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  • 1.
    Havstam, Christina
    et al.
    Sahlgrens Univ Hosp, Dept Otorhinolaryngol Speech & Language Pathol, SE-41345 Gothenburg, Sweden.;Univ Gothenburg, Sahlgrenska Acad, Dept Hlth & Rehabil, Speech & Language Pathol Unit,Inst Neurosci & Phy, Gothenburg, Sweden..
    Brunnegård, Karin
    Umeå Univ, Dept Clin Sci Speech & Language Pathol, Umeå, Sweden..
    Hagberg, Emilie
    Karolinska Univ Hosp, Med Unit Speech & Language Pathol, Stockholm, Sweden.;Karolinska Univ Hosp, Stockholm Craniofacial Team, Stockholm, Sweden.;Karolinska Inst, Dept Clin Sci Intervent & Technol, Div Speech & Language Pathol, Stockholm, Sweden..
    Nelli, Cecilia
    Linköping Univ Hosp, Otorhinolaryngol Clin, Speech & Language Pathol Unit, Linköping, Sweden..
    Okhiria, Åsa
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Surgical Sciences, Plastic Surgery.
    Klintö, Kristina
    Skane Univ Hosp, Dept Otorhinolaryngol, Div Speech Language Pathol, Malmö, Sweden.;Lund Univ, Dept Clin Sci Lund, Div Speech Language Pathol Phoniatr & Audiol, Lund, Sweden..
    Speech in 7-and 10-year-olds born with a unilateral cleft lip and palate: a continued prospective Swedish intercentre study2023In: Journal of Plastic Surgery and Hand Surgery, ISSN 2000-656X, E-ISSN 2000-6764, Vol. 58, p. 149-154Article in journal (Refereed)
    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.

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  • 2.
    Klintö, Kristina
    et al.
    Lund Univ, Dept Clin Sci Lund, Lund, Sweden.;Skane Univ Hosp, Dept Otorhinolaryngol, Div Speech & Language Pathol, S-20502 Malmö, Sweden..
    Hagberg, Emilie
    Karolinska Univ Hosp, Med Unit Speech Therapy, Stockholm, Sweden.;Karolinska Univ Hosp, Stockholm Craniofacial Team, Stockholm, Sweden..
    Havstam, Christina
    Sahlgrens Univ Hosp, Dept Otorhinolaryngol, Div Speech & Language Pathol, Gothenburg, Sweden..
    Nelli, Cecilia
    Linköping Univ Hosp, Speech & Language Therapy Unit, Linköping, Sweden..
    Okhiria, Åsa
    Uppsala Univ Hosp, Dept Speech Language Pathol, Uppsala, Sweden.
    Brunnegård, Karin
    Umeå Univ, Dept Clin Sci Speech & Language Pathol, Umeå, Sweden..
    Reliability of data on percent consonants correct and its associated quality indicator in the Swedish cleft lip and palate registry2024In: Logopedics, Phoniatrics, Vocology, ISSN 1401-5439, E-ISSN 1651-2022, Vol. 49, no 1, p. 27-33Article in journal (Refereed)
    Abstract [en]

    Background

    Data in national health care quality registries must be valid and reliable in order to enable open comparisons of results.

    Aim

    To assess the reliability of data on percent consonants correct (PCC) and its associated quality indicator ≥86% correct consonants in the Swedish quality registry for patients born with cleft lip and palate (CLP) registry.

    Methods

    Six independent speech-language pathologists re-assessed the audio recordings of 96 five-year-olds with PCC data in the CLP registry. Target consonants of a single-word picture-naming test were phonetically transcribed, and PCC was calculated. The reliability of PCC data was assessed with the intraclass correlation coefficient (ICC). The reliability of the quality indicator ≥86% correct consonants was assessed with point-by-point percentage agreement and Cohen’s kappa.

    Results

    Intra- and inter-judge agreement for PCC was excellent with ICCs above 0.9, and so was the agreement of data from the CLP registry and the six judges’ re-assessments. The percentage agreement between all judges and the CLP registry for the quality indicator ≥86% correct consonants was poor (67%). However, in 88% of the cases, results from four judges and the CLP registry agreed, corresponding to good agreement. The mean of all kappa values for six judges and the CLP registry corresponded to good agreement (0.72).

    Conclusions

    The results indicate the PCC data in the CLP registry and the quality indicator ≥86% correct consonants to be reliable. When differences in outcome between treatment centres are detected, the raw data collected should always be re-examined before drawing definitive conclusions.

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  • 3.
    Okhiria, Åsa
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Surgical Sciences, Plastic Surgery.
    Articulation and velopharyngeal function in patients with cleft lip and/or palate: Outcome predictors2024Doctoral 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. 

