 7.0-19.2 years). METHODS: Cephalometric measurements were performed using lateral and frontal cephalograms. RESULTS: Most of the individuals with Saethre-Chotzen syndrome exhibited lower values for SNA, SNB, s-n and s-ar, while their NSL/NL, NSL/ML, NL/ML, and n-s-ba values were higher than the respective mean reference values for healthy individuals. In comparison with age- and sex-matched individuals without craniofacial anomalies, the individuals with SCS showed higher values for the maxillary and mandibular angular measurements, as well as for the menton midline angle. CONCLUSIONS: This sample of 12 unrelated individuals with SCS is the largest collected to date for cephalometric measurements. We found that the syndrome is associated with bimaxillary retrognathism, posterior maxillary and mandibular inclination, neutral sagittal relation as well as a tendency toward an open vertical skeletal relation, a short and flattened skull base, and facial asymmetry, as compared to individuals without the syndrome.

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.

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.

Scandcleft randomized trials of primary surgery for unilateral cleft lip and palate: impact of maxillary dental agenesis on craniofacial growth and dental arch relationship in 8 year olds.

BACKGROUND: It is suggested that dental agenesis affects maxillary protrusion and dental arch relationship in children with unilateral cleft lip and palate (UCLP). In addition, an association between the need for orthognathic surgery and dental agenesis is reported. AIM: The aim was to study the impact of maxillary dental agenesis on craniofacial growth and dental arch relationship in 8-year-old children with UCLP. SUBJECTS AND METHODS: The sample consisted of individuals with UCLP from Scandcleft randomized trials. The participants had available data from diagnosis of maxillary dental agenesis as well as cephalometric measurements (n = 399) and GOSLON assessment (n = 408) at 8 years of age. RESULTS: A statistically significant difference was found for ANB between individuals with agenesis of two or more maxillary teeth (mean 1.52°) in comparison with those with no or only one missing maxillary tooth (mean 3.30° and 2.70°, respectively). Mean NSL/NL was lower among individuals with agenesis of two or more maxillary teeth (mean 9.90°), in comparison with individuals with no or one missing maxillary tooth (mean 11.46° and 11.45°, respectively). The number of individuals with GOSLON score 4-5 was 47.2% in the group with two or more missing maxillary teeth and 26.1% respectively 26.3% in the groups with no or one missing maxillary tooth. No statistically significant difference was found in the comparison between individuals with no agenesis or with agenesis solely of the cleft-side lateral. CONCLUSION: Maxillary dental agenesis impacts on craniofacial growth as well as dental arch relationship and should be considered in orthodontic treatment planning.

Scandcleft trial of primary surgery for unilateral cleft lip and palate: Craniofacial cephalometrics at 8 years.

BACKGROUND: The Scandcleft trial is a randomized controlled trial that includes children with unilateral cleft lip and palate where registrations are standardized and therefore provides the opportunity to describe craniofacial characteristics in a very large sample of patients. OBJECTIVES: The aim of this study was to describe craniofacial growth and morphology in a large study sample of 8-year-old children with unilateral cleft lip and palate (UCLP); before orthodontic treatment and before secondary alveolar bone grafting; and to compare the cephalometric values with age-matched non-cleft children from previous growth studies to identify the differences between untreated cleft- and non-cleft children. MATERIALS: There are 429 eight-year-old UCLP patients in the Scandcleft study group. A total of 408 lateral cephalograms with a mean age of 8.1 years were analysed. Cephalometric analyses were performed digitally. The results from three previously published growth studies on non-cleft children were used for comparison. RESULTS: Cephalometric analyses showed a large variation in craniofacial morphology among the UCLP group. In general, they present with significant maxillary retrusion and reduced intermaxillary relationships compared to the age-matched non-cleft children. In addition, the vertical jaw relationship was decreased, mainly due to decreased maxillary inclination. The upper and lower incisors were retroclined. It can be expected that these differences will increase in significance as the children age. CONCLUSION: Results from this study provide proposed norms for the young UCLP before any orthodontic treatment and can be valuable for the clinician in future treatment planning. TRIAL REGISTRATION: ISRCTN29932826.

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.

Scandcleft randomized trials of primary surgery for unilateral cleft lip and palate: occlusion in 8-year-olds according to the Modified Huddart and Bodenham index.

