| 1. |
- Okhiria, Åsa, et al.
(författare)
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Greater Palatal Cleft Width Predicts an Increased Risk for Unfavorable Outcomes in Cleft Palate Repair.
- 2022
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Ingår i: The Cleft Palate-Craniofacial Journal. - : Sage. - 1055-6656 .- 1545-1569. ; 59:8, s. 1030-1037
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Tidskriftsartikel (refereegranskat)abstract
- OBJECTIVE: To investigate the impact of cleft width and cleft type on the need for secondary surgery and velopharyngeal competence from a longitudinal perspective.DESIGN: Retrospective, longitudinal study.SETTING: A single multidisciplinary craniofacial team at a university hospital.PATIENTS: Consecutive patients with unilateral or bilateral cleft lip and palate and cleft palate only (n = 313) born from 1984 to 2002, treated with 2-stage palatal surgery, were reviewed. A total of 213 patients were included.MAIN OUTCOME MEASURES: The impact of initial cleft width and cleft type on secondary surgery. Assessment of hypernasality, audible nasal emission, and glottal articulation from routine follow-ups from 3 to 16 years of age. The assessments were compared with reassessments of 10% of the recordings.RESULTS: Cleft width, but not cleft type, predicted the need for secondary surgery, either due to palatal dehiscence or velopharyngeal insufficiency. The distribution of cleft width between the scale steps on a 4-point scale for hypernasality and audible nasal emission differed significantly at 5 years of age but not at any other age. Presence of glottal articulation differed significantly at 3 and 5 years of age. No differences between cleft types were seen at any age for any speech variable.CONCLUSIONS: Cleft width emerged as a predictor of the need for secondary surgery as well as more deviance in speech variables related to velopharyngeal competence during the preschool years. Cleft type was not related to the need for secondary surgery nor speech outcome at any age.
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| 2. |
- Okhiria, Åsa, et al.
(författare)
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Longitudinal data on speech outcomes in internationally adopted children compared with non-adopted children with cleft lip and palate
- 2023
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Ingår i: International journal of language and communication disorders. - : Wiley. - 1368-2822 .- 1460-6984. ; 58:5, s. 1440-1453
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Tidskriftsartikel (refereegranskat)abstract
- BackgroundAt the beginning of the 21st century, international adoptions of children with cleft lip and/or palate increased dramatically in Sweden. Many children arrived partially or totally unoperated, despite being at an age when palatoplasty has usually been performed. To date, the speech development of internationally adopted (IA) children has been described up to age 7-8 years, but later development remains unstudied. AimsTo investigate speech development between ages 5 and 10 years in children born with cleft lip and palate (CLP) adopted from China and to compare them with non-adopted (NA) children with CLP. A secondary aim was to compare the frequencies of secondary palatal surgery and number of visits to a speech and language pathologist (SLP) between the groups. Methods & ProceduresIn a longitudinal study, 23 IA children from China were included and matched with 23 NA children born in Sweden. Experienced SLPs blindly reassessed audio recordings from routine follow-ups at ages 5 and 10 years. Velopharyngeal function (VPF) was assessed with the composite score for velopharyngeal competence (VPC-Sum) for single words and rated on a three-point scale (VPC-Rate) in sentence repetition. Target sounds in words and sentences were phonetically transcribed. Per cent correct consonants (PCC) were calculated at word and sentence levels. For in-depth analyses, articulation errors were divided into cleft speech characteristics (CSCs), developmental speech characteristics (DSCs) and s-errors. Information on secondary palatal surgery and number of visits to an SLP was collected. Outcomes & ResultsVPF differed significantly between the groups at both ages when assessed with VPC-Sum, but not with VPC-Rate. Regardless of the method for assessing VPF, a similar proportion in both groups had incompetent VPF but fewer IA than NA children had competent VPF at both ages. IA children had lower PCC at both ages at both word and sentence levels. More IA children had CSCs, DSCs and s-errors at age 5 years, and CSCs and s-errors at age 10. The development of PCC was significant in both groups between ages 5 and 10 years. The proportion of children receiving secondary palatal surgery did not differ significantly between the groups, nor did number of SLP visits. Conclusions & ImplicationsCSCs were more persistent in IA children than in NA children at age 10 years. Interventions should target both cleft and DSCs, be comprehensive and continue past the pre-school years. WHAT THIS PAPER ADDSWhat is already known on this subjectAt the beginning of the 21st century, IA children with cleft lip and/or palate arrived in Sweden partially or totally unoperated, despite being at an age when palatoplasty has usually been performed. Studies up to age 7-8 years show that adopted children, compared with NA peers, have poorer articulation skills, demonstrate both cleft-related and developmental articulation errors, and are more likely to have velopharyngeal incompetence. Several studies also report that adopted children more often require secondary palatal surgery due to fistulas, dehiscence or velopharyngeal incompetence compared with NA peers. What this paper adds to existing knowledgeThis longitudinal study provides additional knowledge based on longer follow-ups than previous studies. It shows that the proportion of children assessed to have incompetent VPF was similar among IA and NA children. It was no significant difference between the groups regarding the proportion that received secondary palatal surgery. However, fewer IA children were assessed to have a competent VPF. Developmental articulation errors have ceased in most IA and all NA children at age 10 years, but significantly more adopted children than NA children still have cleft-related articulation errors. What are the potential or actual clinical implications of this work?Speech and language therapy should target both cleft-related and developmental articulation errors. When needed, treatment must be initiated early, comprehensive, and continued past the pre-school years, not least for adopted children.
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| 3. |
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| 4. |
- Engstrom, Olof, et al.
(författare)
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Proboscis Lateralis : A Novel Surgical Technique Using Tape Elongation and One-Stage Tunneling
- 2022
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Ingår i: The Journal of Craniofacial Surgery. - : Ovid Technologies (Wolters Kluwer Health). - 1049-2275 .- 1536-3732. ; 33:4, s. E438-E439
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Tidskriftsartikel (refereegranskat)abstract
- Proboscis lateralis is a rare craniofacial anomaly characterized by a tubular, trunk-like appendage, often arising from the medial canthal region. It can occur in isolation or in association with a variety of other anomalies. Several treatment options have been described depending on the characteristics of the defect, but due to its infrequency, the body of knowledge is limited. Here, the authors present a case of proboscis lateralis in an 18-month-old girl, being successfully treated with a novel technique including tape elongation and a 1 stage tunneling procedure.
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| 5. |
- Falk Delgado, Alberto, et al.
(författare)
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The Skoog Lip Repair for Unilateral Cleft Lip Deformity : The Uppsala Experience
- 2018
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Ingår i: Plastic and reconstructive surgery (1963). - 0032-1052 .- 1529-4242. ; 141:5, s. 1226-1233
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Tidskriftsartikel (refereegranskat)abstract
- Background: The Uppsala Craniofacial Center has been treating patients with unilateral cleft lip deformity using the lip repair technique described by Tord Skoog. The aim of this study was to determine complications after lip surgery and the incidence and indications for lip revisions in all patients born with unilateral cleft lip from 1960 to 2004.Methods: All patients who were born from 1960 to 2004 with unilateral cleft lip, cleft lip and alveolus, or cleft lip and palate and underwent lip repair were studied retrospectively. The timing, indication, complications of the primary procedure, and type of secondary surgery were recorded. Kruskal-Wallis and Fisher’s exact tests were used, with Bonferroni correction.Results: The study included 443 patients. The total rate of early surgical complications was 6 percent (n = 26). Secondary surgery for short upper lip was performed in 3.8 percent (n = 17), 8.4 percent (n = 37) underwent reduction of excess vermillion, 8.6 percent (n = 38) underwent scar revision, 11 percent (n = 51) underwent revision for incongruent vermillion-cutaneous border, and 10 percent (n = 45) underwent revision for other indications. Altogether, 45 percent had no secondary revisions.Conclusion: In conclusion, the Skoog lip repair is associated with a low total revision rate, and a short-lip deformity is rare.CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, IV.
