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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., (© 2023 The Authors. International Journal of Language & Communication Disorders published by John Wiley & Sons Ltd on behalf of Royal College of Speech and Language Therapists.)

Objective: To evaluate the benefit of anatomical muscle dissection repair for velopharyngeal insufficiency (VPI) in patients with submucous cleft palate (SMCP) with 22q11.2 deletion syndrome., Design: Retrospective blinded randomised analysis of a surgeon's management over 10 years., Setting: The study was performed at a specialised Paediatric hospital in the United Kingdom., Patients: Children with SMCP and 22q11.2 deletion syndrome., Interventions: All participants underwent radical muscle dissection veloplasty., Outcomes Measured: Pre- and post- operative measurements included severity of anatomical defect, speech samples and lateral images which were digitised, randomised then externally and blindly analysed using validated techniques. Stata software was used to perform statistical analysis., Results: 57 children with 22q11.2 deletion syndrome were included in this analysis. Intra-operatively, the majority of cases were identified as SMCP Grade I anomalies. Post-operatively, a statistically significant improvement in hypernasality, resting palate length, palate length at maximum closure, palate excursion and gap size at maximum closure was observed. Secondary surgery was performed for 59% of patients by ten years., Conclusion: Muscle dissection repair improves hypernasality, palate closure function and the closure gap in patients with 22q11.2 deletion syndrome. Although over 50% of patients may require further surgery, muscle dissection repair should be a first step due to its utility at a younger age, when invasive investigations are impossible, its lower morbidity, speech and language benefits or altering the plans for less obstructive secondary surgery when it lead to reduced velo-pharyngeal gap and improved palate mobility even when adequate velo-pharyngeal closure was not achieved., Competing Interests: Declaration of Conflicting InterestsThe author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

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.

To delineate the indications, referral sources and outcomes of radical muscle dissection palate repair in the first 4 years of life for patients with submucous cleft palate (SMCP)., This study presents a retrospective analysis of a single surgeon's management., All children were treated at Great Ormond Street Hospital, United Kingdom., Twenty-three children with nonsyndromic SMCP were included in this study., All participants underwent radical muscle dissection repair before their fourth birthday., Parameters analyzed included: referral sources, indications for referral, extent of anatomical abnormality, and postoperative outcomes., Paediatricians provided the largest proportion of referrals to the cleft lip and palate clinic (39%) due to the presence of cleft lip/palate on prenatal scans or during neonatal examination. Over half (n = 12, 52%) of patients had severe anatomical anomalies being in grade IV (score 8-9), with the classical triad present to some extent in all but 2 of the patients. The main indication for surgery was nasal regurgitation of food and liquid (n = 9, 35%) followed by hypernasality (n = 6, 21%), difficulty feeding (n = 3, 8%), and severe anatomical defect (n = 2; 4%). Postoperatively, the presenting complaint improved in the majority of cases., Nonsyndromic SMCP is often missed, though may present with nasal regurgitation, feeding problems, and/or hypernasality, though may be missed in nonsyndromic children. Early radical muscle dissection repair in the first 4 years of life is safe and effective, facilitating normal speech development.

Objectives: To estimate both the association of surgical variables in complete unilateral cleft lip and palate (cUCLP) in the UK with outcomes at age 5 years, and the association of secondary speech surgery, volume of surgery, and surgeon with the same outcomes., Setting and Sample Population: The Cleft Care UK study, a cross-sectional study of 268 5-year-olds, born from 2005 to 2007, with cUCLP., Materials and Methods: Information on surgical variables was extracted from a standardized questionnaire. Dento-facial outcomes were derived from dental study casts of dental arch relationships. Three speech outcomes - intelligibility, structure and articulation - were derived using the Cleft Audit Protocol for Speech-Augmented tool., Results: Surgical and outcome data were available for 211 (79%) children from all cleft centres in the UK. Later soft palate surgery was associated with a 17% increased chance of a poor intelligibility score (P = .02), and high volume surgery with a 249% increased chance of a good articulation score (P = .01). There were no between surgeon effects identified. No association between the surgical variables examined and dento-facial outcome, or secondary speech surgery by the age of 5 years were found., Conclusion: This study found associations between surgical variables and speech outcomes at 5 years of age, but not between surgical variables and dento-facial outcome, nor between surgical variables and secondary speech surgery. High surgical volume should be maintained, and any changes towards later surgery monitored for changes in speech outcome., (© 2022 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.)

A consortium of global cleft professionals, predominantly from low- and middle-income countries, identified adaptations to cleft care protocols during and after COVID-19 as a priority learning area of need., A multidisciplinary international working group met on a videoconferencing platform in a multi-staged process to make consensus recommendations for adaptations to cleft protocols within resource-constrained settings. Feedback was sought from a roundtable discussion forum and global organizations involved in comprehensive cleft care., Foundational principles were agreed to enable recommendations to be globally relevant and two areas of focus within the specified topic were identified. First the safety aspects of cleft surgery protocols were scrutinized and COVID-19 adaptations, specifically in the pre- and perioperative periods, were highlighted. Second, surgical procedures and cleft care services were prioritized according to their relationship to functional outcomes and time-sensitivity. The surgical procedures assigned the highest priority were emergent interventions for breathing and nutritional requirements and primary palatoplasty. The cleft care services assigned the highest priority were new-born assessments, pediatric support for children with syndromes, management of acute dental or auditory infections and speech pathology intervention., A collaborative, interdisciplinary and international working group delivered consensus recommendations to assist with the provision of cleft care in low- and middle-income countries. At a time of global cleft care delays due to COVID-19, a united approach amongst global cleft care providers will be advantageous to advocate for children born with cleft lip and palate in resource-constrained settings.

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., (© 2021 The Author(s) Published by S. Karger AG, Basel.)

Background: Maxillary hypoplasia is a common skeletal condition in cleft lip and palate (CLP). Maxillary osteotomy is typically used to reposition the maxilla in CLP with maxillary hypoplasia. Previous studies have suggested that vowel articulations are adjusted postsurgically due to altered vocal tract configuration and articulatory reorganization. This acoustic study aims to investigate whether vowels are normalized postoperatively and to explore the nature of articulatory reorganization., Methods and Procedures: A prospective study was conducted to examine the vowel production of a group of individuals with CLP (N = 17) undergoing maxillary osteotomy and a group of normal controls (N = 20), using speech acoustic data. The data were collected at 0 to 3 months presurgery (T1), 3-months (T2), and 12-months (T3) postsurgery. General linear model repeated measures and independent t-tests were undertaken on F1, F2, and vowel space area., Results: General linear model repeated measures revealed no main effects of time for F1 (F [2, 22] = 1.094, P = 0.352), F2 (F [2, 22] = 1.269, P = 0.301), and vowel space area (F [2, 28] = 0.059, P = 0.943). Independent t-tests showed statistically significant differences (P < 0.05) for all acoustic parameters and all vowels between the CLP and the normal groups at all time points., Conclusions: Vowels were not normalized after maxillary osteotomy despite positive anatomical changes within the oral cavity. Individuals with CLP tended to adjust their vowel articulatory gestures to match presurgical patterns. The nature of articulatory reorganization appears to be prompt, sensory-driven, complete, and permanent., Competing Interests: The authors report no conflicts of interest., (Copyright © 2021 by Mutaz B. Habal, MD.)