    List of papers
    1. Greater Palatal Cleft Width Predicts an Increased Risk for Unfavorable Outcomes in Cleft Palate Repair.
    Open this publication in new window or tab >>Greater Palatal Cleft Width Predicts an Increased Risk for Unfavorable Outcomes in Cleft Palate Repair.
    Show others...
    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
    2. Longitudinal data on speech outcomes in internationally adopted children compared with non‐adopted children with cleft lip and palate
    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
    Show others...
    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
    3. The impact of surgical technique and cleft width on the rate of secondary surgery and velopharyngeal function
    Open this publication in new window or tab >>The impact of surgical technique and cleft width on the rate of secondary surgery and velopharyngeal function
    Show others...
    (English)Manuscript (preprint) (Other academic)
    National Category
    Other Health Sciences
    Identifiers
    urn:nbn:se:uu:diva-517722 (URN)
    Available from: 2023-12-12 Created: 2023-12-12 Last updated: 2024-01-14
    4. Associations between cleft type and width and the rate of secondary palatal surgery and articulation proficiency in 5-year-olds with cleft lip and/or palate
    Open this publication in new window or tab >>Associations between cleft type and width and the rate of secondary palatal surgery and articulation proficiency in 5-year-olds with cleft lip and/or palate
    Show others...
    (English)Manuscript (preprint) (Other academic)
    National Category
    Other Medical Sciences not elsewhere specified
    Identifiers
    urn:nbn:se:uu:diva-517194 (URN)
    Available from: 2023-12-05 Created: 2023-12-05 Last updated: 2024-01-14
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    UUThesis_Å-Okhiria-2024
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  • 4.
    Okhiria, Åsa
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Surgical Sciences, Plastic Surgery.
    Jabbari, Fatima
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Surgical Sciences, Plastic Surgery.
    Hakelius, Malin
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Surgical Sciences, Plastic Surgery.
    Blom Johansson, Monica
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Public Health and Caring Sciences, Speech-Language Pathology.
    Nowinski, Daniel
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Surgical Sciences, Plastic Surgery.
    Greater Palatal Cleft Width Predicts an Increased Risk for Unfavorable Outcomes in Cleft Palate Repair.2022In: The Cleft Palate-Craniofacial Journal, ISSN 1055-6656, E-ISSN 1545-1569, Vol. 59, no 8, p. 1030-1037Article in journal (Refereed)
    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.

  • 5.
    Okhiria, Åsa
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Surgical Sciences, Plastic Surgery.
    Persson, Christina
    Blom Johansson, Monica
    Hakelius, Malin
    Jabbari, Fatemeh
    Nowinski, Daniel
    The impact of surgical technique and cleft width on the rate of secondary surgery and velopharyngeal functionManuscript (preprint) (Other academic)
  • 6.
    Okhiria, Åsa
    et al.
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Surgical Sciences, Plastic Surgery.
    Persson, Christina
    Institute of Neuroscience and Physiology, Speech and Language Pathology Unit, Sahlgrenska Academy, University of Gothenburg, Gothenburg, Sweden;Region Västra Götaland, Department of Otorhinolaryngology Sahlgrenska University Hospital Gothenburg Sweden.
    Blom Johansson, Monica
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Public Health and Caring Sciences, Speech-Language Pathology. Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Public Health and Caring Sciences, Research in Disability and Habilitation.
    Hakelius, Malin
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Surgical Sciences, Plastic Surgery.
    Nowinski, Daniel
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Surgical Sciences, Plastic Surgery.
    Longitudinal data on speech outcomes in internationally adopted children compared with non‐adopted children with cleft lip and palate2023In: International journal of language and communication disorders, ISSN 1368-2822, E-ISSN 1460-6984, Vol. 58, no 5, p. 1440-1453Article in journal (Refereed)
    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.

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  • 7. Schölin, Johnna Sahlsten
    et al.
    Jonasson, Åsa
    Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Surgical Sciences. Institute of Neuroscience and Physiology, Speech Language Pathology Unit, Sahlgrenska Academy at University of Gothenburg, Gothenburg, Sweden.
    Axelsson, Jessica
    Havstam, Christina
    Persson, Christina
    Jönsson, Radi
    Mark, Hans
    Surgical, speech, and hearing outcomes at five years of age in internationally adopted children and Swedish-born children with cleft lip and/or palate2020In: Journal of Plastic Surgery and Hand Surgery, ISSN 2000-656X, E-ISSN 2000-6764, Vol. 54, no 1, p. 6-13Article in journal (Refereed)
    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.

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