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Okhiria, Å. (2024). Articulation and velopharyngeal function in patients with cleft lip and/or palate: Outcome predictors. (Doctoral dissertation). Uppsala: Acta Universitatis Upsaliensis
Open this publication in new window or tab >>Articulation and velopharyngeal function in patients with cleft lip and/or palate: Outcome predictors
2024 (English)Doctoral thesis, comprehensive summary (Other academic)
Abstract [en]

Cleft lip and/or palate is the most common congenital craniofacial malformation, requiring multidisciplinary treatment, including surgery and often speech therapy. Palatal surgery restores the anatomical barrier between the oral and nasal cavities as well as the palatal function needed for normal speech.

The present thesis aimed to investigate factors thought to impact surgical and speech outcomes. These factors include the timing of surgery, surgical technique, the surgeon's experience, cleft type, and cleft width.

Study I investigated the impact of cleft type and width on velopharyngeal function (VPF) and secondary surgery rates from ages three to 16. Cleft width was associated with increased surgery rates and signs of velopharyngeal insufficiency (VPI) at ages three and five. Contrary to some previous studies, surgery rates and speech outcomes were not associated with cleft type at any age.

Study II examined VPF and articulation development between ages five and ten in children adopted from China compared to Swedish-born children. Adoptees had palatoplasty later and had more severe articulation difficulties at both ages. The number of adoptees and non-adoptees with VPI was similar, but fewer adoptees had competent VPF.

Study III explored different types of intra-velar veloplasty and the impact of cleft width and surgeon experience on outcomes in 5-year-old children. Radical muscle dissection was not superior to intra-velar veloplasty reinforced by the palatopharyngeal muscle. Cleft width had a more significant impact on secondary surgery rates and VPF than did surgical technique—neither affected articulation proficiency.

Study IV examined the association between cleft type and width and surgical and speech outcomes, especially articulation, in 5-year-old children. Cleft width, not type, indicated articulation proficiency. The same errors occurred across all cleft types. Neither cleft type nor width was significantly associated with secondary surgery rates or VPF.

The present thesis highlights the importance of considering various factors when predicting secondary palatal surgery rates and speech outcomes. Cleft width significantly affects secondary surgery rates, VPF, and articulation proficiency, while cleft type and surgical technique do not. Adopted children are at higher risk of persistent articulation errors, which may partly be due to their later palatoplasty. 

Place, publisher, year, edition, pages
Uppsala: Acta Universitatis Upsaliensis, 2024. p. 62
Series
Digital Comprehensive Summaries of Uppsala Dissertations from the Faculty of Medicine, ISSN 1651-6206 ; 2009
Keywords
cleft lip and palate, cleft width, cleft type, surgical technique, articulation, velopharyngeal function, international adoption
National Category
Surgery
Identifiers
urn:nbn:se:uu:diva-519479 (URN)978-91-513-2012-0 (ISBN)
Public defence
2024-03-01, H:son Holmdahl-salen, Akademiska sjukhuset, ing. 100, 2 tr, Uppsala, 09:15 (English)
Opponent
Supervisors
Available from: 2024-02-09 Created: 2024-01-14 Last updated: 2024-02-09
Okhiria, Å., Persson, C., Blom Johansson, M., Hakelius, M. & Nowinski, D. (2023). Longitudinal data on speech outcomes in internationally adopted children compared with non‐adopted children with cleft lip and palate. International journal of language and communication disorders, 58(5), 1440-1453
Open this publication in new window or tab >>Longitudinal data on speech outcomes in internationally adopted children compared with non‐adopted children with cleft lip and palate
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2023 (English)In: International journal of language and communication disorders, ISSN 1368-2822, E-ISSN 1460-6984, Vol. 58, no 5, p. 1440-1453Article in journal (Refereed) Published
Abstract [en]

Background: At 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.

Aims: To 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 & Procedures: In 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 & Results: VPF 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 & and Implications: CSCs 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.

Place, publisher, year, edition, pages
Wiley, 2023
National Category
Surgery
Identifiers
urn:nbn:se:uu:diva-511350 (URN)10.1111/1460-6984.12869 (DOI)000949286400001 ()
Available from: 2023-09-12 Created: 2023-09-12 Last updated: 2024-01-23Bibliographically approved
Havstam, C., Brunnegård, K., Hagberg, E., Nelli, C., Okhiria, Å. & Klintö, K. (2023). Speech in 7-and 10-year-olds born with a unilateral cleft lip and palate: a continued prospective Swedish intercentre study. Journal of Plastic Surgery and Hand Surgery, 58, 149-154
Open this publication in new window or tab >>Speech in 7-and 10-year-olds born with a unilateral cleft lip and palate: a continued prospective Swedish intercentre study
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2023 (English)In: Journal of Plastic Surgery and Hand Surgery, ISSN 2000-656X, E-ISSN 2000-6764, Vol. 58, p. 149-154Article in journal (Refereed) Published
Abstract [en]

