Long-term Outcome for Two-Stage Palatal Closure With Different Timings for Hard Palate Surgery: Craniofacial Growth and Dental Arch Relation.

OBJECTIVE: The aim was to evaluate dental arch relation and craniofacial growth for individuals born with unilateral cleft lip and palate (UCLP), who had two-stage palatal surgery, with hard palate closure (HPC) at the ages of 3 or 8 years. DESIGN: Longitudinal cohort study. SETTING: Ceft lip and palate team in Gothenburg, Sweden. PATIENTS: The inclusion criteria were nonsyndromic individuals born with complete UCLP who were consecutively operated according to two different surgical protocols; soft palate closure at 6 months followed by hard palate closure at the age of 3 years (HPC3) or at the age of 8 years (HPC8). In this evaluation, 28 individuals had HPC3 and 59 individuals had HPC8. INTERNVENTIONS: The main outcome, longitudinal series of dental casts and lateral radiographs from the ages of 5, 10, 16, and 19 years, were evaluated using GOSLON Yardstick and cephalometric analysis. RESULTS: At the age of 10 years, 78% of the individuals with HPC3 demonstrated GOSLON scores of 1 and 2 and 86% in HPC8. At the age of 19 years, 54% of the individuals exhibited GOSLON scores of 1or 2 when compared with 74% in HPC8. A statistical significant difference for SNA was observed at the age of 5 years (P = .004), with a lower SNA in HPC3, but not at the ages of 10, 16 and 19 years. At the final age, SNA was 75.2° for HPC3 and 76.8° for HPC8. CONCLUSIONS: The decrease in age for HPC did not have an adverse effect on long-term dental arch relationship or craniofacial growth.

Reliability and Predictive Validity of Dental Arch Relationships Using the 5-Year-Olds' Index and the GOSLON Yardstick to Determine Facial Growth.

AIMS: To determine reliability and predictive validity of the 5-year-olds'(5YO) Index and GOSLON Yardstick in 119 patients born with unilateral cleft lip and palate at 5, 7/8, 10, 15/16, and 19 years. METHODS: Five hundred thirty-four dental study models were appraised by 2 teams in 2 centers, twice in each center. Intrateam and interteam reliability in scoring the models was calculated using κ. Dental arch prediction rates were calculated as the proportion of models remaining in the same category (good-scores 1 and 2; fair-score 3; poor-scores 4 and 5) over time. RESULTS: Intrateam and interteam κ statistics ranged from 0.74 to 0.89 and from 0.74 to 0.81, respectively. The 5YO Index and GOSLON Yardstick at 5 years produced almost identical results. The prediction rate of 19-year-old (n = 106) outcome was >80% for those in groups 1 and 2 at 5 years, while for those in groups 4 and 5 prediction was poor (<40%). Prediction of groups 4 and 5 remained poor until 10 years when it increased to 77%. At 15/16 years prediction rate was 93% for those in groups 4 and 5. Prediction of cases in group 3 was very poor at all ages. CONCLUSIONS: These results question the predictive value of "poor" dental arch relationships before 10 years of age. However, the predictive value of "good" dental arch relationship scores over time is good in all age groups. This has implications for audit policies to predict facial growth outcomes.

Vertical maxillary growth after two different surgical protocols in unilateral cleft lip and palate patients.

Objective : The aim of the present study was to compare vertical maxillofacial growth in patients born with unilateral cleft lip and palate (UCLP) who were treated using two different surgical protocols. Design : A retrospective cohort study. Subjects : We studied 92 patients with complete UCLP (61 male and 31 female) treated at Sahlgrenska University Hospital in Gothenburg, Sweden: 46 consecutive patients born between 1965 and 1974 who underwent surgical treatment according to the Wardill-Kilner (W-K) protocol and 46 consecutive patients born between 1982 and 1989 who underwent surgical treatment according to the Gothenburg delayed hard palate closure (DHPC) protocol. Methods : We analyzed lateral cephalograms obtained at 10 years of age. Results : Patients treated according to the Gothenburg DHPC protocol had significantly greater anterior upper facial height, anterior maxillary height, overbite, and inclination of the maxilla than those treated with the W-K protocol. Both techniques led to similar posterior upper facial height. Conclusion : The Gothenburg DHPC protocol in patients with complete UCLP results in more normal anterior maxillary vertical growth and overbite and therefore increased maxillary inclination at 10 years of age.

