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  • 1
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    Characterising the speech phenotype in individuals with craniofacial microsomia: a scoping review protocol
    BMJ ; 2023
    In:  BMJ Open Vol. 13, No. 2 ( 2023-02), p. e069233-
    Details
    In: BMJ Open, BMJ, Vol. 13, No. 2 ( 2023-02), p. e069233-
    Abstract: Asymmetric mandibular hypoplasia, microtia, tongue and laryngeal anomalies, and soft palate and facial nerve dysfunction are clinical features observed in children with craniofacial microsomia (CFM). Despite involvement of all these structures in hearing and speech, there is limited evidence reporting speech outcomes in this population. Systematic reviews of clinical and surgical interventions related to CFM have been published, but no methodological review of speech outcomes exists. This scoping review will summarise what is known about speech production in individuals with CFM as well as illustrate gaps in the existing body of literature that will guide future research. Methods/analysis This review will follow the methodological framework for scoping reviews first reported by Arksey & O’Malley and revised by Levac and others. Databases searched will include Ovid MEDLINE, EMBASE, CINAHL, PsycINFO and grey literature. Articles reporting any parameter of speech production in individuals with CFM will be considered for inclusion. Articles published in a language other than English will be excluded. Articles will be screened in three stages: (1) title review, (2) abstract review and (3) full text review. Ten per cent of articles will be rescreened by a second reviewer. Reference lists will be hand reviewed to identify additional relevant articles. Data charting will capture article metadata, study population and design, CFM diagnostic criteria, speech outcome measurement and key findings. The Preferred Reporting Systems for Systematic Reviews and Meta-Analyses Protocols-Extension for Scoping Reviews checklist will guide reporting of results. Descriptive analysis and data visualisation strategies will be used. Ethics and dissemination Institutional review board approval is not required for a scoping review, as it does not directly involve human subjects. Results will be disseminated through peer-reviewed publication as well as conference presentation.
    Type of Medium: Online Resource
    ISSN: 2044-6055 , 2044-6055
    URL: Article
    URL: Article
    DOI: 10.1136/bmjopen-2022-069233
    DOI: 10.1136/bmjopen-2022-069233.supp1
    Language: English
    Publisher: BMJ
    Publication Date: 2023
    detail.hit.zdb_id: 2599832-8
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  • 2
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    Behavioral Adjustment of Preschool Children With and Without Craniofacial Microsomia
    SAGE Publications ; 2021
    In:  The Cleft Palate-Craniofacial Journal Vol. 58, No. 1 ( 2021-01), p. 42-53
    Details
    In: The Cleft Palate-Craniofacial Journal, SAGE Publications, Vol. 58, No. 1 ( 2021-01), p. 42-53
    Abstract: The study aim was to assess behavioral adjustment in preschool children with and without craniofacial microsomia (CFM). Design: Multisite cohort study of preschoolers with CFM (“cases”) or without CFM (“controls”). Participants: Mothers (89%), fathers (9%), and other caregivers (2%) of 161 preschoolers. Outcome Measure: Child Behavior Check List (CBCL 1.5-5); linear regressions with standardized effect sizes (ES) adjusted for sociodemographic confounds. Results: Child Behavior Check Lists for 89 cases and 72 controls (average age 38.3 ± 1.9 months). Children were male (54%), white (69%), and of Latino ethnicity (47%). Cases had microtia with mandibular hypoplasia (52%), microtia only (30%), or other CFM-associated features (18%). Nearly 20% of cases had extracranial anomalies. Composite CBCL scores were in the average range compared to test norms and similar for cases and controls. On the subscales, cases’ parents reported higher Anxious/Depressed scores (ES = 0.35, P = .04), Stress Problems (ES = 0.40, P = .04), Anxiety Problems (ES = 0.34, P = .04), and Autism Spectrum Problems (ES = 0.41, P = .02); however, the autism subscale primarily reflected speech concerns. Among cases, more problems were reported for children with extracranial anomalies and certain phenotypic categories with small ES. Conclusions: Behavioral adjustment of preschoolers with CFM was comparable to peers. However, parental reports reflected greater concern for internalizing behaviors; thus, anxiety screening and interventions may benefit children with CFM. Among cases, more problems were reported for those with more complex presentations of CFM. Craniofacial microsomia–related speech problems should be distinguished from associated psychosocial symptoms during developmental evaluations.