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 (r s = 0.503-1.000, all p < 0.01) and strong to very strong reliability for videofluoroscopic measurements (r s = 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 ϰ 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 r s = 0.869, p < 0.01) and a weak correlation with unrelated parameters (e.g., amount of forward advancement r s = 0.160, p = 0.526). Criterion validity was found by a moderate correlation between closure ratio r s = -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., (© 2021 Royal College of Speech and Language Therapists.)

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.

Objective: To report speech and velopharyngeal function (VPF) outcomes, and surgical characteristics, at age 5 following early complete palate closure using the Sommerlad protocol., Design: A retrospective, descriptive, cross-sectional consecutive series., Setting: A regional twin site center; a district general hospital and tertiary children's hospital., Participants: Between 1993 and 2006, 877 participants underwent surgery; 712 (81%) were eligible for inclusion; 391 (55%) were included, 321 (45%) excluded. Thirteen percent had bilateral cleft lip and palate (CLP), 40% unilateral CLP, and 47% isolated cleft palate., Intervention: Lip and vomerine flap repair at 3 months of age (BCLP, UCLP) with soft palate closure using radical muscle dissection and retropositioning at a mean age of 6.6 months, range 4 to 23 months, as described and undertaken by Sommerlad., Outcome Measures: Velopharyngeal Composite (VPC) CAPS-A and articulation summary scores derived from analysis using the Cleft Audit Protocol for Speech-Augmented., Results: A VPC-SUM CAPS-A score of "0" was found in 97% reflecting adequate VPF; 73% had no cleft articulation difficulties. The BCLP group had the poorest articulation. Secondary speech surgery rate was 2.6% and 10.7% had fistula repair. Levator muscle quality and degree of retropositioning was associated with speech outcomes related to VPF. 2.6% had current features of VPI., Conclusions: Complete palate closure by around 6 months of age by the surgeon who described the technique led to excellent perceived VPF and very good speech outcomes at age 5, with less speech therapy and secondary speech surgery than commonly reported. These findings serve as a benchmark for the Sommerlad protocol and technique.

Background: A total of 68% of pre-school children with cleft palate have speech problems requiring speech therapy. There is a lack of access to regular targeted therapy. Parent training leads to positive outcomes in early communication skills in cleft palate and non-cleft speech disorders. Connected health has been used to address inadequate access to therapy, providing intervention to those who would not otherwise receive therapy., Aims: To evaluate the speech, activity and participation outcomes of Parent Led, Therapist Supervised, Articulation Therapy (PLAT) compared with routine speech therapy intervention in parent-child dyads., Methods & Procedures: A total of 44 children, aged 2.9-7.5 years, were included in a two-centre, two-phase randomized controlled trial. Informed consent and assent were obtained. Participants and speech and language therapists (SLTs) were unblinded to the groups. Parents, in the parent-trained group (n = 23), attended 2 days' training, received a detailed speech therapy programme, and undertook intervention over 12 weeks supported by the cleft specialist SLT using FaceTime and one face-to-face session. In the control arm (n = 21), parent-child dyads received six therapy sessions over 12 weeks with a research SLT, comparable with usual care. Speech recordings were undertaken pre- and post-intervention. Percent consonant correct (PCC) was analysed by external SLTs blinded to the time and group. Activity and participation were measured using the Intelligibility in Context Scale (ICS) and Focus on Outcomes for Children Under Six (FOCUS) questionnaire., Outcomes & Results: There was no evidence of an interaction between Time and Group or an overall statistical difference between groups for PCC scores. There was a statistically significant difference over time for both groups (words: p < 0.002; confidence interval (CI) = 9.38-16.27; d = 0.57; sentences: p < 0.002; CI = 16.04-25.97; d = 0.23). Effect sizes were medium for words and small for sentences. For intelligibility and participation, there was no evidence of an interaction between Time and Group or an overall statistical difference between groups. A statistically significant difference over time was found for intelligibility (F = 29.97, d.f. = 1, 42, p < 0.001, 95 % CI = 1.45-3.15 d = 0.46) and for participation (F = 14.19, d.f. = 1, 41, p < 0.001 95% CI = 7.63-25.03; d = 0.36) with FOCUS results indicating clinically meaningful (parent-led group) and significant (control group) change in participation., Conclusions & Implications: PLAT can be as effective as routine care in changing speech, activity and participation outcomes for children with cleft palate, when supported by a specialist cleft SLT using connected health. What this paper adds What is already known on this subject Over 50% of children with cleft palate require speech therapy. However, there is a lack of timely, accessible speech therapy services in the UK and Ireland. Previous studies have shown that parents can deliver therapy effectively, and that connected health can support the delivery of speech therapy. This study aims to provide evidence that parent-led therapy with the supervision of a specialist cleft therapist using FaceTime is effective. What this paper adds to existing knowledge This randomized controlled trial indicates that parents can be trained to deliver therapy for children with cleft palate speech disorders, under the supervision of an SLT. This approach results in improved speech, activity and participation outcomes similar to routine care. What are the potential or actual clinical implications of this work? This study indicates that both parent-led articulation therapy and routine care showed meaningful gains in speech, activity and participation, and that parent-led articulation therapy when supported by a cleft SLT using connected health could be an additional service delivery model for children with cleft palate speech disorders., (© 2020 Royal College of Speech and Language Therapists.)

We present a family with a previously undescribed abnormality of the palate and oropharynx which involved the absence of the uvula and the anterior pillar of the fauces, rudimentary posterior pillar of the fauces, and hypernasality. Eight family members over 4 generations are affected in a pattern consistent with autosomal dominant inheritance. A causal role for the FOXF2 gene has been identified and previously reported. We describe the management of the proband, which involved attempting to lengthen the palate and to retroposition the abnormally anteriorly directed velar musculature, along with speech therapy.

Background: There is no consensus in the UK regarding the types of speech samples or parameters of speech that should be assessed at 3 years of age in children with cleft palate ± cleft lip (CP±L), despite cleft units routinely assessing speech at this age. The standardization of assessment practices would facilitate comparisons of outcomes across UK cleft units; earlier identification of speech impairments-which could support more timely treatments; and more reliable recording of therapy impacts and surgical interventions., Aims: To explore assessment practices used to assess speech in 3-year-old children with CP±L, including speech parameters, methods of assessment and the nature of the speech sample used., Methods & Procedures: A broad examination of the literature was undertaken through the use of a scoping review conducted in accordance with Joanna Briggs Institute guidelines. Search terms were generated from a preliminary search and then used in the main search (Medline, CINAHL, Embase, AMED and PsycINFO)., Main Contribution: A combination of approaches (medical, linguistic, developmental and functional) is required to assess CP±L speech at age 3. A developmental approach is recommended at this age, considering the complexity of speech profiles at age 3, in which typically developing speech processes may occur alongside cleft speech characteristics. A combined measure for both nasal emission and turbulence, and an overall measure for velopharyngeal function for speech, show potential for assessment at this age. Categorical ordinal scales are frequently used; the use of continuous scales has yet to be fully explored at age 3. Although single-word assessments, including a subset of words developed for cross-linguistic comparisons, are frequently used, more than one type of speech sample may be needed to assess speech at this age validly. The lack of consensus regarding speech samples highlights a need for further research into the types of speech samples 3-year-olds can complete; the impact of incomplete speech samples on outcome measures (particularly relevant at this age when children may be less able to complete a full sample); the impact of different speech samples on the validity of assessments; and the reliability of listener judgements., Conclusions & Implications: Whilst a medical model and linguistic approaches are often central in assessments of age-3 cleft speech, this review highlights the importance of developmental and functional approaches to assessment. Cross-linguistic single-word assessments show potential, and would facilitate the comparison of UK speech outcomes with other countries. Further research should explore the impact of different speech samples and rating scales on assessment validity and listener reliability., (© 2020 Royal College of Speech and Language Therapists.)