The aim of this study was to report longitudinal speech results in consecutively selected children from each of the six cleft centres in Sweden and to compare the results between centres. The children were born with a non-syndromic unilateral cleft lip and palate, and results from the same cohort at 5 years of age have previously been reported. Background data on medical care in terms of surgery, speech therapy, and hearing between 5 and 10 years of age were collected. Speech recordings of 56 children at 7 years and 54 at 10 years of age were blindly and independently assessed by four speech-language pathologists experienced in cleft palate speech. This resulted in measures of percent consonant correct (PCC) and perceived velopharyngeal competence rated on a three-tier scale. No statistically significant differences were found between centres. PCC scores at 7 years of age ranged from 44 to 100% (median 97.5) and at 10 years of age from 86 to 100% (median 100). Competent or marginally incompetent velopharyngeal function was found in 95% of the 7-year-olds and 98% of the 10-year-olds. Speech results were slightly better than previous reports of speech in children born with a unilateral cleft lip and palate.

Place, publisher, year, edition, pages
Medical Journals Sweden, 2023
Keywords
Speech, unilateral cleft lip and palate, intercentre study
National Category
Otorhinolaryngology Surgery
Identifiers
urn:nbn:se:uu:diva-524327 (URN)10.2340/jphs.v58.15766 (DOI)001157817200024 ()38108470 (PubMedID)
Available from: 2024-03-05 Created: 2024-03-05 Last updated: 2024-03-05Bibliographically approved
Okhiria, Å., Jabbari, F., Hakelius, M., Blom Johansson, M. & Nowinski, D. (2022). Greater Palatal Cleft Width Predicts an Increased Risk for Unfavorable Outcomes in Cleft Palate Repair.. The Cleft Palate-Craniofacial Journal, 59(8), 1030-1037
Open this publication in new window or tab >>Greater Palatal Cleft Width Predicts an Increased Risk for Unfavorable Outcomes in Cleft Palate Repair.
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2022 (English)In: The Cleft Palate-Craniofacial Journal, ISSN 1055-6656, E-ISSN 1545-1569, Vol. 59, no 8, p. 1030-1037Article in journal (Refereed) Published
Abstract [en]

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.

Place, publisher, year, edition, pages
Sage, 2022
Keywords
cleft type, cleft width, nasality, secondary surgery, speech production, velopharyngeal competence, velopharyngeal function
National Category
Surgery
Identifiers
urn:nbn:se:uu:diva-459538 (URN)10.1177/10556656211029537 (DOI)000705465500001 ()34259078 (PubMedID)
Available from: 2021-11-25 Created: 2021-11-25 Last updated: 2024-01-23Bibliographically approved
Klinto, K., Karsten, A., Marcusson, A., Paganini, A., Rizell, S., Cajander, J., . . . Becker, M. (2020). Coverage, reporting degree and design of the Swedish quality registry for patients born with cleft lip and/or palate. BMC Health Services Research, 20(1), Article ID 528.
Open this publication in new window or tab >>Coverage, reporting degree and design of the Swedish quality registry for patients born with cleft lip and/or palate
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2020 (English)In: BMC Health Services Research, E-ISSN 1472-6963, Vol. 20, no 1, article id 528Article in journal (Refereed) Published
Abstract [en]

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.

Keywords
Cleft lip, Cleft palate, Registry, Surgery, Orthodontics, Speech
National Category
Dentistry
Identifiers
urn:nbn:se:uu:diva-418956 (URN)10.1186/s12913-020-05389-x (DOI)000543100100001 ()32522189 (PubMedID)
Available from: 2020-09-09 Created: 2020-09-09 Last updated: 2022-09-15Bibliographically approved
Brunnegård, K., Hagberg, E., Havstam, C., Okhiria, Å. & Klintö, K. (2020). Reliability of Speech Variables and Speech-Related Quality Indicators in the Swedish Cleft Lip and Palate Registry. The Cleft Palate-Craniofacial Journal, 57(6), 715-722
Open this publication in new window or tab >>Reliability of Speech Variables and Speech-Related Quality Indicators in the Swedish Cleft Lip and Palate Registry
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2020 (English)In: The Cleft Palate-Craniofacial Journal, ISSN 1055-6656, E-ISSN 1545-1569, Vol. 57, no 6, p. 715-722Article in journal (Refereed) Published
Abstract [en]

Objective: To assess the reliability of speech variables and speech-related quality indicators in the Swedish quality registry for cleft lip and palate (CLP).