    Type of Medium: Online Resource
    ISSN: 1055-6656 , 1545-1569
    URL: Article
    DOI: 10.1177/1055665620947987
    RVK:
    XA 36625
    Language: English
    Publisher: SAGE Publications
    Publication Date: 2021
    detail.hit.zdb_id: 2030056-6
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  • 3
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    Crowdsourcing to Assess Speech Quality Associated With Velopharyngeal Dysfunction
    SAGE Publications ; 2021
    In:  The Cleft Palate-Craniofacial Journal Vol. 58, No. 1 ( 2021-01), p. 25-34
    Details
    In: The Cleft Palate-Craniofacial Journal, SAGE Publications, Vol. 58, No. 1 ( 2021-01), p. 25-34
    Abstract: To assess crowdsourced responses in the evaluation of speech outcomes in children with velopharyngeal dysfunction (VPD). Design: Fifty deidentified speech samples were compiled. Multiple pairwise comparisons obtained by crowdsourcing were used to produce a rank order of speech quality. Ratings of overall and specific speech characteristics were also collected. Twelve speech-language pathologists (SLPs) who specialize in VPD were asked to complete the same tasks. Crowds and experts completed each task on 2 separate occasions at least 1 week apart. Setting: On-line crowdsourcing platform. Participants: Crowdsource raters were anonymous and at least 18 years of age, North American English speakers with self-reported normal hearing. Speech-language pathologists were recruited from multiple cleft/craniofacial teams. Interventions: None. Main Outcome Measure(s): Correlation of repeated assessments and comparison of crowd and SLP assessments. Results: We obtained 6331 lay person assessments that met inclusion criteria via crowdsourcing within 8 hours. The crowds provided reproducible Elo rankings of speech quality, ρ(48) = .89; P 〈 .0001, and consistent ratings of intelligibility and acceptability (intraclass correlation coefficient [ICC] = .87 and .92) on repeated assessments. There was a significant correlation of those crowd rankings, ρ(10) = .86; P = .0003, and ratings (ICC = .75 and .79) with those of SLPs. The correlation of more specific speech characteristics by the crowds and SLPs was moderate to weak (ICC 〈 0.65). Conclusions: Crowdsourcing shows promise as a rapid way to obtain large numbers of speech assessments. Reliability of repeated assessments was acceptable. Large groups of naive raters yield comparable evaluations of overall speech acceptability, intelligibility, and quality, but are not consistent with expert raters for specific speech characteristics such as resonance and nasal air emission.
    Type of Medium: Online Resource
    ISSN: 1055-6656 , 1545-1569
    URL: Article
    DOI: 10.1177/1055665620948770
    RVK:
    XA 36625
    Language: English
    Publisher: SAGE Publications
    Publication Date: 2021
    detail.hit.zdb_id: 2030056-6
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  • 4
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    Validity and Responsiveness of VELO: A Velopharyngeal Insufficiency Quality of Life Measure
    Wiley ; 2013
    In:  Otolaryngology–Head and Neck Surgery Vol. 149, No. 2 ( 2013-08), p. 304-311
    Details
    In: Otolaryngology–Head and Neck Surgery, Wiley, Vol. 149, No. 2 ( 2013-08), p. 304-311
    Abstract: Test the Velopharyngeal Insufficiency (VPI) Effects on Life Outcomes (VELO) instrument for validity, reliability, and responsiveness. Study Design Observational cohort. Setting Academic tertiary medical center. Subjects Children with VPI (n = 59) and their parents (n = 84) were prospectively enrolled from a pediatric VPI clinic. Methods Pediatric speech language pathologists diagnosed VPI using perceptual speech analysis and rated VPI severity and speech intelligibility deficit (each as minimal, mild, moderate, or severe). All parents and youth 8+ years old (n = 24) completed the VELO instrument and other quality‐of‐life questionnaires at baseline; the first 40 subjects completed the VELO instrument again 2 weeks later. Treatments included Furlow palatoplasty (n = 20), sphincter pharyngoplasty (n = 14), or an obturator (n = 2), and 29 of 36 (81%) subjects completed the questionnaires 3 months posttreatment. VELO was tested with correlations for criterion validity against VPI severity, construct validity against speech intelligibility and velopharyngeal gap size, and concurrent validity against other quality‐of‐life measures ( r 〉 . 40 demonstrating validity); for test‐retest reliability using intraclass correlation ( 〉 .6 demonstrating reliability); and for responsiveness with the 3‐month posttreatment measure using the paired t test. Results Parental responses are reported; youth responses showed similar results. The VELO instrument did not meet criterion validity ( r = –.18, P =. 10), or functional construct validity ( r = –.37, P =. 001), but did meet anatomic construct and concurrent validity (each r 〉 . 50, P 〈 . 01). VELO scores demonstrated excellent test‐retest reliability ( r =. 85, P 〈 . 001) and responsiveness (baseline 54 ± 14 to posttreatment 70 ± 18, P 〈 . 001). Conclusion VELO provides a VPI‐specific quality‐of‐life instrument that demonstrates concurrent validity, test‐retest reliability, and responsiveness to change in quality of life with treatment.
