Speech in 7- and 10-year-olds born with a unilateral cleft lip and palate: a continued prospective Swedish intercentre study.

The aim of this study was to report longitudinal speech results in consecutively selected children from each of the six cleft centres in Sweden and to compare the results between centres. The children were born with a non-syndromic unilateral cleft lip and palate, and results from the same cohort at 5 years of age have previously been reported. Background data on medical care in terms of surgery, speech therapy, and hearing between 5 and 10 years of age were collected. Speech recordings of 56 children at 7 years and 54 at 10 years of age were blindly and independently assessed by four speech-language pathologists experienced in cleft palate speech. This resulted in measures of percent consonant correct (PCC) and perceived velopharyngeal competence rated on a three-tier scale. No statistically significant differences were found between centres. PCC scores at 7 years of age ranged from 44-100% (median 97.5) and at 10 years of age from 86-100% (median 100). Competent or marginally incompetent velopharyngeal function was found in 95% of the 7-year-olds and 98% of the 10-year-olds. Speech results were slightly better than previous reports of speech in children born with a unilateral cleft lip and palate.

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.).

Long-term longitudinal follow-up of individuals with UCLP after Gothenburg twostage palate closure: surgical and speech outcomes.

BACKGROUND: Delayed hard palate closure in unilateral cleft lip and palate (UCLP) patients show on a safe surgical method and good speech outcome, however, occurrence of orally retracted articulation before hard palate closure at 8 years. The aim of this study was to describe surgical and speech outcome in UCLP patients closing the hard palate at 3 years. METHODS: A consecutive of 28 participants were operated with Gothenburg two-stage method including soft palate closure at 6 months and hard palate at 3 years. Surgical and speech outcome were evaluated. Recordings of sentences and spontaneous speech at 5, 10, 16, and 19 years were analyzed blindly and independently by three speech-language pathologists. Compensatory articulation, hypernasality, hyponasality, weak pressure consonants, and nasal air leakage were evaluated on ordinal four-point and intelligibility and perceived velopharyngeal function on three-point scales. RESULTS: Long-term follow-up revealed a safe surgical method. Articulation disorders were present in 25-30% at 5-year but largely not later. About 20% had incompetent velopharyngeal function at 5 years but none at 19 years. Most participants were well intelligible after 5 years. Hard palate closure at 3 years indicated less occurrence of orally retracted articulation compared with a cohort who had hard palate closure at 8.2 years. CONCLUSIONS: Long-term, follow-up of individuals with UCLP after Gothenburg two-stage palate closure including closure of the soft palate closure at 6 months and hard palate at 3 years of age shows a safe surgical method and indicates less retracted oral articulation compared with hard palate closure at 8 years.

Inter-centre comparison of data on surgery and speech outcomes at 5 years of age based on the Swedish quality registry for patients born with cleft palate with or without cleft lip.

BACKGROUND: The objective of the Swedish cleft lip and palate registry (CLP registry) is to promote quality control, research and improvement of treatment, by the comparison of long-term results. The aim was to compare data from the CLP registry among the six treatment centres, regarding data on surgery and speech outcomes at 5 years of age. METHODS: The participants were 430 children born in Sweden from 2009 to 2014, with cleft palate with or without cleft lip and without known syndromes and/or additional malformations. The number of primary and secondary palatal surgeries up to 5 years of age, timing of the last primary palatal surgery, percentage consonants correct, percentage non-oral speech errors and perceived velopharyngeal competence at 5 years were assessed. Multivariable binary logistic regression adjusted for sex and cleft type was used to compare results between the six centres. RESULTS: At one centre (centre 4), the palate was closed in one to three stages, and at the remaining centres in one or two stages. At centre 4, more children underwent a higher number of palatal surgeries, and the last primary palatal surgery was performed at a higher age. Children in centre 4 were also less likely to achieve ≥86% correct consonants (OR = 0.169, P = < 0.001), have no non-oral speech errors (OR = 0.347, P = < 0.001), or have competent or marginally incompetent velopharyngeal competence (OR = 0.244, P = < 0.001), compared to the average results of the other centres. No clear association between patient volume and speech outcome was observed. CONCLUSIONS: The results indicated the risk of a negative speech result if the last primary palatal surgery was performed after 25 months of age. Whether the cleft in the palate was closed in one or two stages did not affect speech outcome. The Swedish CLP registry can be used for open comparisons of treatment results to provide the basis for improvements of treatment methods. If deviating negative results are seen consistently at one centre, this information should be acted upon by further investigation and analysis, making changes to the treatment protocol as needed.

Coverage, reporting degree and design of the Swedish quality registry for patients born with cleft lip and/or palate.

