How differences in anatomy and physiology and other aetiology affect the way we label and describe speech in individuals with cleft lip and palate.

BACKGROUND: Speech in individuals with cleft lip and/or palate (CLP) is a complex myriad of presenting symptoms. It is uniquely associated with the structural difference of velopharyngeal insufficiency (VPI), together with a wide and heterogeneous range of other aetiologies which often co-occur. The nature of the speech sound disorder (SSD) including VPI may also change over the course of an individual's care pathway. Differences in terminology and approaches to analysis are currently used, resulting in confusion internationally. Additionally, current diagnostic labels and classification systems in SSD do not capture the complexity and full nature of speech characteristics in CLP. AIMS: This paper aims to explore the different aetiologies of cleft palate/VPI speech and to relate aetiology with speech characteristic(s). In so doing, it attempts to unravel the different terminology used in the field, describing commonalities and differences, and identifying overlaps with the speech summary patterns used in the United Kingdom and elsewhere. The paper also aims to explore the applicability of current diagnostic labels and classification systems in the non-cleft SSD literature and illustrate certain implications for speech intervention in CLP. METHODS AND PROCEDURES: The different aetiologies were identified from the literature and mapped onto cleft palate/VPI speech characteristics. Different terminology and approaches to analysis are defined and overlaps described. The applicability of current classification systems in SSD is discussed including additional diagnostic labels proposed in the field. OUTCOMES AND RESULTS: Aetiologies of cleft palate/VPI speech identified include developmental (cognitive-linguistic), middle ear disease and fluctuating hearing loss, altered oral structure, abnormal facial growth, VPI-structural (abnormal palate muscle) and VPI-iatrogenic (maxillary advancement surgery). There are four main terminologies used to describe cleft palate/VPI speech: active/passive and compensatory/obligatory, which overlap with the four categories used in the UK speech summary patterns: anterior oral cleft speech characteristics (CSCs), posterior oral CSCs, non-oral CSCs and passive CSCs, although not directly comparable. Current classification systems in non-cleft SSD do not sufficiently capture the full nature and complexity of cleft palate/VPI speech. CONCLUSIONS AND IMPLICATIONS: Our attempt at identifying the heterogeneous range of aetiologies provides clinicians with a better understanding of cleft palate/VPI speech to inform the management pathway and the nature and type of speech intervention required. We hope that the unravelling of the different terminology in relation to the UK speech summary patterns, and those used elsewhere, reduces confusion and provides more clarity for clinicians in the field. Diagnostic labels and classification require international agreement. WHAT THIS PAPER ADDS: What is already known on the subject Speech associated with cleft palate/velopharyngeal insufficiency (VPI) is a complex myriad of speech characteristics with a wide and heterogeneous range of aetiologies. Different terminology and speech summary patterns are used to describe the speech characteristics. The traditional classification of cleft palate/VPI speech is Articulation Disorder, although evidence is building for Phonological Disorder and contrastive approaches in cleft speech intervention. What this paper adds to existing knowledge This paper explores the range of aetiologies of cleft palate/VPI speech (e.g., altered oral structure, abnormal facial growth, abnormal palate muscle and iatrogenic aetiologies) and attempts to relate aetiology with speech characteristic(s). An attempt is made at unravelling the different terminology used in relation to a well-known and validated approach to analysis, used in the United Kingdom and elsewhere. Complexities of current diagnostic labels and classifications in Speech Sound Disorder to describe cleft palate/VPI speech are discussed. What are the potential or actual clinical implications of this work? There needs to be a common language for describing and summarising cleft palate/VPI speech. Speech summary patterns based on narrow phonetic transcription and correct identification of aetiology are essential for the accurate classification of the speech disorder and identification of speech intervention approaches. There is an urgent need for research to identify the most appropriate type of contrastive (phonological) approach in cleft lip and/or palate.

The First Hybrid International Educational Comprehensive Cleft Care Workshop.