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| 6. |
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| 7. |
- Hakelius, Malin, 1965-
(författare)
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Interactions between Malignant Keratinocytes and Fibroblasts : Studies in Head and Neck Squamous Cell Carcinoma
- 2014
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Doktorsavhandling (övrigt vetenskapligt/konstnärligt)abstract
- Carcinoma growth requires a supportive tumor stroma. The concept of reciprocal interactions between tumor and stromal cells has become widely acknowledged and the connective tissue activation seen in the malignant process has been likened to that of a healing wound. Little is, however, known about the specific characteristics of these interactions, distinguishing them from the interplay occurring between epithelial and stromal cells in wound healing. In order to study differences in the humoral effects of malignant and benign epithelial cells on fibroblasts, we used an in vitro coculture model with human oral squamous cell carcinoma cells (SCC) or normal oral keratinocytes (NOK) on one side of a semi-permeable membrane and fibroblasts seeded in gels on the other. Pro-collagens α1(I) and α1(III) were more downregulated in NOK cocultures compared to SCC cocultures. IL-1α was identified as a major keratinocyte-derived soluble factor behind the effects observed. We concluded that SCC are less antifibrotic compared to NOK. There was also a differential expression among enzymes involved in ECM turnover. The urokinase-type plasminogen activator (uPA) and plasminogen activator inhibitor-1 (PAI-1) were both upregulated by NOK, but not by SCC. Here, rIL-1ra caused further upregulation of PAI-1. Global gene expression in fibroblasts was assessed using Affymetrix™ arrays. In total, 82 transcripts were considered differentially expressed; 52 were up- and 30 were downregulated in SCC compared to NOK cocultures. Among the differentially expressed genes there was an enrichment of genes related to collagens and to a nonspecific, innate-type response. The innate response marker pentraxin (PTX3) was upregulated by keratinocyte-derrived IL-1α in both NOK and SCC cocultures. We observed a considerably higher IL-1α / IL-1ra quotient in SCC cocultures, however, while PTX3 mRNA upregulation was higher in SCC cocultures, there was no difference in the level of PTX3 secreted protein. Taken together, we concluded that NOK and SCC regulate genes important for ECM composition and for the innate immune-response differentially. IL-1α was identified as one important mediator of the observed effects. In general, SCC appeared to be more profibrotic in their effects on fibroblasts.
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| 8. |
- Hakelius, Malin, 1965-, et al.
(författare)
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Normal oral keratinocytes and head and neck squamous carcinoma cells induce an innate response in fibroblasts
- 2016
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Ingår i: Anticancer Research. - 0250-7005 .- 1791-7530. ; 36:5, s. 2131-2137
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Tidskriftsartikel (övrigt vetenskapligt/konstnärligt)abstract
- Background: Tumor stroma is similar to the connective tissue of chronic inflammation. The extracellular matrix of tumors is formed by cancer-associated fibroblasts that also modulate the inflammatory response. Materials and Methods: We studied the ability of oral keratinocytes (NOK) and oral squamous cell carcinoma cells (SCC) to induce an innate immune response in fibroblasts. Co-cultures with fibroblasts in collagen gels and keratinocytes in inserts were used. Pentraxin 3 (PTX3) was used as an indicator of an innate immune response. Results: SCC and NOK up-regulated fibroblast mRNA expression and protein release of PTX3. mRNA levels were more pronounced in cultures with malignant cells. The induction of PTX3 was abrogated by an interleukin-1 receptor antagonist Conclusion: Keratinocytes have the capacity to induce an interleukin-1-dependent innate immune response by fibroblasts in vitro. This could be important for subsequent fibroblast modulation of the inflammatory reaction in non-malignant and malignant disease processes.