BACKGROUND: The objective of the Swedish cleft lip and palate (CLP) registry is to promote quality control, research and improvement of treatment, by comparison of the long-term results of surgery, orthodontics and speech from all six Swedish CLP centres. The purpose of the study was to investigate the coverage and reporting degree of the Swedish CLP registry, and to describe the design of the registry and discuss questions of reliability and validity of the data included. METHODS: All six Swedish CLP centres participate in the registry. All children in Sweden with cleft lip and/or cleft palate, born from 2009 onwards, are included in the registry. Baseline data such as cleft type (ICD-10 diagnosis), heredity, birth weight and additional deformities and/or syndromes, as well as pre-surgical treatment, are recorded at first visit. Data on surgical treatment are recorded continuously. Treatment outcome regarding dentofacial development and speech are recorded at follow-ups at 5, 10, 16 and 19 years of age. Data on dentofacial development are also recorded 1 year after orthognathic surgery. In addition, data on babbling and speech are recorded at 18 months of age. Coverage degree and reporting degree of surgery was assessed by comparison with registrations in the Swedish Central patient registry. Reporting degree of orthodontic and speech registrations at 5 years of age was assessed by comparison with registrations at baseline. RESULTS: The average coverage degree for children born 2009 to 2018 was 95.1%. For cleft-related surgeries, the average reporting degree was 92.4%. Average reporting degree of orthodontic registrations and speech registrations at age 5 years was 92 and 97.5% respectively. CONCLUSION: In order to achieve valid and reliable data in a healthcare quality registry, the degree of coverage and reporting needs to be high, the variables included should be limited and checked for reliability, and the professionals must calibrate themselves regularly. The Swedish CLP registry fulfils these requirements.

Palatal augmentation prostheses in individuals treated for head and neck cancer: Effects on speech and oral transport.

BACKGROUND: Negative side effects affecting articulation and oral transport are common after treatment for head and neck cancer (HNC). The aim of this study was to assess the effects on speech and patient-reported experience of palatal augmentation prostheses (PAP) in this patient group. METHODS: Twenty consecutive patients who had received a PAP were included. We performed a blinded randomized listener assessment of recordings of the patients' speech with and without the PAP. Patient-reported experiences were collected from medical records. RESULTS: The listener assessment showed that production of velar sounds improved significantly for 12 of 19 patients; 12 of 19 patients also reported easier or better speech with the PAP. All six patients treated for tongue cancer reported speech improvement. Seven of 20 patients reported better transport of food or saliva. CONCLUSION: A PAP can help patients treated for HNC to more precisely articulated velar consonants and can benefit oral transport.

Surgical, speech, and hearing outcomes at five years of age in internationally adopted children and Swedish-born children with cleft lip and/or palate.

Internationally adopted children (IAC) with a cleft lip and/or palate (CL/P) tend to arrive with un-operated palates at an age at which their Swedish-born peers have completed their primary palate surgery. Our aim of the present study was to analyze surgical, speech and hearing outcomes of IAC at age 5 and compare with those of a matched group of Swedish-born children. Fifty children with CL/P born in 1994-2005 participated in the study. Twenty-five IAC were matched according to age, sex and cleft type with 25 Swedish-born children. Audio recordings were perceptually analyzed by two experienced, blinded speech-language pathologists. Hearing and speech statuses were evaluated on the same day for all children. Surgical timing and complications as in fistulas and requirement for secondary velopharyngeal (VP) surgery, speech evaluation results, and present hearing status were analyzed for all children of age 5 years. Results showed that primary palatal surgery was delayed by a mean of 21 months in IAC. IAC had a higher prevalence of velopharyngeal impairment that was statistically significant, a higher fistula rate, and experienced more secondary surgery than Swedish-born peers. Hearing loss due to middle ear disease was slightly more common among IAC, whereas the rate of treatment with tympanostomy tubes was similar between the two groups. In conclusion, IAC with CL/P represent a challenge for CL/P teams because of the heterogeneous nature of the patient group and difficulties associated with delayed treatment, and the results show the importance of close follow-up over time.

Speech in 5-year-olds born with unilateral cleft lip and palate: a Prospective Swedish Intercenter Study.

Studies on the impact of cleft palate surgery on speech with stringent methodology are called for, since we still do not know the best timing or the best method for surgery. The purpose was to report on speech outcome for all Swedish-speaking 5-year-olds born with a non-syndromic unilateral cleft lip and palate (UCLP), in 2008-2010, treated at Sweden's six cleft palate centres, and to compare speech outcomes between centres. Speech was assessed in 57 children with percent consonants correct adjusted for age (PCC-A), based on phonetic transcriptions from audio recordings by five independent judges. Also, hypernasality and perceived velopharyngeal function were assessed. The median PCC-A for all children was 93.9, and medians in the different groups varied from 89.9 to 96.8. In the total group, 9 children (16%) had more than mild hypernasality. Twenty-two children (38.5%) were perceived as having competent/sufficient velopharyngeal function, 25 (44%) as having marginally incompetent/insufficient velopharyngeal function, and 10 children (17.5%) as having incompetent/insufficient velopharyngeal function. Ten children were treated with secondary speech improving surgery and/or fistula surgery. No significant differences among the six groups, with eight to ten children in each group, were found. The results were similar to those in other studies on speech of children with UCLP, but poorer than results in normative data of Swedish-speaking 5-year-olds without UCLP. Indications of differences in frequency of surgical treatment and speech treatment between centres were observed.