BACKGROUND: The Scandcleft international multicenter study is a prospective clinical trial of the long-term outcome after four different surgical protocols for palatal closure in patients born with unilateral cleft lip and palate (UCLP). This paper is one of a series of follow-up studies in 8-year olds. OBJECTIVES: To evaluate the dental occlusion of 8-year-old patients after four different protocols of primary surgery for UCLP. TRIAL DESIGN: Ten cleft centres in five countries tested three different surgical procedures for primary palatal repair in three parallel trials (Arms B, C, and D) against a common procedure (Arm A). METHODS: Initially 448 children born with non-syndromic UCLP were included in the project. At 8 years of age, 428 children remained in the study. Dental casts of 411 patients (270 boys, 141 girls), mean age 8.1 years (range 7.0-10.0) were taken. The casts were blindly assessed with the Modified Huddart and Bodenham (MHB) index by four orthodontists. The main outcome measures were anterior (+2 to -6) and posterior (0 to -8) mean scores. Comparisons were made with previous data in 5-year-olds. RESULTS: The inter- and intra-examiner reliability was good to excellent (0.75-0.90; 0.73-0.97), respectively. The mean total scores varied from -7.09 (Trial 2C) to -10.13 (Trial 3D). The mean anterior scores varied from -1.75 (Trial 2C) to -3.18 (Trial 1A). The mean posterior cleft-side scores varied from -4.32 (Trial 1B) to -5.21 (Trial 3D) and the mean non-cleft-side scores varied from -0.88 (Trial 2C) to -2.40 (Trial 3A). No significant differences were found within the trials. A significant difference was found between Trials 2 and 3 (Arm C/D) for the total score (P = 0.004). CONCLUSIONS: There was no evidence of clinically significant differences in occlusion between the two surgical methods in each trial or between the trials. All mean scores showed more negative values in 8-year-olds compared with previously reported values in 5-year-olds. TRIAL REGISTRATION: ISRCTN29932826.

Validation of reported dentoalveolar relationships in the Swedish Quality Registry for Cleft Lip and Palate.

OBJECTIVES: The present study validated data that had been reported to the Swedish Quality Registry for Cleft Lip and Palate (CLP) under new requirements from 2016, when use of the 5-year-old (5YO) and the Modified Huddart and Bodenham (MHB) indices for rating occlusion in children born with unilateral CLP (UCLP) was introduced. MATERIALS AND METHODS: The sample included blinded study casts (n = 97) and photos (n = 4) of 5-year-old children who had been born with UCLP in 2009-2011 and were enrolled at one of six cleft centres in Sweden. Fourteen orthodontists from the centres assessed the patients (n = 101) using the 5YO and the MHB indices. Median 5YO and MHB scores of the 14 assessments were compared with original registry data (n = 61). Each centre devised code keys to protect the identities of their patients in the registry. RESULTS: Interrater agreement among the 14 orthodontists was good for the 5YO index (quadratic-weighted kappa: 0.72-0.92) and the MHB index (intraclass correlation coefficient: 0.991-0.994). Comparisons of median 5YOs for each identifiable child with their registry data (n = 61) found total agreement for 70.5 per cent. Comparisons between median MHBs and registry data showed very good or good agreement in 93.4 per cent of the cases. LIMITATIONS: Two teams lost their code keys, which reduced the sample to 61 patients. CONCLUSIONS: The dentoalveolar outcome data in the CLP registry was trustworthy. There was good agreement among the Swedish cleft teams assessing the 5YO and MHB indices in children born with UCLP at age 5 years.

Scandcleft randomized trials of primary surgery for unilateral cleft lip and palate: maxillary growth at eight years of age.