    Type of Medium: Online Resource
    ISSN: 0194-5998 , 1097-6817
    URL: Article
    DOI: 10.1177/0194599813486081
    Language: English
    Publisher: Wiley
    Publication Date: 2013
    detail.hit.zdb_id: 2008453-5
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  • 5
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    Speech Outcomes After Sphincter Pharyngoplasty for Velopharyngeal Insufficiency
    Wiley ; 2021
    In:  The Laryngoscope Vol. 131, No. 6 ( 2021-06)
    Details
    In: The Laryngoscope, Wiley, Vol. 131, No. 6 ( 2021-06)
    Abstract: To investigate perceptual speech outcomes following sphincter pharyngoplasty (SP) and to identify patient characteristics associated with velopharyngeal insufficiency (VPI) resolution or improvement. Methods Retrospective review of prospectively collected data was performed of consecutive patients that underwent SP for management of VPI between 1994 and 2016 at a single tertiary care pediatric hospital. Demographic data, nasendoscopic findings, and speech characteristics were recorded using a standardized protocol. Pre‐ and post‐operative VPI was graded on a five‐point Likert scale. Frequency of post‐operative VPI resolution and improvement was assessed and associations with patient characteristics were analyzed. The association between odds of VPI resolution or improvement and five patient characteristics identified a priori was performed controlling for confounding factors. Results Two‐hundred ninety‐six subjects were included. All patients had at least minimal VPI pre‐operatively; 72% were graded moderate or severe. Sixty‐four percent experienced resolution and 83% improved at least one point on the VPI‐severity scale. Of the five patient characteristics, only history of cleft palate repair was significantly associated with decreased odds of VPI improvement but not resolution when controlling for other variables. Conclusions Sphincter pharyngoplasty resulted in resolution of VPI in 64% and improvement in 83% of subjects. Children with a history of cleft palate had significantly decreased odds of VPI improvement compared to those without a history of cleft palate. Neither syndrome diagnosis nor 22q11 deletion had a significant association with speech outcomes after sphincter pharyngoplasty. Level of Evidence 4 Laryngoscope , 131:E2046–E2052, 2021
    Type of Medium: Online Resource
    ISSN: 0023-852X , 1531-4995
    URL: Issue
    URL: Article
    DOI: 10.1002/lary.v131.6
    DOI: 10.1002/lary.29189
    Language: English
    Publisher: Wiley
    Publication Date: 2021
    detail.hit.zdb_id: 2026089-1
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  • 6
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    Abstract: Speech and Surgical Outcomes in International Adoptees with Cleft Palate
    Ovid Technologies (Wolters Kluwer Health) ; 2016
    In:  Plastic and Reconstructive Surgery - Global Open Vol. 4, No. 9S ( 2016-09), p. 16-17
    Details
    In: Plastic and Reconstructive Surgery - Global Open, Ovid Technologies (Wolters Kluwer Health), Vol. 4, No. 9S ( 2016-09), p. 16-17
    Type of Medium: Online Resource
    ISSN: 2169-7574
    URL: Article
    DOI: 10.1097/01.GOX.0000502896.99994.01
    Language: English
    Publisher: Ovid Technologies (Wolters Kluwer Health)
    Publication Date: 2016
    detail.hit.zdb_id: 2723993-7
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  • 7
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    Sphincter Pharyngoplasty for Velopharyngeal Dysfunction: Impact of 22q11.2 Deletion Syndrome
    Wiley ; 2023
    In:  The Laryngoscope Vol. 133, No. 10 ( 2023-10), p. 2813-2820
    Details
    In: The Laryngoscope, Wiley, Vol. 133, No. 10 ( 2023-10), p. 2813-2820
    Abstract: Patients with 22q11.2 deletion syndrome (22q11DelS) often present with velopharyngeal dysfunction (VPD). VPD in patients with 22q11DelS is multifactorial beyond velopharyngeal insufficiency (VPI) alone, and differences in surgical outcomes are poorly understood. Our objective was to determine whether patients with 22q11DelS have an increased risk for persistent VPI after sphincter pharyngoplasty compared to patients without 22q11DelS. Methods We completed a retrospective cohort study of patients with 22q11DelS undergoing sphincter pharyngoplasty between 1995 and 2019 using a VPD clinic database. Patients with 22q11DelS were compared to a cohort of 2:1 frequency‐matched (age, degree of velopharyngeal closure) patients without 22q11DelS. Variables included patient characteristics, surgical history, perceptual speech evaluation, and degree of closure on nasopharyngoscopic evaluations. Primary outcomes included postoperative VPI severity and hypernasality. Speech and nasopharyngoscopic characteristics were compared using Fisher's exact test. Postoperative VPI severity and hypernasality were compared between groups via relative risks (RR) from mixed effects Poisson regression models, with random effects of age and velopharyngeal