Background/aims: Percent consonant correct (PCC) was originally described by Shriberg and Kwiatkowski [J Speech Hear Disord. 1982 Aug;47(3):256-70] as a severity metric for phonological speech disorders, and has been adapted and used in many studies on speech sound disorders. It is well-recognized that cleft speech is complex, consisting of several interacting parameters assessed simultaneously, with error sounds not in the listener's own language. In speech outcome studies, narrow phonetic transcription and the reporting of intra- and inter-rater reliability are acknowledged as the gold standard. However, cleft speech brings special challenges to this task, as complex speech disorders are known to be associated with low transcriber agreement. Recent studies informed the decision to use PCC as the primary outcome measure in a cleft speech intervention study, given its common usage and familiarity. The aim was to specifically evaluate the intra- and inter-rater reliability of PCC in an intervention study, in contrast to other types of speech outcome studies., Methods: Two trained and experienced listeners analyzed 119 recordings, randomly selected from five data points before, during, and following intervention. The PCC score was separately calculated for words and sentences/phrases., Results: Using intraclass correlations (ICCs), Phase 1 results showed poor reliability for targets elicited for words (ICC = 0.07) and sentences/phrases (ICC = 0.42). Differences in classification of errors as glottal stops and consonant deletion accounted for this. Following further training, a second reliability study was undertaken showing improvement in the number of targets elicited in words (ICC = 0.85) and sentences/phrases (ICC = 0.94). There was very good inter-rater reliability for the PCC score on the word dataset (ICC = 0.9) and the sentence dataset (ICC = 0.88). Very good intra-rater reliability (ICC = 1.0) was found for the PCC score in both words and sentences/phrases for each listener. One listener consistently gave higher modified PCC scores., Conclusions: In cleft speech intervention studies, reliability of the number of targets elicited should be reported. Listeners need to distinguish between glottal articulation and consonant deletion, in order that the PCC score is meaningful. Attention should be paid to where listeners are reliable, but their pattern of scores consistently differs but in a consistent way. More research is needed on measuring the resolution of articulation difficulties in cleft intervention studies., (© 2019 S. Karger AG, Basel.)

Objective: We highlight a major study that investigated the impact of reconfigured cleft care in the United Kingdom some 15 years after centralization. We argue that centralization as an intervention has a major impact on outcomes., Setting: Audit clinics held in Cleft Centers in the United Kingdom., Patients, Participants: Five-year-olds born between April 1, 2005, and March 31, 2007, with nonsyndromic unilateral cleft lip and palate., Interventions: Centralization of cleft care., Main Outcome Measure(s): We collected routine clinical measures (speech recordings, hearing, photographs, models, oral health, psychosocial factors) in a very similar way to a previous survey., Results: We identified 359 eligible children and recruited 268 (74.7%) to the study. Overall, their outcomes were better post-centralization. There have been marked improvements in dentoalveolar arch relationships and in speech whereas the prevalence of dental caries and hearing loss are unchanged., Conclusions: Centralized cleft care has changed UK outcomes considerably and there is no argument for returning to a dispersed model of treatment.

Objective: To investigate the reliability of ordinal versus visual analogue scaling (VAS) ratings for perceptual judgments of nasal resonance, nasal airflow, understandability, and acceptability in speakers with cleft palate., Design: Within-subjects comparative study., Setting: Multisite., Participants: Five specialist speech and language therapists from U.K. Regional Cleft Centres., Outcome Measures: Participants rated 30 audio speech samples obtained from the Speech and Language Therapy archives of Great Ormond Street Hospital. They rated the identified speech parameters using each scaling method, with 1 month between rating tasks. The model of best fit was determined to examine validity, and both intra- and inter-rater reliability were also computed., Results: VAS ratings were valid for all parameters when plotted against ordinal ratings, and the model of best fit revealed only a slightly stronger curvilinear than linear relationship between the scaling methods. Intra-rater reliability was high for both rating methods across all six speech parameters. There was also high inter-rater reliability for both ordinal and VAS ratings of hypernasality, nasal emission, nasal turbulence, understandability, and acceptability, and for the ordinal ratings of hyponasality., Conclusions: Perceptual judgments of nasal resonance, nasal airflow, understandability, and acceptability were similar using VAS and ordinal scaling, indicating that both scaling methods were appropriate for measuring the cleft speech parameters. VAS, however, may offer statistical advantages, and there is a growing body of evidence advocating its use for the measurement of prothetic speech parameters.

Objective: The aim of this study was to investigate the impact of an intensive 10-week course of articulation therapy on articulation errors in cleft lip and palate patients who have Velopharyngeal Insufficiency (VPI), non-oral and passive cleft speech characteristics., Methods: Five children with cleft palate (+/-cleft lip) with VPI and non-oral and passive cleft speech characteristics underwent 40 intensive articulation therapies over 10 weeks in a single case experimental design. The percentage of non-oral CSCs (NCSCs), passive CSCs (PCSCs), stimulable consonants (SC), correct consonants in word imitation (CCI), and correct consonants in picture naming (CCN) were captured at baseline, during intervention and in follow up phases. Visual analysis and two effect size indexes of Percentage of Nonoverlapping Data and Percentage of Improvement Rate Difference were analyzed., Results: Articulation therapy resulted in visible decrease in NCSCs for all 5 participants across the intervention phases. Intervention was effective in changing percentage of passive CSCs in two different ways; it reduced the PCSCs in three cases and resulted in an increase in PCSCs in the other two cases. This was interpreted as intervention having changed the non-oral CSCs to consonants produced within the oral cavity but with passive characteristics affecting manner of production including weakness, nasalized plosives and nasal realizations of plosives and fricatives. Percent SC increased throughout the intervention period in all five patients. All participants demonstrated an increase in percentage of CCI and CCN suggesting an increase in the consonant inventory. Follow-up data showed that all the subjects were able to maintain their ability to articulate learned phonemes correctly even after a 4-week break from intervention., Conclusion: This single case experimental study supports the hypothesis that speech intervention in patients with VPI can result in an improvement in oral placements and passive CSCs., (Copyright © 2016 Elsevier Ireland Ltd. All rights reserved.)