OBJECTIVES: To assess differences in craniofacial growth at 8 years of age according to the different protocols for primary cleft surgery in the Scandcleft project. DESIGN AND SETTING: Prospective, randomized, controlled clinical trial (RCT) involving 10 centres, including non-syndromic Caucasians with unilateral cleft lip and palate (UCLP). In Trial 1, a common surgical method (1a) with soft palate closure at 3-4 months of age and hard palate closure at 12 months of age was tested against similar surgery but with hard palate repair at 36 months (delayed hard palate closure) (1b). In Trial 2, the common method (2a) was tested against simultaneous closure of both hard and soft palate at 1 year (2c). In Trial 3, the common method (3a) was tested against hard palate closure together with lip closure at 3 months of age and soft palate closure at 1 year of age (3d). Participants were randomly allocated by use of a dice. Operator blinding was not possible but all raters of all outcomes were blinded. SUBJECTS AND METHODS: The total number of participating patients at 8 years of age was 429. Lateral cephalograms (n = 408) were analysed. The cephalometric angles SNA and ANB were chosen for assessing maxillary growth for this part of the presentation. RESULTS: Within each trial (Trial 1a/1b, Trial 2a/2c, and Trial 3a/3d), there was no difference in cephalometric values between the common and the local arm. There were no statistically significant differences in the SNA and ANB angles between the common arm in Trial 1a (mean SNA 77.8, mean ANB 2.6) and Trial 2a (mean SNA 79.8, mean ANB 3.6) and no difference between Trial 1a and Trial 3a, but a statistical difference could be seen between Trial 2a and Trial 3a (mean SNA 76.9, mean ANB 1.7). However, the confidence interval was rather large. Intra- and inter-rater reliability were within acceptable range. CONCLUSIONS: The timing and the surgical method is not of major importance as far as growth outcomes (SNA and ANB) in UCLP are concerned. REGISTRATION: ISRCTN29932826. PROTOCOL: The protocol was not published before trial commencement.

Scandcleft randomized trials of primary surgery for unilateral cleft lip and palate. Dental arch relationships in 8 year-olds.

BACKGROUND AND TRIAL DESIGN: The Scandcleft intercentre study evaluates the outcomes of four surgical protocols for treatment of children with unilateral cleft lip and palate (UCLP). Originally 10 cleft centres in Denmark, Finland, Norway, Sweden, and the UK participated in a set of three randomized trials of primary surgery. Three groups of centres (Trials 1, 2, and 3) tested their traditional local surgical protocols (Arms B, C, and D) against a common protocol (Arm A). OBJECTIVES: To evaluate dental arch relationships at age 8 years after four different protocols of primary surgery for UCLP. These results are secondary outcomes of the overall trial. METHODS: Study models of 411 children (270 boys, 141 girls) with non-syndromic UCLP at a mean age of 8.1 (range 7.0-10.0) years were available. Dental arch relationships were analysed using the GOSLON Yardstick by a blinded panel of 11 orthodontists. To assess reliability, Kappa statistics were calculated. The trials were tested statistically with t-tests. RESULTS: Comparisons within each trial showed no statistically significant differences in the mean 8-year index scores or their distributions between the common protocol and the local team protocol. The mean index scores were Trial 1: Arm A 3.03, Arm B 2.82, Trial 2: Arm A 2.78, Arm C 2.64, and Trial 3: Arm A 3.06, Arm D 3.08. Comparisons between the trials detected a significantly (P < 0.005) better mean index score Trial 2 Arm C than in Trial 3 Arm D. The intra- and inter-rater reliabilities were acceptable. CONCLUSION: The results of these three trials do not provide evidence that one surgical protocol is better than the others. TRIAL REGISTRATION: ISRCTN29932826.

Scandcleft randomized trials of primary surgery for unilateral cleft lip and palate: dental anomalies in 8-year olds.

BACKGROUND: Children born with unilateral cleft lip and palate (UCLP) are reported to display several dental anomalies including agenesis, supernumeraries, as well as variations in dental size, shape, and path of eruption. The extensive sample of individuals with UCLP included in the Scandcleft randomized control trials offers the opportunity to study more rare conditions, which is seldom possible with limited samples. OBJECTIVES: The aim was to study dental anomalies at 8 years of age in children born with UCLP included in the Scandcleft randomized control trials. METHODS: Panoramic and intraoral radiographs from 425 individuals (279 males and 146 females) with a mean age of 8.1 years were assessed by four orthodontists regarding dental anomalies. RESULTS: Agenesis was found in 52.5 per cent and supernumerary teeth in 16.9 per cent of the participants. The cleft lateral was missing in 43.8 per cent and was found peg shaped in 44.7 per cent. The distribution of ectopic eruption was 14.6 per cent, mainly affecting maxillary first molars, while transposition was found in 3.4 per cent of the individuals. In addition, infraocclusion of one or several primary molars was registered in 7.2 per cent of the participants. CONCLUSION: We conclude that 8-year-old children born with UCLP display multiple dental anomalies. The Scandcleft sample allowed rarely studied conditions such as infraocclusion of primary molars and transposition to be studied in children born with UCLP. TRIAL REGISTRATION: ISRCTN29932826.

