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.).

Longitudinal study of the development of obstruent correctness from ages 3 to 5 years in 108 Danish children with unilateral cleft lip and palate: a sub-study within a multicentre randomized controlled trial.

BACKGROUND: Speech-sound development in preschoolers with unilateral cleft lip and palate (UCLP) as a group is delayed/disordered, and obstruents comprise the most vulnerable sound class. AIMS: To evaluate the development of obstruent correctness (PCC-obs) and error types (cleft speech characteristics (CSCs) and developmental speech characteristics (DSCs)) from ages 3-5 and to investigate possible predictors (error types, velopharyngeal dysfunction (VPD) and gender) of PCC-obs at age 5 in two groups of children with UCLP. METHODS & PROCEDURES: Subgroup analysis was conducted within a multicentre randomized controlled trial (RCT) of primary surgery (Scandcleft Project). A total of 125 Danish children with UCLP received lip and soft palate repair around 4 months of age and early hard palate closure at 12 months (EHPC group) or late hard palate closure at 36 months (LHPC group). Audio and video recordings of a naming test were available for 108 children at ages 3 and 5, and recordings were transcribed phonetically by blinded raters. OUTCOMES & RESULTS: PCC-obs scores increased significantly from ages 3-5 in both groups, but with small effect sizes in the EHPC group that had higher scores at age 3 than the LHPC group. DSCs decreased in both groups whereas CSCs only decreased in the LHPC group that had more CSCs at age 3 than the EHPC group. The frequency of CSCs at age 3 was a significant predictor of PCC-obs scores at age 5 in both groups. DSCs significantly improved the logistic regression model in the EHPC group, whereas VPD and gender did not significantly improve the model in either group. CONCLUSIONS & IMPLICATIONS: Although PCC-obs developed significantly from ages 3 to 5, children with UCLP as a group did not catch up to typically developing Danish children at age 5. Furthermore, the LHPC group at age 5 did not reach the 3-year level of the EHPC group, which means that delaying hard palate closure until age 3 is detrimental to obstruent development. Both CSCs and DSCs at age 3 were important predictors of PCC-obs at age 5 and should be considered when determining need for intervention.

Influence of timing of delayed hard palate closure on articulation skills in 3-year-old Danish children with unilateral cleft lip and palate.

BACKGROUND: Differing results regarding articulation skills in young children with cleft palate (CP) have been reported and often interpreted as a consequence of different surgical protocols. AIMS: To assess the influence of different timing of hard palate closure in a two-stage procedure on articulation skills in 3-year-olds born with unilateral cleft lip and palate (UCLP). Secondary aims were to compare results with peers without CP, and to investigate if there are gender differences in articulation skills. Furthermore, burden of treatment was to be estimated in terms of secondary surgery, hearing and speech therapy. METHODS & PROCEDURES: A randomized controlled trial (RCT). Early hard palate closure (EHPC) at 12 months versus late hard palate closure (LHPC) at 36 months in a two-stage procedure was tested in a cohort of 126 Danish-speaking children born with non-syndromic UCLP. All participants had the lip and soft palate closed around 4 months of age. Audio and video recordings of a naming test were available from 113 children (32 girls and 81 boys) and were transcribed phonetically. Recordings were obtained prior to hard palate closure in the LHPC group. The main outcome measures were percentage consonants correct adjusted (PCC-A) and consonant errors from blinded assessments. Results from 36 Danish-speaking children without CP obtained previously by Willadsen in 2012 were used for comparison. OUTCOMES & RESULTS: Children with EHPC produced significantly more target consonants correctly (83%) than children with LHPC (48%; p < .001). In addition, children with LHPC produced significantly more active cleft speech characteristics than children with EHPC (p < .001). Boys achieved significantly lower PCC-A scores than girls (p = .04) and produced significantly more consonant errors than girls (p = .02). No significant differences were found between groups regarding burden of treatment. The control group performed significantly better than the EHPC and LHPC groups on all compared variables.

Development and validation of a screening procedure to identify speech-language delay in toddlers with cleft palate.

The purpose of this study was to develop and validate a clinically useful speech-language screening procedure for young children with cleft palate ± cleft lip (CP) to identify those in need of speech-language intervention. Twenty-two children with CP were assigned to a +/- need for intervention conditions based on assessment of consonant inventory using a real-time listening procedure in combination with parent-reported expressive vocabulary. These measures allowed evaluation of early speech-language skills found to correlate significantly with later speech-language performance in longitudinal studies of children with CP. The external validity of this screening procedure was evaluated by comparing the +/- need for intervention assignment determined by the screening procedure to experienced speech-language pathologist (SLP)s' clinical judgement of whether or not a child needed early intervention. The results of real-time listening assessment showed good-excellent inter-rater agreement on different consonant inventory measures. Furthermore, there was almost perfect agreement between the children selected for intervention with the screening procedure and the clinical judgement of experienced SLPs indicate that the screening procedure is a valid way of identifying children with CP who need early intervention.