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- Pegelow, M., et al.
(author)
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Reliability and Predictive Validity of Dental Arch Relationships Using the 5-Year-Olds’ Index and the GOSLON Yardstick to Determine Facial Growth
- 2021
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In: Cleft Palate-Craniofacial Journal. - : SAGE Publications. - 1055-6656 .- 1545-1569. ; 58:5, s. 619-627
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Journal article (peer-reviewed)abstract
- 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. © American Cleft Palate-Craniofacial Association. All rights reserved 2020.
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- Pegelow, M., et al.
(author)
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Validation of reported dentoalveolar relationships in the Swedish Quality Registry for Cleft Lip and Palate
- 2020
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In: European journal of orthodontics. - : Oxford University Press (OUP). - 1460-2210 .- 0141-5387. ; 42:1, s. 30-35
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Journal article (peer-reviewed)abstract
- 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. © The Author(s) 2019. Published by Oxford University Press on behalf of the European Orthodontic Society.
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- Pegelow, M., et al.
(author)
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Association and Mutation Analyses of the IRF6 Gene in Families With Nonsyndromic and Syndromic Cleft Lip and/or Cleft Palate
- 2014
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In: The Cleft Palate-Craniofacial Journal. - : SAGE Publications. - 1055-6656 .- 1545-1569. ; 51:1, s. 49-55
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Journal article (peer-reviewed)abstract
- Objectives: (1) To detect interferon regulatory factor 6 gene (IRF6) mutations in newly recruited Van der Woude syndrome (VWS) and popliteal pterygium syndrome (PPS) families. (2) To test for association, in nonsyndromic cleft lip and/or cleft palate (NSCL/P) and in VWS/PPS families, the single nucleotide polymorphism (SNP) rs642961, from the IRF6 enhancer AP-2 alpha region, alone or as haplotype with rs2235371, a coding SNP (Val274Ile). Design: IRF6 mutation screening was performed by direct sequencing and genotyping of rs642961 and rs2235371 by TaqMan technology. Patients: Seventy-one Swedish NSCL/P families, 24 Finnish cleft palate (CP) families, and 24 VWS/PPS families (seven newly recruited) were studied. Results: Allelic and genotypic frequencies in each phenotype were compared to those of the controls, and no significant difference could be observed. IRF6 gene mutation was detected in six of the seven new VWS/PPS families. Association analysis of the entire VWS/PPS sample set revealed the A allele from rs642961 to be a risk allele. Significant association was detected in the Swedish CP subset of our NSCL/P collection where the G-C haplotype for rs642961-rs2235371 were at risk (P = .013). Conclusions: Our results do not support the previously reported association between the A allele of rs642961 and the NSCL phenotype. However, in the VWS/PPS families, the A allele was a risk allele and was, in a large majority (>80%), transmitted on the same chromosome as the IRF6 mutation.
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- Peterson, P, et al.
(author)
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Mean GOSLON Yardstick Scores After 3 Different Treatment Protocols-A Long-term Study of Patients With Unilateral Cleft Lip and Palate
- 2019
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In: The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association. - : SAGE Publications. - 1545-1569. ; 56:2, s. 236-247
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Journal article (peer-reviewed)abstract
- (1) To evaluate dental arch relationships, with the Great Ormond Street, London and Oslo (GOSLON) Yardstick, of participants with Unilateral cleft lip and palate (UCLP) and treated with 1-stage palatal closure with 3 different surgical protocols (2) to compare the mean GOSLON ratings with other CLP centers. Design: Retrospective study of medical charts and dental models. Setting: Karolinska University Hospital, Stockholm, Sweden. Participants: Eighty-seven patients with UCLP operated with 1-stage palatal repair. Thirty-five were operated with Veau-Wardill-Kilner (VWK) technique 1975 to 1986, 31 with minimal incision technique (MIT) from 1987 to 1997, and 21 according to MIT with muscle reconstruction (MITmr) 1998 to 2004. Interventions: Dental casts at ages 5 (n = 87), 7 to 8 (n = 27), 10 (n = 81), 16 (n = 61), and 19 (n = 35) years were rated by 10 assessors with the GOSLON Yardstick. Information of other interventions was retrieved from patients’ charts. Main outcome measures: Mean GOSLON ratings. Results: A total of 82% of the participants were rated as having excellent to satisfactory outcome. Weighted κ statistics for the 10 assessors was good for inter-rater agreement and good/very good for intra-rater agreement. Conclusions: The mean GOSLON score in the Stockholm overall material at age 10 was 2.67. The VWK technique resulted in a greater need of orthognathic surgery than the MIT ( P < .01). The MITmr did not produce better dental arch relationships than MIT at age 5 ( P < .05). The best dental arch relationships were found in the MIT group at 10 years, mean 2.58, which is not significantly different from other centers with excellent outcome except Gothenburg and Vienna.
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- Kadi, H, et al.
(author)
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A CBCT Based Assessment of Canine Eruption and Development Following Alveolar Bone Grafting in Patients Born With Unilateral Cleft lip and/or Palate
- 2023
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In: The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association. - : SAGE Publications. - 1545-1569. ; 60:4, s. 386-394
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Journal article (peer-reviewed)abstract
- To three-dimensionally assess and visualize the eruption path and development of the maxillary canine following alveolar bone grafting in patients born with cleft lip and palate. A further objective of this analysis was to assess how the presence of the lateral incisor impacts the eruption path of the canine. Observational follow-up study. Stockholm Craniofacial Team, Karolinska University Hospital, Sweden. Thirty children born with non-syndromic unilateral cleft lip with or without palate were consecutively included. CBCT scans of the maxilla were taken six months before and six months after the alveolar bone-grafting surgery for each patient. Canine eruption (angulation and vertical movement) and canine development (length and volume). There was a significant difference pre- and post-operatively of the canine angulation between the cleft and non-cleft sides. The mean angulation on the cleft side was 14.7° (SD = 11.1°) while on the non-cleft side, it was 4.9° (SD = 9.2°). No significant differences were noted between cleft-side and non-cleft side canines in terms of amount of vertical eruption and volumetric development. Absence of the lateral incisor did not significantly contribute to either canine angulation or its vertical eruption on the cleft-side. Higher angulation of the canine on the cleft side indicates a higher risk of future canine impaction. Presence or absence of the lateral incisor did not significantly affect canine angulation or its vertical eruption. Increased age and children born with total cleft lip and palate imply a higher risk of angulated canines on the cleft side.
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