Scandcleft randomised trials of primary surgery for unilateral cleft lip and palate: 4. Speech outcomes in 5-year-olds - velopharyngeal competency and hypernasality.

BACKGROUND AND AIM: Adequate velopharyngeal function and speech are main goals in the treatment of cleft palate. The objective was to investigate if there were differences in velopharyngeal competency (VPC) and hypernasality at age 5 years in children with unilateral cleft lip and palate (UCLP) operated on with different surgical methods for primary palatal repair. A secondary aim was to estimate burden of care in terms of received additional secondary surgeries and speech therapy. DESIGN: Three parallel group, randomised clinical trials were undertaken as an international multicentre study by 10 cleft teams in five countries: Denmark, Finland, Sweden, Norway, and the UK. METHODS: Three different surgical protocols for primary palatal repair were tested against a common procedure in the total cohort of 448 children born with a non-syndromic UCLP. Speech audio and video recordings of 391 children (136 girls, 255 boys) were available and perceptually analysed. The main outcome measures were VPC and hypernasality from blinded assessments. RESULTS: There were no statistically significant differences between the prevalences in the arms in any of the trials. VPC: Trial 1, A: 58%, B: 61%; Trial 2, A: 57%, C: 54%; Trial 3, A: 35%, D: 51%. No hypernasality: Trial 1, A: 54%, B: 44%; Trial 2, A: 47%, C: 51%; Trial 3, A: 34%, D: 49%. CONCLUSIONS: No differences were found regarding VPC and hypernasality at age 5 years after different methods for primary palatal repair. The burden of care in terms of secondary pharyngeal surgeries, number of fistulae, and speech therapy visits differed. TRIAL REGISTRATION: ISRCTN29932826.

Scandcleft randomised trials of primary surgery for unilateral cleft lip and Palate: 9. Parental report of social and emotional experiences related to their 5-year-old child's cleft diagnosis.

BACKGROUND AND AIM: Parents of children with a cleft lip and palate may be emotionally affected by the child's diagnosis. Their experiences and perceptions are important when evaluating the complexity of satisfactory treatment outcomes. The objective was to examine parents' social and emotional experiences related to their child's cleft diagnosis, and their perceptions of the child's adjustment to living with a visible difference. DESIGN: International multicentre study by 10 cleft teams in five countries: Denmark, Finland, Sweden, Norway, and the UK. METHODS: A cohort of 448 children born with a non-syndromic UCLP were included. A total of 356 parents completed the Scandcleft Parent Questionnaire. RESULTS: The majority of parents experienced practical and emotional support from family, friends, and health professionals. Nevertheless, parents had to cope with other people's reactions to the cleft, experiences that were described as ranging from hurtful to neutral and/or positive. According to parents, 39% of the children had experienced cleft-related comments and/or teasing. More than half of the parents reported specific worries related to their child's future. CONCLUSION: While the majority of the parents experienced positive support and coped well with the child's diagnosis, some parents were at risk for psychological and emotional challenges that should be identified by the cleft team. To optimise outcomes and the child's adjustment, these parents should be offered psychological support when necessary. TRIAL REGISTRATION: ISRCTN29932826.

Scandcleft randomised trials of primary surgery for unilateral cleft lip and palate: 5. Speech outcomes in 5-year-olds - consonant proficiency and errors.

BACKGROUND AND AIM: Normal articulation before school start is a main objective in cleft palate treatment. The aim was to investigate if differences exist in consonant proficiency at age 5 years between children with unilateral cleft lip and palate (UCLP) randomised to different surgical protocols for primary palatal repair. A secondary aim was to estimate burden of care in terms of received additional secondary surgeries and speech therapy. DESIGN: Three parallel group, randomised clinical trials were undertaken as an international multicentre study by 10 cleft teams in five countries: Denmark, Finland, Norway, Sweden, and the UK. METHODS: Three different surgical protocols for primary palatal repair were tested against a common procedure in the total cohort of 448 children born with non-syndromic UCLP. Speech audio- and video-recordings of 391 children (136 girls and 255 boys) were available and transcribed phonetically. The main outcome measure was Percent Consonants Correct (PCC) from blinded assessments. RESULTS: In Trial 1, arm A showed statistically significant higher PCC scores (82%) than arm B (78%) (p = .045). No significant differences were found between prevalences in Trial 2, A: 79%, C: 82%; or Trial 3, A: 80%, D: 85%. Across all trials, girls achieved better PCC scores, excluding s-errors, than boys (91.0% and 87.5%, respectively) (p = .01). CONCLUSIONS: PCC scores were higher in arm A than B in Trial 1, whereas no differences were found between arms in Trials 2 or 3. The burden of care in terms of secondary pharyngeal surgeries, number of fistulae, and speech therapy visits differed. TRIAL REGISTRATION: ISRCTN29932826.