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.

Long-term, longitudinal follow-up of individuals with unilateral cleft lip and palate after the Gothenburg primary early veloplasty and delayed hard palate closure protocol: speech outcome.

OBJECTIVE: To investigate long-term, longitudinal speech outcome in patients born with unilateral cleft lip and palate treated according to a two-stage primary palatal protocol with early veloplasty and delayed hard palate closure. DESIGN: Retrospective, longitudinal cohort study. Setting : A university hospital in western Sweden. SUBJECTS: A consecutive series of 55 patients from the total cohort of 65 were included. All patients had surgical procedures at Sahlgrenska University Hospital, Gothenburg, Sweden. METHODS: Standardized audio recordings were blindly analyzed at 5, 7, 16, and 19 years of age and after at a clinical visit at 10 years of age. Typical cleft speech variables were rated independently on ordinal scales. Intelligibility and perceived velopharyngeal function were assessed also. Prevalences of speech characteristics were determined, and interrater and intrarater agreement were calculated. Results : Prominent hypernasality, nasal air leakage, and retracted oral articulation at 5 years were markedly reduced throughout the years with low prevalences at ages 16 and 19 years. Perceived velopharyngeal competence was noted in 82% at age 16 and 87% at age 19 years along with normal intelligibility. Pharyngeal flap surgery was performed in 6 of the 55 patients (11%). CONCLUSIONS: Long-term speech outcome in patients with two-stage palatoplasty with early soft palate repair was considered good and improved even before hard palate repair. The typical retracted oral articulation was quite frequent during the early ages; whereas, nonoral misarticulations were almost nonexistent, implying good velopharyngeal competence.

Long-term, longitudinal follow-up of individuals with UCLP after the Gothenburg primary early veloplasty and delayed hard palate closure protocol: maxillofacial growth outcome.

OBJECTIVE: To study long-term maxillofacial development in a sample of patients born with unilateral cleft lip and palate. The patients' palatal repair had been performed according to a two-stage protocol with early velar closure and delayed hard palate surgery. DESIGN: Retrospective, longitudinal cohort study. SUBJECTS: The sample consisted of 50 consecutive patients with unilateral cleft lip and palate born from 1980 to 1989. All of them had been operated on at the Sahlgrenska University Hospital in Gothenburg, Sweden. METHODS: Certain maxillary casts as well as lateral roentgencephalograms, obtained at 5, 10, 16, and 19 years of age, were analyzed. Results : Our patients' maxillofacial growth was very good even up to the final examination in early adulthood. We ascribe the advantageous midfacial morphology of our patients particularly to limited growth restriction from palatal scars due to the surgical protocol used in our cleft center. CONCLUSION: The two-stage protocol we advocate for repair of the palatal cleft resulted in very satisfactory growth outcome. A palatal scar, which might impair maxillary development, was created only by the velar surgery. If this scar is located close to the posterior border of the hard palate, it might result in less maxillary growth restriction than if it is positioned further anteriorly.

Isolated cleft palate requires different surgical protocols depending on cleft type.

A staged protocol for isolated cleft palate (CPO), comprising the early repair of the soft palate at 6 months and delayed repair of the eventual cleft in the hard palate until 4 years, designed to improve maxillary growth, was introduced. CPO is frequently associated with additional congenital conditions. The study evaluates this surgical protocol for clefts in the soft palate (CPS) and for clefts in the hard and soft palate (CPH), with or without additional malformation, regarding primary and secondary surgical interventions needed for cleft closure and for correction of velopharyngeal insufficiency until 10 years of age. Of 94 consecutive children with CPO, divided into four groups with (+) or without (-) additional malformations (CPS + or CPS - and CPH + or CPH-), hard palate repair was required in 53%, performed with small local flaps in 21% and with bilateral mucoperiosteal flaps in 32%. The total incidence of soft palate re-repair was 2% and the fistula repair of the hard palate was 5%. The total incidence of secondary velopharyngeal surgery was 17% until 10 years, varying from 0% for CPS - and 15% for CPH-, to 28% for CPS + and 30% for CPH+. The described staged protocol for repair of CPO is found to be safe in terms of perioperative surgical results, with comparatively low need for secondary interventions. Furthermore, the study indicates that the presence of a cleft in the hard palate and/or additional conditions have a negative impact on the development of the velopharyngeal function.