OBJECTIVE: Describe the first hybrid global simulation-based comprehensive cleft care workshop, evaluate impact on participants, and compare experiences based on in-person versus virtual attendance. DESIGN: Cross-sectional survey-based evaluation. SETTING: International comprehensive cleft care workshop. PARTICIPANTS: Total of 489 participants. INTERVENTIONS: Three-day simulation-based hybrid comprehensive cleft care workshop. MAIN OUTCOME MEASURES: Participant demographic data, perceived barriers and interventions needed for global comprehensive cleft care delivery, participant workshop satisfaction, and perceived short-term impact on practice stratified by in-person versus virtual attendance. RESULTS: The workshop included 489 participants from 5 continents. The response rate was 39.9%. Participants perceived financial factors (30.3%) the most significant barrier and improvement in training (39.8%) as the most important intervention to overcome barriers facing cleft care delivery in low to middle-income countries. All participants reported a high level of satisfaction with the workshop and a strong positive perceived short-term impact on their practice. Importantly, while this was true for both in-person and virtual attendees, in-person attendees reported a significantly higher satisfaction with the workshop (28.63 ± 3.08 vs 27.63 ± 3.93; P = .04) and perceived impact on their clinical practice (22.37 ± 3.42 vs 21.02 ± 3.45 P = .01). CONCLUSION: Hybrid simulation-based educational comprehensive cleft care workshops are overall well received by participants and have a positive perceived impact on their clinical practices. In-person attendance is associated with significantly higher satisfaction and perceived impact on practice. Considering that financial and health constraints may limit live meeting attendance, future efforts will focus on making in-person and virtual attendance more comparable.

Speech Telepractice and Treatment Intensity in a Cantonese-Speaking Case with 22q11.2 Deletion Syndrome Following Late Diagnosis and Management of Velopharyngeal Dysfunction.

This case report explores clinical treatment efficacy in a Cantonese-speaking child with 22q11.2 Deletion Syndrome where diagnosis and management of velopharyngeal dysfunction can be considered late. All treatment sessions were undertaken via telepractice during the peak of the COVID-19 pandemic in Hong Kong. A hybrid of specialized cleft palate speech treatment techniques and traditional treatment approaches in Speech Sound Disorders were utilized. Treatment intensity components including dose, dose form, session duration, and total intervention duration were documented.

The Impact of Maxillary Osteotomy on Fricatives in Cleft Lip and Palate: A Perceptual Speech and Acoustic Study.

INTRODUCTION: Abnormal facial growth is a recognized outcome in cleft lip and palate (CLP), resulting in a concave profile and a class III occlusal status. Maxillary osteotomy (MO) is undertaken to correct this facial deformity, and the surgery can impact speech articulation, although the evidence remains limited and ill-defined for the CLP population. AIMS: The aim of the study was to investigate the impact of MO on the production of the fricatives /f/ and /s/, using perceptual and acoustic analyses, and to explore the nature of speech changes. METHODS: Twenty participants with CLP were seen 0-3 months pre-operatively (T1) and 3 months (T2) and 12 months (T3) after MO. A normal group (N = 20) was similarly recruited. Perceptual speech data was collected according to a validated framework and ratings made on audio and audio-video recordings (VIDRat). Spectral moments were centre of gravity (CG), standard deviation (SD), skewness (SK) and kurtosis (KU). Reliability studies were carried out for all speech analyses. RESULTS: For the CLP group, VIDRat identified dentalization/interdentalization as the main type of pre-operative error for /s/ with a statistically significant improvement over time, χ2(2) = 6.889, p = 0.032. Effect sizes were medium between T1 and T3 (d = 0.631) and small between T2 and T3 (d = 0.194). For the acoustic data, effect sizes were similarly medium between T1 and T2 (e.g., SK, /f/ d = 0.579, /s/ d = 0.642) and small between T1 and T3 across all acoustic parameters. Independent t tests showed mainly statistically significant differences between both groups at all time points with large effect sizes (e.g., T2 CG, t = -4.571, p < 0.001, d =1.581), indicating that /s/ was not normalized post-operatively. For /f/, differences tended to be at T1 with large effect sizes (e.g., CG, t = -2.307, p = 0.028, d = 0.797), reflecting normalization. CONCLUSIONS AND IMPLICATIONS: This is the first speech acoustic study on /f/ for individuals with CLP undergoing MO. The surgery has a positive impact on /f/ and /s/, which appear to stabilize 3 months post-operatively. Speech changes are an automatic and a direct consequence of the physical changes brought about by MO, effecting articulatory re-organization. The results of the study have direct clinical implications for the clinical care pathway for patients with CLP undergoing MO.

A perceptual outcome measure of velopharyngeal function based on the Cleft Audit Protocol for Speech-Augmented (CAPS-A VPC-Sum): Validation through a speech osteotomy study.