Vertical maxillary growth after two different surgical protocols in unilateral cleft lip and palate patients.

Objective : The aim of the present study was to compare vertical maxillofacial growth in patients born with unilateral cleft lip and palate (UCLP) who were treated using two different surgical protocols. Design : A retrospective cohort study. Subjects : We studied 92 patients with complete UCLP (61 male and 31 female) treated at Sahlgrenska University Hospital in Gothenburg, Sweden: 46 consecutive patients born between 1965 and 1974 who underwent surgical treatment according to the Wardill-Kilner (W-K) protocol and 46 consecutive patients born between 1982 and 1989 who underwent surgical treatment according to the Gothenburg delayed hard palate closure (DHPC) protocol. Methods : We analyzed lateral cephalograms obtained at 10 years of age. Results : Patients treated according to the Gothenburg DHPC protocol had significantly greater anterior upper facial height, anterior maxillary height, overbite, and inclination of the maxilla than those treated with the W-K protocol. Both techniques led to similar posterior upper facial height. Conclusion : The Gothenburg DHPC protocol in patients with complete UCLP results in more normal anterior maxillary vertical growth and overbite and therefore increased maxillary inclination at 10 years of age.

45,X/46,XX karyotype mitigates the aberrant craniofacial morphology in Turner syndrome.

The aim of this project was to study the impact on craniofacial morphology from Turner syndrome (TS) karyotype, number of intact X chromosomal p-arms, and age as well as to compare craniofacial morphology in TS with healthy females. Lateral radiographs from 108 females with TS, ranging from 5.4 to 61.6 years, were analysed. The TS females were divided into four karyotype groups: 1. monosomy (45,X), 2. mosaic (45,X/46,XX), 3. isochromosome, and 4. other, as well as according to the number of intact X chromosomal p-arms. The karyotype was found to have an impact on craniofacial growth, where the mosaic group, with presence of 46,XX cell lines, seems to exhibit less mandibular retrognathism as well as fewer statistically significant differences compared to the reference group than the 45,X karyotype. Isochromosomes had more significant differences versus the reference group than 45,X/46,XX but fewer than 45,X. To our knowledge, this is the first time the 45,X/46,XX and isochromosome karyotypes are divided into separate groups studying craniofacial morphology. Impact from p-arm was found on both maxillary and mandibular length. Compared to healthy females, TS expressed a shorter posterior and flattened cranial base, retrognathic, short and posteriorly rotated maxilla and mandible, increased height of ramus, and relatively shorter posterior facial height. The impact of age was found mainly on mandibular morphology since mandibular retrognathism and length were more discrepant in older TS females than younger.

Palatal height and dental arch dimensions in Turner syndrome karyotypes.

The aim of this project was to study the impact from Turner syndrome (TS) karyotype and age on dental arch morphology and palatal height and to compare the variables in TS with reference data from non-TS females with normal occlusion. Plaster casts from 76 females with TS (6-50 years) were analysed with respect to dentoalveolar arch dimensions and palatal height. The TS females were divided into the karyotype categories: i) 45,X ii) 45,X/46,XX iii) isochromosome, and iv) other. The 45,X/46,XX karyotype exhibited fewer statistically significant variables differing from the reference group compared with other karyotypes. TS females showed increased dentoalveolar depths, decreased maxillary but increased mandibular width, decreased posterior segments, and decreased mandibular circumference compared with the reference group. In opposition to previous reports, the palatal height did not differ compared with non-TS females. Age had an impact on nine of the variables. We conclude that the present dental arch deviations are reflecting the high frequency of malocclusions reported in TS and the subsequent need for orthodontic treatment, which might possibly be lower in the 45,X/46,XX karyotype. The palatal height did not differ from the reference group, but instead the narrow maxilla might contribute to an illusion of a higher palate. We therefore suggest using the nomination 'narrow palatal vault' instead of the commonly used term 'high palatal vault'.

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.