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| 9. |
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| 10. |
- Jabbari, Fatima, et al.
(författare)
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Secondary Alveolar Bone Grafting in Patients Born With Unilateral Cleft Lip and Palate : A 20-Year Follow-up
- 2018
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Ingår i: The Cleft Palate-Craniofacial Journal. - : SAGE Publications. - 1055-6656 .- 1545-1569. ; 55:2, s. 173-179
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Tidskriftsartikel (refereegranskat)abstract
- Objective: To identify factors of oral health important for the final outcome, after secondary alveolar bone grafting in patients born with unilateral cleft lip and palate and compare occlusal radiographs with cone beam computed tomography (CBCT) in assessment of alveolar bone height. Design: Observational follow-up study. Setting: Cleft Lip and Palate Team, Craniofacial Center, Uppsala University Hospital, Sweden. Patients: 40 nonsyndromic, Caucasian patients with unilateral complete cleft lip and palate. Interventions: Clinical examination, CBCT, and occlusal radiographs. Main Outcome Measurements: Alveolar bone height was evaluated according to Bergland index at a 20-year follow-up. Results: The alveolar bone height in the cleft area was significantly reduced compared to a previously reported 10-year follow-up in the same cohort by total (P = .045) and by subgroup with dental restoration (P = .0078). This was positively correlated with the gingival bleeding index (GBI) (r = 0.51, P = .0008) and presence of dental restorations in the cleft area (r = 0.45, P = .0170). There was no difference in the Bergland index generated from scoring the alveolar bone height on occlusal radiographs as with the equivalent index on CBCT. Conclusion: Patients rehabilitated with complex dental restoration seems to be at higher risk for progression of bone loss in the cleft area. Supportive periodontal therapy should be implemented after complex dental restorations in cleft patients. Conventional occlusal radiographs provide an adequate image for evaluating postoperative bone height in clinical follow-up.
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| 11. |
- Jabbari, Fatemeh, et al.
(författare)
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Skoog Primary Periosteoplasty versus Secondary Alveolar Bone grafting in Unilateral Cleft Lip and Alveolus : Long.term effects on alveolar Bone Formation and Maxillary Growth
- 2017
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Ingår i: Plastic and reconstructive surgery (1963). - 0032-1052 .- 1529-4242. ; 139:1, s. 137-148
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Tidskriftsartikel (refereegranskat)abstract
- Background: Clefts involving the alveolus are treated using one of two strategies: primary periosteoplasty at the time of lip repair or secondary alveolar bone grafting at mixed dentition. Most teams favor secondary alveolar bone grafting because of its high success rate, and concerns have been raised that primary periosteoplasty may interfere with maxillary growth. However, primary periosteoplasty may obviate the need for future bone grafting and is still practiced in some centers. Few studies compare the long-term outcomes of these two strategies.Methods: Fifty-seven consecutive patients born with unilateral cleft lip and alveolus were studied retrospectively. All patients underwent primary lip repair using Skoog's method; 28 patients underwent primary periosteoplasty at the time of lip repair and the remaining 29 underwent secondary alveolar bone grafting at mixed dentition. Occlusal radiographs obtained at ages 10 and 16 years were analyzed for alveolar bone height. Cephalometric analysis assessed growth at ages 5, 10, and 18 years.Results: Seventeen of 28 patients treated using primary periosteoplasty required later secondary bone grafting, and the bone height at age 16 years was lower in the primary periosteoplasty group (p < 0.0001). There was a more pronounced decrease in maxillary protrusion from ages 5 to 10 years in the primary periosteoplasty group (p < 0.03). However, at age 18 there was no significant difference in maxillary growth between the two groups.Conclusion: Primary periosteoplasty did not seem to inhibit long-term maxillary growth but was ineffective as a method of reconstructing the alveolar cleft.
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| 12. |
- Klintö, Kristina, et al.