BACKGROUND: The status of the velopharyngeal mechanism can be inferred from perceptual ratings of specified speech parameters. Several studies have proposed the measure of an overall velopharyngeal composite score based on these perceptual ratings and have reported good validity. The Cleft Audit Protocol for Speech-Augmented (CAPS-A) is a validated and reliable perceptual framework for the assessment of cleft speech and velopharyngeal function used by all Regional Cleft Services in the UK and Ireland. An overall velopharyngeal composite summary score based on the CAPS-A would serve as an important surgical outcome measure of speech. AIMS: To develop and validate a velopharyngeal composite summary score based on perceptual ratings made on the CAPS-A (CAPS-A VPC-Sum) using data from a maxillary osteotomy (MO) study. METHODS & PROCEDURES: There were two surgical groups: a cleft lip and palate (CLP) (N = 20) group and a non-CLP group (N = 10), and a normal control group (N = 20). Participants in groups 1 and 2 were seen for perceptual and instrumental assessments of speech and velopharyngeal function preoperatively (T1), 3 months (T2) and 12 months (T3) postoperatively. Perceptual speech data were collected and rated by independent listeners using CAPS-A. OUTCOMES & RESULTS: Moderate to strong interrater reliability for perceptual data (rs = 0.503-1.000, all p < 0.01) and strong to very strong reliability for videofluoroscopic measurements (rs = 0.746-0.947) were found. Construct validity of the CAPS-A VPC-Sum was shown by an increase in postoperative scores for the CLP group only Ï°2 (2) = 9.769, p = 0.008 and significant differences between the CLP and the other two groups at T2 and T3 using independent t-tests. Convergent and divergent validity was indicated by a positive moderate correlation with related parameters (e.g., hypernasality rs = 0.869, p < 0.01) and a weak correlation with unrelated parameters (e.g., amount of forward advancement rs = 0.160, p = 0.526). Criterion validity was found by a moderate correlation between closure ratio rs = -0.541, p = 0.020 and CAPS-A VPC-Sum. CONCLUSIONS & IMPLICATIONS: A velopharyngeal composite score based on perceptually rated parameters serves as an important surgical speech outcome measure. The CAPS-A VPC-Sum is a useful, reliable and valid outcome measure of velopharyngeal function. There are added positive implications for other clinicians using geographically and language-specific adapted versions of the CAPS-A internationally. WHAT THIS PAPER ADDS: What is already known on this subject Velopharyngeal composite scores based on perceptually rated speech parameters have been shown to have both clinical and research utility, serving as a useful surgical outcome measure. However, such a composite score must be specifically validated on the perceptual speech framework upon which it is based, as there are differences in measurement methods and terminology across cleft speech perceptual frameworks internationally. What this paper adds to existing knowledge The CAPS-A is a nationally used tool in the UK and Ireland for audit and research purposes with validated and adapted international versions. This paper reports on the validation of the derived velopharyngeal composite score measure based on the CAPS-A and an English-speaking sample, providing evidence of its validity through a speech osteotomy study. What are the potential or actual clinical implications of this work? This work provides CLP teams who use CAPS-A with a validated surgical speech outcome measure of velopharyngeal function. It has positive implications also for adapted versions of the CAPS-A internationally.

Identifying Predictors of Acquired Velopharyngeal Insufficiency in Cleft Lip and Palate Following Maxillary Osteotomy Using Multiple Regression Analyses.

BACKGROUND: Maxillary osteotomy is typically undertaken to correct abnormal facial growth in cleft lip and palate. The surgery can cause velopharyngeal insufficiency resulting in hypernasality. This study aims to identify valid predictors of acquired velopharyngeal insufficiency following maxillary osteotomy by using a range of perceptual and instrumental speech investigations and multiple regression. METHODS: A prospective study was undertaken consisting of a consecutive series of patients with cleft lip and palate (N = 20) undergoing maxillary osteotomy by a single surgeon. Participants were seen at: 0 to 3 months pre-surgery (T1), 3-months (T2), and 12-months (T3) post-surgery. Hypernasality was rated using the cleft audit protocol for speech-augmented (CAPS-A) and visual analog scales, and nasalance was measured on the Nasometer II 6400. For lateral videofluorosopic and nasendoscopic images, visual perceptual ratings and quantitative ratiometric measurements were undertaken. Multiple regression analyses were undertaken to identify predictors. RESULTS: T3 models with hypernasality as the dependent variable were found to be a good fit and significant (eg, CAPS-A: R2 = 0.920, F(11,7) = 7.303, P = 0.007). Closure ratio (a quantitative ratiometric measurement) and proportion of palate contacting the posterior pharyngeal wall (a visual perceptual rating) were identified as significant predictors for the CAPS-A model (P = 0.030, P = 0.002).