Objective: To describe the results of two reliability studies and to assess the effect of training on interrater reliability scores., Design: The first study (1) examined interrater and intrarater reliability scores (weighted and unweighted kappas) and (2) compared interrater reliability scores before and after training on the use of the Cleft Audit Protocol for Speech-Augmented (CAPS-A) with British English-speaking children. The second study examined interrater and intrarater reliability on a modified version of the CAPS-A (CAPS-A Americleft Modification) with American and Canadian English-speaking children. Finally, comparisons were made between the interrater and intrarater reliability scores obtained for Study 1 and Study 2., Participants: The participants were speech-language pathologists from the Americleft Speech Project., Results: In Study 1, interrater reliability scores improved for 6 of the 13 parameters following training on the CAPS-A protocol. Comparison of the reliability results for the two studies indicated lower scores for Study 2 compared with Study 1. However, this appeared to be an artifact of the kappa statistic that occurred due to insufficient variability in the reliability samples for Study 2. When percent agreement scores were also calculated, the ratings appeared similar across Study 1 and Study 2., Conclusion: The findings of this study suggested that improvements in interrater reliability could be obtained following a program of systematic training. However, improvements were not uniform across all parameters. Acceptable levels of reliability were achieved for those parameters most important for evaluation of velopharyngeal function.

Background: Approximately 75% of children with cleft palate (CP) have Otitis Media with Effusion (OME) histories. Evidence for the effective management of OME in these children is lacking. The inconsistency in outcome measurement in previous studies has led to a call for the development of a Core Outcome Set (COS). Despite the increase in the number of published COS, involvement of patients in the COS development process, and methods to integrate the views of patients and health professionals, to date have been limited., Methods and Findings: A list of outcomes measured in previous research was identified through reviewing the literature. Opinion on the importance of each of these outcomes was then sought from key stakeholders: Ear, Nose and Throat (ENT) surgeons, audiologists, cleft surgeons, speech and language therapists, specialist cleft nurses, psychologists, parents and children. The opinion of health professionals was sought in a three round Delphi survey where participants were asked to score each outcome using a bespoke online system. Parents and children were also asked to score outcomes in a survey and provided an in-depth insight into having OME through semi-structured interviews. The results of the Delphi survey, interviews and parent/patient survey were brought together in a final consensus meeting with representation from all stakeholders. A final set of eleven outcomes reached the definition of "consensus in" to form the recommended COS: hearing; chronic otitis media (COM); OME; receptive language skills; speech development; psycho social development; acute otitis media (AOM); cholesteatoma; side effects of treatment; listening skills; otalgia., Conclusions: We have produced a recommendation about the outcomes that should be measured, as a minimum, in studies of the management of OME in children with CP. The development process included input from key stakeholders and used novel methodology to integrate the opinion of healthcare professionals, parents and children.

Objective : Submucous cleft palate (SMCP) is a congenital condition associated with abnormal development of the soft palate musculature. In a proportion of cases, this results in velopharyngeal insufficiency (VPI), the treatment for which includes pharyngeal flap surgery, pharyngoplasty, and palate reconstruction. The aim of this paper is to determine whether there is superiority of one or more types of surgical procedure over the others in improving speech in patients with VPI secondary to SMCP. Methodology : Nine databases, including MEDLINE and EMBASE, were searched between inception and January 2013 to identify articles published relating to the surgical management of SMCP. Only studies that reported outcome measures for postoperative speech were included in the systematic review. Results : Twenty-six studies analyzing the outcomes of surgery for VPI in patients with SMCP met the inclusion criteria. In these studies, speech outcomes were measured either in a binary fashion (i.e., normal speech or evidence of VPI) or using scales of VPI severity. Of the 26 studies, only two utilized blinded speech assessment, and 12 included both preoperative and postoperative speech assessment. Conclusions : The review found little evidence to support any specific surgical intervention. This is in large part due to the inclusion of mixed etiologies within study populations and the lack of unbiased validated preoperative and postoperative speech assessment. Further methodologically rigorous studies need to be conducted to provide a secure evidence base for the surgical management of SMCP.

Objective: To develop national standards for speech outcomes and processes of care for children with cleft palate ± lip and to test the standards using national data., Design, Setting, and Participants: In this large, multicenter, prospective cohort study, speech recordings of 1110 five-year-olds with cleft palate involvement (born 2001 to 2003) were collected by 12 cleft centers in Great Britain and Ireland. Recordings were analyzed by consensus by specialist speech and language therapists using the Cleft Audit Protocol for Speech-Augmented. Results were benchmarked against evidence-based process and speech outcome standards and statistical analysis undertaken., Results: From the 1110 children audited, 48% (530) had speech within the normal range. This was not significantly different from the agreed standard of 50% (P = .20, CI = 45-50%). Sixty-six percent (734) had speech with no evidence of structurally related speech problems or history of speech-related secondary surgery. This was significantly below the standard of 70% (P = .007, CI = 62-69%). Sixty percent (666) had no serious cleft-related articulation errors. This was significantly better than the agreed standard of 50% (P < .001, CI = 67-73%). More than 80% of 2-year-olds received a specialist speech and language assessment against a benchmark of 100%., Conclusions: Developing standards has facilitated more meaningful reporting of speech outcomes and treatment processes. Evidence-based standards were defined and extensively tested, enabling centers to compare their performance with national trends. One 5-year outcome standard was achievable; the other two standards will require modification through the mandatory annual national audit program.

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., (© 2013 Royal College of Speech and Language Therapists.)

Objective :  (1) To examine the evidence for the effectiveness of differences in timing and type of speech and language therapy for children with cleft palate with or without a cleft lip and (2) to identify types of interventions assessed. Design :  Nine databases, including MEDLINE and EMBASE, were searched between inception and March 2011 to identify published articles relating to speech and language therapy for children with cleft palate with or without cleft lip. Studies that included at least 10 participants and reported outcome measures for speech and/or language measures were included. Studies where the experimental group had less than 90% of children with cleft palate with or without cleft lip were excluded. Two reviewers independently completed inclusion assessment, data extraction, and risk of bias assessment for all studies identified. Results :  A total of 17 papers were evaluated: six randomized control trials and 11 observational studies. Studies varied widely on risk of bias, intervention used, and outcome measures reported. None of the studies had a low risk of bias. In terms of intervention approaches, seven studies evaluated linguistic approaches and 10 evaluated motor approaches. Outcomes measures did not support either approach over the other, and based on data reported it was difficult to ascertain which approach is more effective for children with cleft palate with or without cleft lip. Conclusions :  The review found little evidence to support any specific intervention. Key uncertainties need to be identified and adequately powered, methodologically rigorous studies conducted to provide a secure evidence base for speech-language therapy practice in children with cleft palate with or without cleft lip.