Scandcleft randomised trials of primary surgery for unilateral cleft lip and palate: 2. Surgical results.

BACKGROUND: Longstanding uncertainty surrounds the selection of surgical protocols for unilateral cleft lip and palate, and randomised trials have only rarely been performed. The Scandcleft Project consists of three trials commenced in 1997 involving ten centres in Denmark, Finland, Norway, Sweden, and the UK. Three groups of centres tested a newly-defined common technique for palatal repair (Arm A) against their local protocols (Arms B, C, D). Arm A was familiar to most of the surgeons in Trial 1, but not to the surgeons in the other Trials. AIM: To evaluate surgical events and complications of the 448 (293 boys, 155 girls) patients with complete unilateral cleft lip and palate (UCLP) enrolled in the three trials. METHOD: The three trials were carried out in parallel in adherence with a fully developed, ethically approved protocol. Operative time, bleeding, complications, and major dehiscence during and after both primary surgeries were recorded by the surgeon. Rates of fistula and surgery for velopharyngeal incompetence (VPI) were assessed until the youngest patient of the study had reached the age of 9 years. Pearson Chi-square statistical analysis was used to compare the outcomes. RESULTS: No significant differences in bleeding, infection, anaesthetic complications or length of hospital stay between the different arms were found for Trial 1. However, in Trials 2 and 3 there were more airway problems in Arm A than with the traditional local protocols (Arms C or D). In Trial 3 fistula and VPI surgery rates were also higher in Arm A. CONCLUSIONS: The results do not provide statistical evidence that any technique is better than others, but indicate that surgery was more problematic for surgeons who were still gaining experience with an unfamiliar surgical protocol. TRIAL REGISTRATION: ISRCTN29932826.

A Scandcleft randomised trials of primary surgery for unilateral cleft lip and palate: 1. Planning and management.

BACKGROUND AND AIMS: Longstanding uncertainty surrounds the selection of surgical protocols for the closure of unilateral cleft lip and palate, and randomised trials have only rarely been performed. This paper is an introduction to three randomised trials of primary surgery for children born with complete unilateral cleft lip and palate (UCLP). It presents the protocol developed for the trials in CONSORT format, and describes the management structure that was developed to achieve the long-term engagement and commitment required to complete the project. METHOD: Ten established national or regional cleft centres participated. Lip and soft palate closure at 3-4 months, and hard palate closure at 12 months served as a common method in each trial. Trial 1 compared this with hard palate closure at 36 months. Trial 2 compared it with lip closure at 3-4 months and hard and soft palate closure at 12 months. Trial 3 compared it with lip and hard palate closure at 3-4 months and soft palate closure at 12 months. The primary outcomes were speech and dentofacial development, with a series of perioperative and longer-term secondary outcomes. RESULTS: Recruitment of 448 infants took place over a 9-year period, with 99.8% subsequent retention at 5 years. CONCLUSION: The series of reports that follow this introductory paper include comparisons at age 5 of surgical outcomes, speech outcomes, measures of dentofacial development and appearance, and parental satisfaction. The outcomes recorded and the numbers analysed for each outcome and time point are described in the series. TRIAL REGISTRATION: ISRCTN29932826.

Maxillary dental arch and occlusion in patients with unilateral cleft lip and palate treated with different delays in closure of the hard palate after early velar repair.

We wanted to find out if growth of the maxilla in 26 patients with unilateral cleft lip and palate (UCLP) was adversely affected by having the residual cleft of the hard palate repaired earlier than had been done previously in a 2-stage palatal closure protocol. The ages at repair of the hard palate of the present patients ranged from 38 to 89 months. Dental casts from ages about 3 years (before any repair of the hard palate), 5, 7, and 10 years of age were analysed. The results indicated that earlier repair of the cleft in the hard palate did not influence maxillary growth differently from the later repair.

Speech development in patients with unilateral cleft lip and palate treated with different delays in closure of the hard palate after early velar repair: a longitudinal perspective.