Scandcleft randomised trials of primary surgery for unilateral cleft lip and palate: 10. Parental perceptions of appearance and treatment outcomes in their 5-year-old child.

BACKGROUND AND AIM: Few studies have explored children's emotional and behavioural reactions to cleft surgery and treatment-related stress. The objective was to investigate parents' evaluations of appearance and treatment outcomes in their 5-year-old child with unilateral cleft lip and palate (UCLP), and their perceptions of how their child was coping with treatment, comparing this information with recorded postsurgical complications. DESIGN: Three parallel group randomised clinical trials were undertaken as an international multicentre study by 10 cleft teams in five countries: Denmark, Finland, Sweden, Norway, and the UK. METHODS: Three different surgical procedures for primary palatal repair were tested against a common procedure in the total cohort of 448 children born with a non-syndromic UCLP. A total of 356 parents completed the Scandcleft Parent Questionnaire, and 346 parents completed the Cleft Evaluation Profile. RESULTS: The results indicated that the majority of parents were satisfied with cleft-related features of their child's appearance. Further, most children coped well with treatment according to their parents. Nevertheless, 17.5% of the children showed minor or short-term reactions after treatment experiences, and 2% had major or lasting difficulties. There were no significant relationships between parent perceptions of treatment-related problems and the occurrence of post-surgical medical complications. CONCLUSIONS: Most parents reported satisfaction with their child's appearance. However, treatment-related problems were described in some children, urging cleft centres to be aware of potential negative emotional and behavioural reactions to treatment in some young children, with a view to preventing the development of more severe treatment-related anxiety. TRIAL REGISTRATION: ISRCTN29932826.

Speech and satisfaction with outcome of treatment in young adults with unilateral or bilateral complete clefts.

Thirty-five young adults (22-32 years old, mean 25) born with complete unilateral or bilateral clefts participated in a follow-up of speech, appearance, and teeth after treatment. They completed a questionnaire about their satisfaction with their speech, nose, lip, and teeth on visual analogue scales, and indicated on two overall questions how often they thought about their cleft, and how often they were asked questions about their speech, nose, or lip. Their speech was recorded and assessed blindly and independently by two speech and language pathologists. Participants' satisfaction with their speech did not correlate significantly with the speech assessments. Satisfaction with the nose had the highest correlation with the overall questions. No participant indicated more dissatisfaction with speech than the midpoint of the scale, making conclusions about covariance between satisfaction with speech and the overall questions difficult.

Evaluation of VPI-assessment with videofluoroscopy and nasoendoscopy.

UNLABELLED: The purpose of this study was to investigate how different amounts of visual assessment information influence the recommended treatment for velopharyngeal insufficiency (VPI). Is a patient presented with videofluoroscopy (VF) in lateral projection recommended the same treatment as when frontal projection, nasoendoscopy, or both, are added? Retrospective material with video recorded assessment of VPI was blinded and copied in random order. Each patient was then presented in four separate combinations: VF in lateral projection; VF in lateral and frontal projection; VF in lateral projection and nasoendoscopy; and VF in lateral and frontal projection and nasoendoscopy (all of the available assessment material). The cleft palate team of Göteborg, Sweden, mutually rated velopharyngeal function and recommended action based on the presented material. SUBJECTS: Nineteen consecutive patients (median age 7:5 years, range 4:4-19:7) investigated with VF in lateral and frontal projection and nasoendoscopy during 1997-99 at the cleft palate centre in Göteborg, Sweden. Post operative assessments were excluded. Percent agreement and Kappa calculations were used to compare the different combinations of parts of information to all of the available information. RESULTS: Thirteen of the 19 patients (68%) were recommended the same action regardless of the amount of presented information. Percent agreement (Kappa) between parts and all of the available information: VF in lateral projection 84% (0.75), VF in lateral and frontal projection 79% (0.74), and VF in lateral projection and nasoendoscopy 84% (0.72). CONCLUSIONS: VF in lateral projection is recommended to be the first step in visualising velopharyngeal function, and nasoendoscopy the next when further investigation is required.