Background: Nasometry has supplemented perceptual assessments of nasality, using speech stimuli, which are devoid of nasal consonants. However, such speech stimuli are not representative of conversational speech. A weak relationship has been found in previous studies between perceptual ratings of hypernasality and nasalance scores for passages containing nasal consonants., Aims: This study aimed to evaluate the relationship between perceptual assessment and acoustic measurements of nasality using controlled speech stimuli., Methods & Procedure: A perceptual scale (the Temple Street Scale) describing nasality was devised for this study. Fifty children presenting with nasality were assessed using the Temple Street Scale and nasalance scores were obtained for specified speech samples using the Nasometer (Kay Elemetrics 6200.3). The relationship between the perceptual ratings and the nasometry results was evaluated using correlation analysis, test sensitivity, specificity, and overall efficiency., Outcomes & Results: Correlation coefficients for perceptual ratings of nasality and nasalance scores ranged from 0.69 to 0.74. The sensitivity of the Nasometer ranged from 0.83 to 0.88; its specificity ranged from 0.78 to 0.95; while its overall efficiency was between 0.82 and 0.92., Conclusions: The strong relationship between perceptual and acoustic assessments of nasality indicated that the Temple Street Scale and the Nasometer are both valid clinical tools for the evaluation of nasality when a carefully constructed speech sample is used. The need to use the Nasometer as a supplement to perceptual assessment is highlighted.

Objective: To achieve consistency and uniformity in reporting speech outcomes in individuals born with cleft palate with or without cleft lip using perceptual parameters that characterize their speech production behavior regardless of the language or languages spoken., Design: A working group of six individuals experienced in speech and cleft palate was formed to develop a system of universal parameters for reporting speech outcomes in individuals born with cleft palate. The system was adopted in conjunction with a workshop held in Washington, D.C., that was devoted to developing the universal system. The system, which was refined further following the workshop, involves a three-stage plan consisting of (1) evaluation, (2) mapping, and (3) reporting. The current report focuses primarily on the third stage, reporting speech outcomes., Results: A set of five universal speech parameters has been devised for the reporting stage. These consist of (1) hypernasality, (2) hyponasality, (3) audible nasal air emission and/or nasal turbulence, (4) consonant production errors, and (5) voice disorder. Also included are speech understandability and speech acceptability, global parameters that can be reported for any type of speech disorder. The parameters are described in detail, and guidelines for speech-sampling content and scoring procedures in relation to the parameters are presented., Conclusion: A plan has been developed to document speech outcomes in individuals with cleft palate, regardless of the spoken language, using a set of five universal reporting parameters and two global speech parameters.

Objectives: To develop an assessment tool for use in intercenter audit studies of cleft speech and to test its acceptability, validity, and reliability. The tool is to be used systematically to record and report speech outcomes, providing an indication of treatment needs and continuing burden of care., Setting: Regional Cleft Center, U.K., Methods: The Cleft Audit Protocol for Speech-Augmented (CAPS-A) was developed by three cleft speech experts who identified the key features required from existing assessment measures. Criterion validity was assessed by comparing the Cleft Audit Protocol for Speech-Augmented outcomes reported for 20 cases with clinical assessment results and other investigations. Intra- and interrater reliability were tested following the training of specialist speech and language therapists who used the Cleft Audit Protocol for Speech-Augmented on two occasions to assess 10 cases. The raters evaluated acceptability and ease of using a questionnaire., Results: The mean percentage agreement for criterion validity in each section was 87% (range 70% to 100%). Both intra- and interexaminer reliability were rated as good/very good (Kappa 0.61 to 1.00) for seven sections and moderate (Kappa 0.41 to 0.60) for three sections. Raters reported that the Cleft Audit Protocol for Speech-Augmented was acceptable and easy to use with appropriate training., Conclusion: An acceptable, valid, and reliable cleft speech audit tool has been developed based on a small sample. The Cleft Audit Protocol for Speech-Augmented is recommended for use in intercenter audit studies in the U.K. and Ireland and could be used in other English-speaking countries. In addition, it has wider applicability for use in reporting speech outcomes of surgical procedures.

Background: Perceptual speech assessment is central to the evaluation of speech outcomes associated with cleft palate and velopharyngeal dysfunction. However, the complexity of this process is perhaps sometimes underestimated., Aims: To draw together the many different strands in the complex process of perceptual speech assessment and analysis, and make recommendations for practice., Main Contribution: This review examines issues such as data sampling, data collection/recording, archiving, the advantages and disadvantages of lay and specialist listeners, approaches to data analysis, reliability, and data interpretation., Conclusions: The need to capture meaningfully the sound of speech based on detailed phonetic analysis is argued. Also described are some of the differences in measuring speech for clinical, audit and research activities. Blind independent analysis of speech data by specialist therapists is recommended as the gold standard methodological approach when reporting audit and research outcomes. The requirement for ongoing training in listening skills for specialist therapists is advocated. The limitations of an impairment-based-only approach to measurement are also illustrated, indicating the need to develop outcome measures that incorporate more functional issues that affect quality of life.

Objectives: This study was designed to determine whether velar surgery was worthwhile for submucous cleft palate (SMCP) and evaluate whether results were dependent on the degree of the anatomical abnormality., Design: A prospective study of a consecutive series of patients fulfilling the entry criteria, assessed blindly from records arranged randomly., Patients: Fifty-eight patients diagnosed with SMCP and operated on by a single surgeon between June 1991 and April 1997 were reviewed. Forty patients fulfilled the entry criteria. Minimum follow-up was 6 years., Intervention: Radical reconstruction of the soft palate musculature was performed by one surgeon using the operating microscope. A scoring system was devised for grading the anatomical severity of submucous cleft (SMCP score)., Main Outcome Measures: Postoperative hypernasality and nasal emission scores and the degrees of improvement were considered the primary outcome measures, and the degree of velopharyngeal closure was also assessed., Results: There were highly significant improvements in hypernasality, nasal emission, and velopharyngeal closure. A preoperative gap size of more than 13 mm was associated with less satisfactory outcomes, but gap size was not predictive of improvement. Severity of the SMCP did not correlate with the degree of preoperative speech abnormality but was a significant predictor of outcome of surgery, with the less severe (total SMCP score of 0 to 3) having less satisfactory end results and lesser degrees of improvement. Patients with less abnormal muscle anatomy had lesser degrees of improvement., Conclusion: Repair of the muscle abnormality in SMCP is recommended as the first line of treatment in most cases.

Objectives: To analyze the results of surgery for velopharyngeal incompetence (VPI) in velocardiofacial syndrome., Design: Prospective data collection, with randomized, blind assessment of speech and velopharyngeal function on lateral videofluoroscopy and nasendoscopy., Setting: Two-site, tertiary referral cleft unit., Patients: Forty-two consecutive patients with the 22q11 deletion underwent surgery for symptomatic VPI by a single surgeon., Interventions: Intraoral examinations, lateral videofluoroscopy (+/- nasendoscopy) and intraoperative evaluation of the position of the velar muscles through the operating microscope. Based on these findings, either a radical dissection and retropositioning of the velar muscles (submucous cleft palate [SMCP repair]) or a Hynes pharyngoplasty (posterior pharyngeal wall augmentation pharyngoplasty) was performed. As anticipated, a proportion of patients undergoing SMCP repair subsequently required a Hynes. The aim of this staged approach was to maximize velar function, thereby enabling a less obstructive pharyngoplasty to be performed. Thus, there were three surgical groups for analysis: SMCP alone, Hynes alone, and SMCP+Hynes., Main Outcome Measures: Blind perceptual rating of resonance and nasal airflow; blind assessment of velopharyngeal function on lateral videofluoroscopy and nasendoscopy; and identification of predictive factors., Results: Significant improvement in hypernasality in all three groups. The SMCP+Hynes group also showed significant improvement in nasal emission. There were significant improvements in the extended and resting velar lengths following SMCP repair and a trend toward increased velocity of closure., Conclusions: Depending on velopharyngeal anatomy and function, there is a role for SMCP repair, Hynes pharyngoplasty, and a staged combination of SMCP+Hynes, all of which are procedures with a low morbidity.

