Timing of Primary Surgery for Cleft Palate.

BACKGROUND: Among infants with isolated cleft palate, whether primary surgery at 6 months of age is more beneficial than surgery at 12 months of age with respect to speech outcomes, hearing outcomes, dentofacial development, and safety is unknown. METHODS: We randomly assigned infants with nonsyndromic isolated cleft palate, in a 1:1 ratio, to undergo standardized primary surgery at 6 months of age (6-month group) or at 12 months of age (12-month group) for closure of the cleft. Standardized assessments of quality-checked video and audio recordings at 1, 3, and 5 years of age were performed independently by speech and language therapists who were unaware of the trial-group assignments. The primary outcome was velopharyngeal insufficiency at 5 years of age, defined as a velopharyngeal composite summary score of at least 4 (scores range from 0 to 6, with higher scores indicating greater severity). Secondary outcomes included speech development, postoperative complications, hearing sensitivity, dentofacial development, and growth. RESULTS: We randomly assigned 558 infants at 23 centers across Europe and South America to undergo surgery at 6 months of age (281 infants) or at 12 months of age (277 infants). Speech recordings from 235 infants (83.6%) in the 6-month group and 226 (81.6%) in the 12-month group were analyzable. Insufficient velopharyngeal function at 5 years of age was observed in 21 of 235 infants (8.9%) in the 6-month group as compared with 34 of 226 (15.0%) in the 12-month group (risk ratio, 0.59; 95% confidence interval, 0.36 to 0.99; P = 0.04). Postoperative complications were infrequent and similar in the 6-month and 12-month groups. Four serious adverse events were reported (three in the 6-month group and one in the 12-month group) and had resolved at follow-up. CONCLUSIONS: Medically fit infants who underwent primary surgery for isolated cleft palate in adequately resourced settings at 6 months of age were less likely to have velopharyngeal insufficiency at the age of 5 years than those who had surgery at 12 months of age. (Funded by the National Institute of Dental and Craniofacial Research; TOPS ClinicalTrials.gov number, NCT00993551.).

Analysis and Reporting of Randomized Trials in Cleft Palate Surgery: Learning from the Timing of Primary Surgery (TOPS) Trial.

The Timing of Primary Surgery (TOPS) trial was published August 2023 in the New England Journal of Medicine and is a milestone achievement for a study focused on cleft palate. Due to the complexity of outcome reporting in cleft and the rarity of such comparative trials, TOPS presents a useful opportunity to critically review the design, analysis and reporting strategies utilised. This perspective article focused on the inclusion of participants, the choice of the primary outcome measure and the analysis of ordinal data within the trial. Considerations for future comparative studies in cleft care are discussed.

Inter-rater reliability in classification of canonical babbling status based on canonical babbling ratio in infants with isolated cleft palate randomised to Timing of Primary Surgery for Cleft Palate (TOPS).

Canonical babbling (CB) is commonly defined as present when at least 15% of all syllables produced are canonical, in other words a canonical babbling ratio (CBR) ≥0.15. However, there is limited knowledge about inter-rater reliability in classification of CB status based on CBR and inter-rater differences in assessment of CBR. We investigated inter-rater reliability of experienced Speech Language Therapists (SLTs) on: classification of CB status based on CBR ≥ 0.15, CBRs and the total number of syllables per infant used to calculate CBR.Each infant (n = 484) was video-recorded at a clinical site in play interaction with their parent as part of the randomised controlled trial Timing of Primary Surgery for Cleft Palate. Each recording was subsequently assessed by three independent SLTs, from a pool of 29 SLTs. They assessed the recordings in real time.The three assessing SLTs agreed in classification of CB status in 423 (87.4%) infants, with higher complete agreement for canonical (91%; 326/358) than non-canonical (77%; 97/126). The average difference in CBR and total number of syllables identified between the SLT assessments of each infant was 0.12 and 95, respectively.This study provided new evidence that one trained SLT can reliably classify CB status (CBR ≥ 0.15) in real time when there is clear distinction between the observed CBR and the boundary (0.15); however, when the observed CBR approaches the boundary multiple SLT assessments are beneficial. Thus, we recommend to include assessment of inter-rater reliability, if the purpose is to compare CBR and total syllable count across infants or studies.Trial registration number here: www.clinicaltrials.gov, identifier NCT00993551.

A cross-sectional cohort study of speech in five-year-olds with cleft palate ± lip to support development of national audit standards: benchmarking speech standards in the United Kingdom.

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.

Articulating the issues: speech assessment and intervention in cleft lip and palate.

This paper describes the impact of cleft lip and palate on speech. It provides an overview for the dental clinician of the key issues affecting speech development and clarity. The paper summarises the complex speech mechanism and cleft-related factors that affect speech, including palatal, dental and occlusal anomalies. It outlines the framework for speech assessment throughout the cleft pathway and provides a description of cleft speech disorder, as well as describing treatment approaches for cleft speech and velopharyngeal dysfunction.This is followed by a spotlight on speech prosthetics for treating nasal speech, with an emphasis on joint management by the Speech and Language Therapist and Consultant in Restorative Dentistry. This includes the core multidisciplinary approach, clinician and patient-reported outcome measures, and brief discussion of national developments in this area.The importance of multidisciplinary cleft care is highlighted and, within this, the essential interaction between speech and dental clinicians in providing routine care, as well as in delivering a highly specialist speech prosthetics service.

Multiple Individual Audit Outcome Web--a novel tool to evaluate patient outcomes.

Multiple Individual Audit Outcome Web (MIAOW) is a pictorial representation of outcomes, which is used widely in industry, finance, and sports, but not, to the best of our knowledge, in our specialty. It was developed to display multiple audit outcomes for patients in cleft surgery and we show its use in that area, but equally it could be applied to other areas such as orthognathic surgery, and head and neck oncology. We report how it can be used to display surgical outcomes.