closure. Results 134 patients (51 22q11DelS, 83 matched) were included, with mean age of 7.3 years (standard deviation 3.0) and 50% male. Cohorts had similar preoperative speech characteristics and nasopharyngoscopic findings. Patients with 22q11DelS had similar postoperative VP function as patients without 22q11DelS (RR 0.85, CI 0.46–1.57 for VPI severity, RR 0.83, CI 0.45–1.53 for hypernasality). Even after adjusting by preoperative variables, no differences were seen between both groups. Conclusion Matched for age and pre‐operative velopharyngeal closure, patients with and without 22q11DelS and VPI had similar benefits after sphincter pharyngoplasty. Level of Evidence Non‐randomized controlled cohort study, 3 Laryngoscope , 133:2813–2820, 2023
    Type of Medium: Online Resource
    ISSN: 0023-852X , 1531-4995
    URL: Issue
    URL: Article
    DOI: 10.1002/lary.v133.10
    DOI: 10.1002/lary.30579
    Language: English
    Publisher: Wiley
    Publication Date: 2023
    detail.hit.zdb_id: 2026089-1
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  • 8
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    Modification and Evaluation of a Velopharyngeal Insufficiency Quality-of-Life Instrument
    American Medical Association (AMA) ; 2012
    In:  Archives of Otolaryngology–Head & Neck Surgery Vol. 138, No. 10 ( 2012-10-01), p. 929-
    Details
    In: Archives of Otolaryngology–Head & Neck Surgery, American Medical Association (AMA), Vol. 138, No. 10 ( 2012-10-01), p. 929-
    Type of Medium: Online Resource
    ISSN: 0886-4470
    URL: Article
    DOI: 10.1001/2013.jamaoto.122
    Language: English
    Publisher: American Medical Association (AMA)
    Publication Date: 2012
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  • 9
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    Home Language Environment of Children With Orofacial Clefts as a Target for Intervention
    Ovid Technologies (Wolters Kluwer Health) ; 2021
    In:  Journal of Craniofacial Surgery Vol. 32, No. 2 ( 2021-03), p. 500-504
    Details
    In: Journal of Craniofacial Surgery, Ovid Technologies (Wolters Kluwer Health), Vol. 32, No. 2 ( 2021-03), p. 500-504
    Abstract: To evaluate the home language environment (HLE) in children with orofacial clefts as a potential modifiable target for language and literacy intervention. Design: Feasibility study examining longitudinal trends in HLE and responses to parent-focused literacy intervention. Setting: Tertiary care children's hospital. Participants: HLE data were collected for 38 children with orofacial clefts between ages 7 and 23 months. Twenty-seven participants received parent-focused literacy intervention. Interventions: Reach Out and Read, a literacy intervention, was introduced during a clinic visit. To assess response, participants were randomized to age at intervention (9, 18, or 24 months). Main Outcome Measures: Primary outcome measures included measurements from recordings in the home language environment of adult word count, child vocalizations, and conversational turns. Results: Baseline (preintervention) results showed lower adult word count and conversational turns for caregivers and children with cleft lip and palate, as well as for those from lower socioeconomic groups. After the literacy intervention was introduced, this cohort showed increasing measures of child and caregiver vocalizations, particularly when introduced at 18 months. Conclusions: Although these results are preliminary, findings suggest that HLE characteristics vary as a function of children's cleft type as well as family socioeconomic status. Further, our caregiver-focused literacy intervention was feasible and resulted in short-term improvements in HLE. This is the first study to document HLE as a target for intervention in children with oral clefts. These findings support further research on HLE and caregiver-focused intervention to improve language/literacy outcomes for children with oral clefts.
    Type of Medium: Online Resource
    ISSN: 1049-2275 , 1536-3732
    URL: Article
    DOI: 10.1097/SCS.0000000000007060
    Language: English
    Publisher: Ovid Technologies (Wolters Kluwer Health)
    Publication Date: 2021
    detail.hit.zdb_id: 2060546-8
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  • 10
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    Cleft Palate Repair and Velopharyngeal Dysfunction
    Ovid Technologies (Wolters Kluwer Health) ; 2014
    In:  Plastic & Reconstructive Surgery Vol. 133, No. 6 ( 2014-06), p. 852e-864e
    Details
    In: Plastic & Reconstructive Surgery, Ovid Technologies (Wolters Kluwer Health), Vol. 133, No. 6 ( 2014-06), p. 852e-864e
    Type of Medium: Online Resource
    ISSN: 0032-1052
    URL: Article
    DOI: 10.1097/PRS.0000000000000184
    Language: English
    Publisher: Ovid Technologies (Wolters Kluwer Health)
    Publication Date: 2014
    detail.hit.zdb_id: 2037030-1
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