Background and Aims: Impaired velopharyngeal closure function is sometimes a complication of a standard Le Fort I maxillary advancement in cleft palate patients. The transpalatal Le Fort I osteotomy has been suggested as an alternative technique that may avoid this problem. The aim of this pilot study was to examine the effects of the transpalatal approach on velopharyngeal function in a series of cleft palate patients., Patients: Sixteen consecutive patients with a history of cleft palate exhibiting maxillary hypoplasia who underwent a transpalatal Le Fort I osteotomy., Methods: All patients had a simultaneous audio/video speech recording and nasopharyngoscopy examination prior to maxillary advancement, followed by a repeat of the same examinations at least 1 year post-operatively. Velopharyngeal function was measured in two ways: by direct observation using nasopharyngoscopy, and indirectly by means of perceptual assessment. Reliability studies of the two measures were performed with satisfactory results., Results: No statistically significant difference was found between the pre- and post-operative data in either the perceptual speech assessment or nasopharyngoscopy examination., Conclusion: These results indicate that maxillary advancement by transpalatal Le Fort I osteotomy does not adversely affect velopharyngeal closure function., (Copyright 2002 European Association for Cranio-Maxillofacial Surgery. Published by Elsevier Science Ltd.)

Objective: To analyze the results of a consecutive series of palate re-repairs performed using the operating microscope and identify predictive factors for outcome., Design: Prospective data collection, with blind assessment of randomized recordings of speech and velar function on lateral videofluoroscopy and nasendoscopy., Patients: One hundred twenty-nine consecutive patients with previously repaired cleft palates and symptomatic velopharyngeal incompetence (VPI) and evidence of anterior insertion of the levator veli palatini underwent palate re-repairs by a single surgeon from 1992 to 1998. Syndromic patients, those who had significant additional surgical procedures at the time of re-repair (23 patients), and all patients with inadequate pre- or postoperative speech recordings were excluded, leaving a total of 85 patients in the study., Interventions: Palate re-repairs, with radical dissection and retropositioning of the velar muscles, were performed using the operating microscope with intraoperative grading of anatomical and surgical findings., Main Outcome Measures: Pre- and postoperative perceptual speech assessments using the Cleft Audit Protocol for Speech (CAPS) score, measurement of velar function on lateral videofluoroscopy, and assessment of nasendoscopy recordings., Results: There were significant improvements in hypernasality, nasal emission, and nasal turbulence and measures of velar function on lateral videofluoroscopy, with improvement in the closure ratio, velopharyngeal gap at closure, velar excursion, velar movement angle, and velar velocity., Conclusions: Palate re-repair has been shown to be effective in treating VPI following cleft palate repair, both in patients who have not had an intravelar veloplasty and those who have had a previous attempt at muscle dissection and retropositioning. Palate re-repair has a lower morbidity and is more physiological than a pharyngoplasty or pharyngeal flap.

Objective: To evaluate the benefit of anatomical muscle dissection repair for velopharyngeal insufficiency (VPI) in patients with submucous cleft palate (SMCP) with 22q11.2 deletion syndrome. Design: Retrospective blinded randomised analysis of a surgeon's management over 10 years. Setting: The study was performed at a specialised Paediatric hospital in the United Kingdom. Patients: Children with SMCP and 22q11.2 deletion syndrome. Interventions: All participants underwent radical muscle dissection veloplasty. Outcomes measured: Pre- and post- operative measurements included severity of anatomical defect, speech samples and lateral images which were digitised, randomised then externally and blindly analysed using validated techniques. Stata software was used to perform statistical analysis. Results: 57 children with 22q11.2 deletion syndrome were included in this analysis. Intra-operatively, the majority of cases were identified as SMCP Grade I anomalies. Post-operatively, a statistically significant improvement in hypernasality, resting palate length, palate length at maximum closure, palate excursion and gap size at maximum closure was observed. Secondary surgery was performed for 59% of patients by ten years. Conclusion: Muscle dissection repair improves hypernasality, palate closure function and the closure gap in patients with 22q11.2 deletion syndrome. Although over 50% of patients may require further surgery, muscle dissection repair should be a first step due to its utility at a younger age, when invasive investigations are impossible, its lower morbidity, speech and language benefits or altering the plans for less obstructive secondary surgery when it lead to reduced velo-pharyngeal gap and improved palate mobility even when adequate velo-pharyngeal closure was not achieved. [ABSTRACT FROM AUTHOR]

Purpose: This study aimed to investigate the experiences of parents of children with cleft-related speech sound disorders who had received in-depth training, following which they were the active agents of intervention, supported by a specialist cleft therapist. Method: All parents (N = 23) who had participated in the parent training arm of a previously published two-phase, two-center, 12-week randomized controlled trial in Dublin, Ireland, and London, United Kingdom, were invited to participate in the study. Eighteen families consented, and in three families, both parents took part, resulting in 21 participants. Sixteen parents participated in one of the four focus groups conducted in each location after each phase of the trial. Three families (n = 5) participated in semistructured telephone interviews. Focus groups and interviews were conducted by professionals who were not involved in the trial. Results: Focus groups and interviews were analyzed using a thematic analysis. Four main themes with subthemes emerged: growth of parents (knowledge, skills, insights, and emotional experiences), undertaking parent-led articulation therapy (bespoke program, flexibility, challenges, supervision/support), changes in the children (speech, confidence, behavior), and the future (ongoing therapy and recommendation of this approach). Conclusions: This study answered the research question, detailing how in-depth training and parent-led therapist-supervised interventions are acceptable to parents. Speech/language pathologists should encourage trained parents to take a direct role in intervention with their children, with supervision and support from therapists. Future research should investigate the experiences of parents with lower educational attainment, identify parent factors that supported implementation, and report children's experiences. [ABSTRACT FROM AUTHOR]