Design: Retrospective study.

Setting: Primary care university hospitals.

Participants: Fifty-two 5-year-old children with unilateral CLP and 41 with bilateral CLP.

Main Outcome Measures: Registry data for "percent nonoral errors" and "perceived velopharyngeal competence" (VPC) were compared to reassessments by 4 independent judges based on audio recordings. Interjudge agreement for "percent consonants correct" (PCC) and the reliability of 3 quality indicators were also assessed. Agreement was calculated with single measures intraclass correlation coefficient (ICC) for articulation outcomes, quadratic weighted kappa and ICC for VPC, and percentage agreement and kappa for quality indicators.

Results: When the agreement between registry data and the judges' reassessments was assessed, the ICC was 0.79 for percent nonoral errors. For VPC, the kappa coefficient was 0.66 to 0.75 and the ICC was 0.73. Interjudge agreement for PCC calculated with ICC was 0.85. For the quality indicator "proportion of children with >= 86% correct consonants," all 4 judges were in agreement for 72% of the cases. For "proportion of children without nonoral speech errors" and "proportion of children with competent or marginally incompetent velopharyngeal function," the agreement between registry data and the 4 judges was 89% and 85%, respectively.

Conclusions: The results indicate that registry data on PCC, percent nonoral errors, VPC, and the quality indicators "proportion of children without nonoral speech errors" and "proportion of children with competent or marginally incompetent velopharyngeal function" are reliable.

Place, publisher, year, edition, pages
ALLIANCE COMMUNICATIONS GROUP DIVISION ALLEN PRESS, 2020
Keywords
quality registry, cleft lip and palate, speech variables, quality indicators, reliability
National Category
Otorhinolaryngology
Identifiers
urn:nbn:se:uu:diva-414289 (URN)10.1177/1055665619894497 (DOI)000535938900007 ()31906706 (PubMedID)
Funder
Swedish Association of Local Authorities and Regions
Available from: 2020-06-26 Created: 2020-06-26 Last updated: 2020-06-26Bibliographically approved
Schölin, J. S., Jonasson, Å., Axelsson, J., Havstam, C., Persson, C., Jönsson, R. & Mark, H. (2020). Surgical, speech, and hearing outcomes at five years of age in internationally adopted children and Swedish-born children with cleft lip and/or palate. Journal of Plastic Surgery and Hand Surgery, 54(1), 6-13
Open this publication in new window or tab >>Surgical, speech, and hearing outcomes at five years of age in internationally adopted children and Swedish-born children with cleft lip and/or palate
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2020 (English)In: Journal of Plastic Surgery and Hand Surgery, ISSN 2000-656X, E-ISSN 2000-6764, Vol. 54, no 1, p. 6-13Article in journal (Refereed) Published
Abstract [en]

Internationally adopted children (IAC) with a cleft lip and/or palate (CL/P) tend to arrive with un-operated palates at an age at which their Swedish-born peers have completed their primary palate surgery. Our aim of the present study was to analyze surgical, speech and hearing outcomes of IAC at age 5 and compare with those of a matched group of Swedish-born children. Fifty children with CL/P born in 1994-2005 participated in the study. Twenty-five IAC were matched according to age, sex and cleft type with 25 Swedish-born children. Audio recordings were perceptually analyzed by two experienced, blinded speech-language pathologists. Hearing and speech statuses were evaluated on the same day for all children. Surgical timing and complications as in fistulas and requirement for secondary velopharyngeal (VP) surgery, speech evaluation results, and present hearing status were analyzed for all children of age 5 years. Results showed that primary palatal surgery was delayed by a mean of 21 months in IAC. IAC had a higher prevalence of velopharyngeal impairment that was statistically significant, a higher fistula rate, and experienced more secondary surgery than Swedish-born peers. Hearing loss due to middle ear disease was slightly more common among IAC, whereas the rate of treatment with tympanostomy tubes was similar between the two groups. In conclusion, IAC with CL/P represent a challenge for CL/P teams because of the heterogeneous nature of the patient group and difficulties associated with delayed treatment, and the results show the importance of close follow-up over time.

Place, publisher, year, edition, pages
Informa UK Limited, 2020
Keywords
Cleft palate, cleft palate surgery, hearing, internationally adopted, palatal fistulas, speech, velopharyngeal function
National Category
Surgery
Identifiers
urn:nbn:se:uu:diva-459539 (URN)10.1080/2000656X.2019.1650056 (DOI)000481063000001 ()31393744 (PubMedID)
Available from: 2021-11-25 Created: 2021-11-25 Last updated: 2023-11-27Bibliographically approved
Organisations
Identifiers
ORCID iD: ORCID iD iconorcid.org/0000-0002-8656-8064

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