(författare)
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Coverage, reporting degree and design of the Swedish quality registry for patients born with cleft lip and/or palate
- 2020
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Ingår i: BMC Health Services Research. - : BMJ Publishing Group Ltd. - 1472-6963. ; 20:1
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Tidskriftsartikel (refereegranskat)abstract
- 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.
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| 13. |
- Tillman, Karin K., 1977-, et al.
(författare)
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Bilateral orofacial clefts confer increased risk of psychiatric morbidity relative to unilateral clefts
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Annan publikation (övrigt vetenskapligt/konstnärligt)abstract
- BackgroundNonsyndromic orofacial clefts (OFC) are associated with neurodevelopmental disorders. The aim of this study was to compare children with bilateral clefts with unilateral clefts regarding psychiatric disorders.MethodsWe utilized data on all children born in Sweden between 1973 and 2012 with unilateral cleft lip (UCL), bilateral cleft lip (BCL), unilateral cleft lip and palate (UCLP) and bilateral cleft lip and palate (BCLP). We performed Cox regression analyses with direct comparisons between bilateral and unilateral clefts compared to matched community cohorts and adjusted for confounders.ResultsChildren with BCL showed risk increases for any psychiatric disorder (aHR 2.13, 95% CI 1.04-4.36), including intellectual disability (aHR 5.31, 95% CI 1.29-21.78) compared to UCL. Children with BCLP demonstrated risk increases for any psychiatric disorder (aHR 1.55, 95% CI 1.20-2.01), including speech and language disorders (aHR 1.99, 95% CI 1.00-3.97), neurodevelopmental disorders (aHR 1.66, 95% CI 1.11-2.47) and other psychiatric disorders (aHR 1.54, 95% CI 1.11-2.15), such as personality disorders (aHR 5.76, 95% CI 2.13- 15.55) compared to UCLP.ConclusionsThis large nationwide register study shows for the first time that individuals with bilateral clefts demonstrate elevated risks of psychiatric disorders compared to unilateral clefts.
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| 14. |
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| 15. |
- Tillman, Karin K., et al.
(författare)
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Increased Risk for Neurodevelopmental Disorders in Children With Orofacial Clefts
- 2018
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Ingår i: Journal of the American Academy of Child and Adolescent Psychiatry. - : Elsevier BV. - 0890-8567 .- 1527-5418. ; 57:11, s. 876-883
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Tidskriftsartikel (refereegranskat)abstract
- OBJECTIVE: Children with orofacial clefts (OFC) may have an increased risk of poor mental health. This study aimed to investigate the risk of psychiatric diagnoses in individuals with OFC, stratified by cleft type.METHOD: A nationwide register-based cohort of all individuals born with nonsyndromic OFC in Sweden between 1973 and 2012 (n = 7,842) was compared to a matched cohort (n = 78,409) as well as to their unaffected siblings (n = 9,637). The risk of psychiatric diagnoses, suicide attempts, and suicides was examined by crude and adjusted Cox regression models. Effect modification by sex was investigated with interaction terms in the models.RESULTS: Children with cleft lip (CL) had a significantly higher risk of any psychiatric disorder, intellectual disability, and language disorders; children with cleft lip and palate (CLP) had, in addition, an increased risk of autism spectrum disorder (ASD). Children with cleft palate only (CPO) had risk increases for the same diagnoses as children with CL and CLP, but with higher hazard ratios, and also for psychotic disorders, attention-deficit/hyperactivity disorder (ADHD), and other behavioral or emotional disorders in childhood. Sex stratification indicated higher risk increases among females in CL and CLP but not in CPO. Siblings without OFC were less likely to be diagnosed with any psychiatric disorder, intellectual disability, language disorder, ASD, or ADHD compared to their siblings with OFC.CONCLUSION: Children with nonsyndromic clefts had a significantly higher risk of neurodevelopmental disorders. This risk is unlikely to be explained by familial influences such as inherited genetic or shared environmental factors.
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