Objective : To delineate the indications, referral sources and outcomes of radical muscle dissection palate repair in the first 4 years of life for patients with submucous cleft palate (SMCP). Design : This study presents a retrospective analysis of a single surgeon's management. Setting : All children were treated at Great Ormond Street Hospital, United Kingdom. Patients : Twenty-three children with nonsyndromic SMCP were included in this study. Interventions : All participants underwent radical muscle dissection repair before their fourth birthday. Outcome Measured : Parameters analyzed included: referral sources, indications for referral, extent of anatomical abnormality, and postoperative outcomes. Results : Paediatricians provided the largest proportion of referrals to the cleft lip and palate clinic (39%) due to the presence of cleft lip/palate on prenatal scans or during neonatal examination. Over half (n = 12, 52%) of patients had severe anatomical anomalies being in grade IV (score 8-9), with the classical triad present to some extent in all but 2 of the patients. The main indication for surgery was nasal regurgitation of food and liquid (n = 9, 35%) followed by hypernasality (n = 6, 21%), difficulty feeding (n = 3, 8%), and severe anatomical defect (n = 2; 4%). Postoperatively, the presenting complaint improved in the majority of cases. Conclusion : Nonsyndromic SMCP is often missed, though may present with nasal regurgitation, feeding problems, and/or hypernasality, though may be missed in nonsyndromic children. Early radical muscle dissection repair in the first 4 years of life is safe and effective, facilitating normal speech development. [ABSTRACT FROM AUTHOR]

Objective : A consortium of global cleft professionals, predominantly from low- and middle-income countries, identified adaptations to cleft care protocols during and after COVID-19 as a priority learning area of need. Design : A multidisciplinary international working group met on a videoconferencing platform in a multi-staged process to make consensus recommendations for adaptations to cleft protocols within resource-constrained settings. Feedback was sought from a roundtable discussion forum and global organizations involved in comprehensive cleft care. Results : Foundational principles were agreed to enable recommendations to be globally relevant and two areas of focus within the specified topic were identified. First the safety aspects of cleft surgery protocols were scrutinized and COVID-19 adaptations, specifically in the pre- and perioperative periods, were highlighted. Second, surgical procedures and cleft care services were prioritized according to their relationship to functional outcomes and time-sensitivity. The surgical procedures assigned the highest priority were emergent interventions for breathing and nutritional requirements and primary palatoplasty. The cleft care services assigned the highest priority were new-born assessments, pediatric support for children with syndromes, management of acute dental or auditory infections and speech pathology intervention. Conclusions : A collaborative, interdisciplinary and international working group delivered consensus recommendations to assist with the provision of cleft care in low- and middle-income countries. At a time of global cleft care delays due to COVID-19, a united approach amongst global cleft care providers will be advantageous to advocate for children born with cleft lip and palate in resource-constrained settings. [ABSTRACT FROM AUTHOR]

Objective: To date, the recording of outcomes of interventions for velopharyngeal dysfunction (VPD) has not been standardized. This makes a comparison of results between studies challenging. The aim of this study was to develop a core outcome set (COS) for reporting outcomes in studies examining the management of VPD. Design: A two-round Delphi consensus process was used to develop the COS. Patients, Participants: The expert Delphi panel comprised patients and caregivers of patients with VPD, surgeons and speech and language therapists specializing in cleft palate, and researchers with expertise in VPD. Interventions: A long list of outcomes was derived from the published literature. In each round of a Delphi survey, participants were asked to score outcomes using the Grading of Recommendations, Assessment, Development, and Evaluations scale of 1 to 9, with 1 to 3 labeled "not important," 4 to 6 labeled "important but not critical," and 7 to 9 labeled "critical." Main outcome measure: Consensus criteria were specified a priori. Outcomes with a rating of 75% or more of the panel rating 7 to 9 and 25% or fewer rating 1 to 3 were included in the COS. Results: A total of 31 core outcomes were identified from the Delphi process. This list was condensed to combine topic areas to produce a final COS of 10 outcomes, including both processes of care and patient-reported outcomes that should be considered for reporting in future studies of VPD. Conclusions: Implementation of the COS-VPD will facilitate consistency of outcomes data collection and comparison of results across studies. [ABSTRACT FROM AUTHOR]

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. [ABSTRACT FROM AUTHOR]

Objective: To report speech and velopharyngeal function (VPF) outcomes, and surgical characteristics, at age 5 following early complete palate closure using the Sommerlad protocol. Design: A retrospective, descriptive, cross-sectional consecutive series. Setting: A regional twin site center; a district general hospital and tertiary children's hospital. Participants: Between 1993 and 2006, 877 participants underwent surgery; 712 (81%) were eligible for inclusion; 391 (55%) were included, 321 (45%) excluded. Thirteen percent had bilateral cleft lip and palate (CLP), 40% unilateral CLP, and 47% isolated cleft palate. Intervention: Lip and vomerine flap repair at 3 months of age (BCLP, UCLP) with soft palate closure using radical muscle dissection and retropositioning at a mean age of 6.6 months, range 4 to 23 months, as described and undertaken by Sommerlad. Outcome Measures: Velopharyngeal Composite (VPC) CAPS-A and articulation summary scores derived from analysis using the Cleft Audit Protocol for Speech-Augmented. Results: A VPC-SUM CAPS-A score of "0" was found in 97% reflecting adequate VPF; 73% had no cleft articulation difficulties. The BCLP group had the poorest articulation. Secondary speech surgery rate was 2.6% and 10.7% had fistula repair. Levator muscle quality and degree of retropositioning was associated with speech outcomes related to VPF. 2.6% had current features of VPI. Conclusions: Complete palate closure by around 6 months of age by the surgeon who described the technique led to excellent perceived VPF and very good speech outcomes at age 5, with less speech therapy and secondary speech surgery than commonly reported. These findings serve as a benchmark for the Sommerlad protocol and technique. [ABSTRACT FROM AUTHOR]

We present a family with a previously undescribed abnormality of the palate and oropharynx which involved the absence of the uvula and the anterior pillar of the fauces, rudimentary posterior pillar of the fauces, and hypernasality. Eight family members over 4 generations are affected in a pattern consistent with autosomal dominant inheritance. A causal role for the FOXF2 gene has been identified and previously reported. We describe the management of the proband, which involved attempting to lengthen the palate and to retroposition the abnormally anteriorly directed velar musculature, along with speech therapy. [ABSTRACT FROM AUTHOR]

Purpose: Speech and language disorders are hallmark features of 22q11.2 deletion syndrome (22qDS). Learning disabilities, cognitive deficits, palate abnormalities, velopharyngeal dysfunction, behavioral differences, and various medical and psychiatric conditions are also major features of this syndrome. The goal of this document is to summarize the state of the art of current clinical and scientific knowledge regarding 22qDS for speech-language pathologists (SLPs) and provide recommendations for clinical management. Method: Best practices for management of individuals with 22qDS were developed by consensus of an expert international group of SLPs and researchers with expertise in 22qDS. These care recommendations are based on the authors' research, clinical experience, and literature review. Results: This document describes the features of 22qDS as well as evaluation procedures, treatment protocols, and associated management recommendations for SLPs for the often complex communication disorders present in this population. Conclusion: Early diagnosis and appropriate management of speech-language disorders in 22qDS is essential to optimize outcomes and to minimize the long-term effects of communication impairments. Knowledge of this diagnosis also allows anticipatory care and guidance regarding associated features for families, health care, and educational professionals. [ABSTRACT FROM AUTHOR]