Effect of Maxillary Osteotomy on Speech in Cleft Lip and Palate: Instrumental Outcomes of Velopharyngeal Function.

OBJECTIVE: To investigate the effect of maxillary osteotomy on velopharyngeal function in cleft lip and palate (CLP) using instrumental measures. DESIGN: A prospective study. PARTICIPANTS: A consecutive series of 20 patients with CLP undergoing maxillary osteotomy by a single surgeon were seen at 0 to 3 months presurgery (T1), 3 months (T2), and 12 months (T3) post-surgery. INTERVENTIONS: Nasalance was measured on the Nasometer II 6400. For videofluoroscopy and nasendoscopy data, visual perceptual ratings, for example, palatal lift angle (PLAn), and quantitative ratiometric measurements, for example, closure ratio (CRa), were made using a validated methodology and computer software. Reliability studies were undertaken for all instrumental measures. MAIN OUTCOME MEASURES: Repeated measures analysis of variance (with time at 3 levels) for nasalance and each velar parameter. Planned comparisons across pairs of time points (T1-T2, T1-T3, and T2-T3) including effect sizes. RESULTS: A significant difference over time was found for nasalance (P = .001) and planned comparisons across pairs of time points were significant between T1 and T2 (P = .008), T1 and T3 (P = .002), but not between T2 and T3 (P = .459) providing evidence that maxillary osteotomy can impact on nasalance adversely and that the changes seen are permanent and stable. There were also significant differences over time for PLAn (P = .012) and CRa (P = -.059) and planned comparisons for both velar parameters reflected similar findings to those of nasalance. CONCLUSIONS: Maxillary osteotomy can adversely affect velopharyngeal function in patients with CLP. The study provides evidence for a much earlier post-surgery review even as early as 3 months after surgery.

Effect of maxillary osteotomy on speech in cleft lip and palate: perceptual outcomes of velopharyngeal function.

BACKGROUND: Abnormal facial growth is a well-known sequelae of cleft lip and palate (CLP) resulting in maxillary retrusion and a class III malocclusion. In 10-50% of cases, surgical correction involving advancement of the maxilla typically by osteotomy methods is required and normally undertaken in adolescence when facial growth is complete. Current evidence for the impact of the surgery on velopharyngeal function is weak and mixed. AIMS: The first objective of the study was to investigate the nature of the effect of maxillary osteotomy on the perceptual outcomes of velopharyngeal function in CLP. The second objective was to establish if speech changes seen early at 3 months post-operation persisted for a year after/following surgery', when it is considered that the maxilla is relatively stable. METHODS & PROCEDURES: Twenty consecutive patients with CLP undergoing maxillary osteotomy by a single surgeon were seen pre-operatively (T1), 3 months (T2) and 12 months (T3) post-operation. A non-cleft control group (NonCLP) undergoing surgery was also recruited. Speech data were collected using the Cleft Audit Protocol for Speech-Augmented (CAPS-A). A velopharyngeal composite score-summary (VPC-SUM) was derived from specific CAPS-A-rated parameters. An external CAPS-A-trained therapist, blinded to the study, rated the randomized samples and inter-rater reliability was established. OUTCOMES & RESULTS: For the CLP group, hypernasality and nasal turbulence increased significantly post-operation. Planned comparisons were significant for T1-T2 only with a medium effect size. For hypernasality, the CLP group differed statistically from the NonCLP group at T2 and T3. For nasal turbulence, the CLP group differed statistically from the NonCLP group at T2. For VPC-SUM, there were statistically significant changes post-operatively between T1-T2 and T1-T3 only with medium effect sizes for the CLP group only. CONCLUSIONS & IMPLICATIONS: This study provides evidence that maxillary osteotomy affects patients with and without CLP differently. In patients with CLP, surgery may impact negatively on velopharyngeal function for speech and changes seen early on at 3 months post-operatively appear to persist at 12 months postoperatively. The findings in this study have implications for the speech care pathway of patients with CLP undergoing maxillary osteotomy in terms of assessment, review and management.