We wanted to find out if different timing of delayed repair of the hard palate in a two-stage procedure had an impact on the speech of 26 patients with unilateral cleft lip and palate (UCLP). The soft palate was closed at the age of 7 months and the hard palate between 38 and 89 months of age. Speech audio recordings at the age of 3 years (baseline, before any repair of the hard palate) and at the ages of 5, 7, and 10 years (the latter obtained at least one year after closure) were analysed. We used standardised speech assessments at routine follow-up and assessment by one external listener. The prevalence of speech errors caused by the cleft was similar to those described in previous reports from our centre in which hard palate repair was delayed. Unexpectedly, the results showed no difference in speech production related to timing of hard palate repair, except for nasal air leakage at the age of 7 years.

Height of the palatal vault after two different surgical procedures: study of the difference in patients with complete unilateral cleft lip and palate.

The present study compared the height of the palatal vault in dental casts from 320 10-year-old children with unilateral cleft lip and palate (UCLP) operated on with the push-back technique according to Wardill-Kilner (W-K) with patients operated on with delayed hard palate closure (DHPC). The palatal height in patients operated on with the DHPC technique was found to be significantly higher than in patients operated on with the W-K technique. This coincides with better maxillary growth and better speech in the DHPC group.

Alveolar bone grafting.

In patients with cleft lip and palate, bone grafting in the mixed dentition in the residual alveolar cleft has become a well-established procedure. The main advantages can be summarised as follows: stabilisation of the maxillary arch; facilitation of eruption of the canine and sometimes facilitation of the lateral incisor eruption; providing bony support to the teeth adjacent to the cleft; raising the alar base of the nose; facilitation of closure of an oro-nasal fistula; making it possible to insert a titanium fixture in the grafted site and to obtain favourable periodontal conditions of the teeth within and adjacent to the cleft. The timing of the ABG surgery take into consideration not only eruption of the canine but also that of the lateral incisor, if present. The best time for bone grafting surgery is when a thin shell of bone still covers the soon erupting lateral incisor or canine tooth close to the cleft.

Analysis of dental arch relationships in Swedish unilateral cleft lip and palate subjects: 20-year longitudinal consecutive series treated with delayed hard palate closure.

OBJECTIVE: To evaluate the dental arch relationships for a consecutive series from Goteborg, Sweden, who had delayed hard palate closure. DESIGN: Retrospective study. SETTING: Sahlgrenska University Hospital, Goteborg, Sweden. PATIENTS: The dental study models of 104 consecutive unilateral cleft lip and palate subjects. The study cohort was born between 1979 and 1994. Longitudinal records were available at ages 5 (n = 94), 10 (n = 97), 16 (n = 59), and 19 years (n = 46). Five assessors rated models according to the GOSLON Yardstick on two separate occasions each. INTERVENTIONS: These patients had been operated upon according to the Goteborg protocol of delayed hard palate closure (at age 8 years). RESULTS: 85% of subjects were rated in groups 1 and 2 (excellent or very good outcome), 12% were rated in group 3 (satisfactory), and 3% were assigned to group 4 (poor). No patients presented in Group 5 (very poor). Weighted kappa statistics for double determination of Yardstick allocation for five assessors demonstrated values between .65 and .90 for interrater agreement (good/very good) and between .70 and .90 for intrarater agreement (very good). CONCLUSIONS: Delayed hard palate closure as practiced in Goteborg since 1979 has produced the best GOSLON Yardstick ratings in a consecutive series of patients ever recorded worldwide, since the Yardstick was first used in 1983. However, it is noteworthy that a new protocol has been introduced in Goteborg since 1994, in which hard palate closure is done at 3 years due to concerns regarding speech.

Peripheral postganglionic sympathicoplegia mimicking cluster headache attacks.

After dissection with complete occlusion of the internal carotid artery, a 58-year-old man started to suffer from intense cluster headache-like attacks. Magnetic imaging showed signs of nonsymptomatic cerebral emboli, which could be dated to have occurred in temporal relation to the start of the attacks, all on the right side. This case and two similar ones indicate that peripheral postganglionic sympathicoplegia can cause attacks with similar pain characteristics, accompanying symptoms, duration, and regularity as in cluster headache.

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.