Objective: We highlight a major study that investigated the impact of reconfigured cleft care in the United Kingdom some 15 years after centralization. We argue that centralization as an intervention has a major impact on outcomes. Setting: Audit clinics held in Cleft Centers in the United Kingdom. Patients, Participants: Five-year-olds born between April 1, 2005, and March 31, 2007, with nonsyndromic unilateral cleft lip and palate. Interventions: Centralization of cleft care. Main Outcome Measure(s): We collected routine clinical measures (speech recordings, hearing, photographs, models, oral health, psychosocial factors) in a very similar way to a previous survey. Results: We identified 359 eligible children and recruited 268 (74.7%) to the study. Overall, their outcomes were better post-centralization. There have been marked improvements in dentoalveolar arch relationships and in speech whereas the prevalence of dental caries and hearing loss are unchanged. Conclusions: Centralized cleft care has changed UK outcomes considerably and there is no argument for returning to a dispersed model of treatment. [ABSTRACT FROM AUTHOR]

The article discusses research which investigated the reliability of ordinal against visual analogue scaling (VAS) ratings for perceptual judgments of nasal resonance, nasal airflow, understandability, and acceptability in speakers with cleft palate. Topics discussed include the rating of 30 audio speech samples taken from the Speech and Language Therapy archives of Great Ormond Street Hospital in Great Britain, and details of the research findings.

Objective: To describe the results of two reliability studies and to assess the effect of training on interrater reliability scores. Design: The first study (1) examined interrater and intrarater reliability scores (weighted and unweighted kappas) and (2) compared interrater reliability scores before and after training on the use of the Cleft Audit Protocol for Speech-Augmented (CAPS-A) with British English-speaking children. The second study examined interrater and intrarater reliability on a modified version of the CAPS-A (CAPS-A Americleft Modification) with American and Canadian English-speaking children. Finally, comparisons were made between the interrater and intrarater reliability scores obtained for Study 1 and Study 2. Participants: The participants were speech-language pathologists from the Americleft Speech Project. Results: In Study 1, interrater reliability scores improved for 6 of the 13 parameters following training on the CAPS-A protocol. Comparison of the reliability results for the two studies indicated lower scores for Study 2 compared with Study 1. However, this appeared to be an artifact of the kappa statistic that occurred due to insufficient variability in the reliability samples for Study 2. When percent agreement scores were also calculated, the ratings appeared similar across Study 1 and Study 2. Conclusion: The findings of this study suggested that improvements in interrater reliability could be obtained following a program of systematic training. However, improvements were not uniform across all parameters. Acceptable levels of reliability were achieved for those parameters most important for evaluation of velopharyngeal function. [ABSTRACT FROM AUTHOR]

Objective: Submucous cleft palate (SMCP) is a congenital condition associated with abnormal development of the soft palate musculature. In a proportion of cases, this results in velopharyngeal insufficiency (VPI), the treatment for which includes pharyngeal flap surgery, pharyngoplasty, and palate reconstruction. The aim of this paper is to determine whether there is superiority of one or more types of surgical procedure over the others in improving speech in patients with VPI secondary to SMCP. Methodology: Nine databases, including MEDLINE and EMBASE, were searched between inception and January 2013 to identify articles published relating to the surgical management of SMCP. Only studies that reported outcome measures for postoperative speech were included in the systematic review. Results: Twenty-six studies analyzing the outcomes of surgery for VPI in patients with SMCP met the inclusion criteria. In these studies, speech outcomes were measured either in a binary fashion (i.e., normal speech or evidence of VPI) or using scales of VPI severity. Of the 26 studies, only two utilized blinded speech assessment, and 12 included both preoperative and postoperative speech assessment. Conclusions: The review found little evidence to support any specific surgical intervention. This is in large part due to the inclusion of mixed etiologies within study populations and the lack of unbiased validated preoperative and postoperative speech assessment. Further methodologically rigorous studies need to be conducted to provide a secure evidence base for the surgical management of SMCP. [ABSTRACT FROM AUTHOR]

Objective: To develop national standards for speech outcomes and processes of care for children with cleft palate ± lip and to test the standards using national data. Design, Setting, and Participants: In this large, multicenter, prospective cohort study, speech recordings of 1110 five-year-olds with cleft palate involvement (born 2001 to 2003) were collected by 12 cleft centers in Great Britain and Ireland. Recordings were analyzed by consensus by specialist speech and language therapists using the Cleft Audit Protocol for Speech-Augmented. Results were benchmarked against evidence-based process and speech outcome standards and statistical analysis undertaken. Results: From the 1110 children audited, 48% (530) had speech within the normal range. This was not significantly different from the agreed standard of 50% (P = .20, CI = 45-50%). Sixty-six percent (734) had speech with no evidence of structurally related speech problems or history of speech-related secondary surgery. This was significantly below the standard of 70% (P= .007, CI = 62-69%). Sixty percent (666) had no serious cleft-related articulation errors. This was significantly better than the agreed standard of 50% (P < .001, CI = 67-73%). More than 80% of 2-year-olds received a specialist speech and language assessment against a benchmark of 100%. Conclusions: Developing standards has facilitated more meaningful reporting of speech outcomes and treatment processes. Evidence-based standards were defined and extensively tested, enabling centers to compare their performance with national trends. One 5-year outcome standard was achievable; the other two standards will require modification through the mandatory annual national audit program. [ABSTRACT FROM AUTHOR]

This review is part of a series of papers by the editorial board reflecting on the 1964 volume of the journal, thereby marking the 50th anniversary of the Cleft-Palate Craniofacial Journal. Twelve speech-related contributions from that year are reviewed. The papers are sorted into four broad thematic categories: quantitative assessment of hypernasality, perceptual assessment of speech, radiological imaging of velopharyngeal function, and speech prosthetics. The 1964 contributions are discussed with regards to their relevance to today's research and clinical practice. [ABSTRACT FROM AUTHOR]

Objective: To undertake a critical and systematic review of the literature on the impact of maxillary advancement on speech outcomes in order to identify current best evidence. Design and Main Outcome Measures: The following databases were searched: PubMed, CINAHL, and The Cochrane Controlled Trials Register. In addition, reference lists were hand searched for additional articles. Using a predefined framework and set criteria, evidence was evaluated using the assignment of levels of evidence (at least Level III on the evidence hierarchy), calculation of post-hoc power (⩾ 0.8), effect size (Cohen's d ⩾ 0.5), and adaptation of the parameters as set out by The Cochrane Collaboration. Results: Of the 40 studies identified, the majority (68%) fell within Level lll.ii, representing cohort-type studies and a fifth (20%) within Level IV, the weakest form of evidence. Power and effect size calculations were only possible in 9 studies for different speech outcomes, and only seven studies met the set criteria for best evidence. Accordingly, current best evidence for articulation exists only for a noncleft population, is conflicting for resonance and nasalance, and is mixed for velopharyngeal function depending on which instrumental measure is used. Conclusions: There is an obvious need for further prospective research in the field with strong speech methodology such as the undertaking of interrater and intrarater reliability, adequate follow-up, and sufficient sample sizes based on a priori power analyses. Methodologic issues are discussed and recommendations made. [ABSTRACT FROM AUTHOR]