The Cleft Palate Craniofacial Journal

Acoustic studies of oral-nasal balance disorders to date have focused on hypernasality. However, in patients with cleft palate, nasal obstruction may also be present, so that hypernasality and hyponasality co-occur. In this study, normal speakers simulated different disorders of oral-nasal balance. Linear discriminant analysis was used to create a tentative diagnostic formula based on the long-term averaged spectra (LTAS) of the speech stimuli.

Eleven female participants were recorded while reading nonnasal and nasal speech stimuli. LTASs of the recordings were run for their normal oral-nasal balance and their simulations of hyponasal, hypernasal, and mixed oral-nasal balance. The amplitude values (in decibels) were extracted in 100-Hz intervals over a range of 4 kHz.

A repeated-measures analysis of variance of the normalized amplitudes revealed a resonance condition–frequency band amplitude interaction effect (P < .001). A linear discriminant analysis of the participants’ LTAS led to formulas correctly classifying 80.7% of the oral-nasal balance conditions.

The simulations produced distinctive spectra enabling the creation of formulas that predicted the oral-nasal balance above chance level. Future research with speakers with oral-nasal balance disorders will be needed to investigate the potential of this approach for the clinical diagnosis of disorders of oral-nasal balance.

The aim was to compare the oral health–related quality of life among 11- to 14-year-old patients with cleft lip and/or palate (CLP) and schoolchildren without CLP. The validity and reliability of the Finnish Child Perception Questionnaire designed for 11- to 14-year-olds (CPQ_11-14) was also assessed.

Participants in this cross-sectional questionnaire survey study were children aged 11 to 14 years from two groups. The CLP sample included all children of this age who had had CLP selected from the regional treatment register (N = 51). The school sample included children from four school classes (N = 82). Informed consent from parents was obtained. Ethical clearance and parental informed consent were obtained.

Oral health–related quality of life was measured with the CPQ11-14.

The CPQ_11-14 total and oral symptoms, functional limitations, emotional well-being, and social well-being subscores were poorer among patients with CLP than among schoolchildren without CLP (mean scores: 55.5 versus 15.0; 11.9 versus 5.1; 14.0 versus 2.8; 12.6 versus 4.2; and 17.1 versus 2.9, respectively; all P < .001 for Mann-Whitney tests). Cronbach alpha value was 0.97 for total scale and between 0.81 and 0.94 for subscales. Among all children in the school sample, intraclass correlation coefficient was 0.79 for total scale and varied between 0.65 and 0.74 for subscales.

The oral health–related quality of life of Finnish children with CLP was considerably poorer than that of their peers in overall and all dimensions, especially social well-being. The CPQ11-14 showed appropriate reliability and validity.

A significant proportion of patients with cleft palate experience persisting velopharyngeal dysfunction (VPD) after primary surgery. Pharyngoplasty is the most common procedure to correct inadequate velopharyngeal closure. Although it is often effective, it poses a risk for postoperative airway obstruction. The mucomuscular buccinator flap is a more recent alternative: In the largest case series to date, we outline its use and evaluate its effectiveness in managing persistent VPD.

Over 9 years, 103 buccinator flap procedures were performed by the Cambridge group to improve velar function. Clinical records were retrospectively assessed: 60 patients were analyzed by two expert speech and language therapists external to the group using the Cleft Audit Protocol for Speech—Augmented. In a subset of patients, the buccinator flap was interpolated between the limbs of a large mucosal Z-plasty. Consensus listening was undertaken, and interrater reliability was calculated for 24.17% of the cohort. The remaining samples were assessed by a single listener following calibrated consensus listening.

There was a significant reduction in VPD (P < .001). Preoperatively, 68.5% of patients demonstrated marked VPD, falling to 24.1% postoperatively. The buccinator procedure demonstrated significant improvement for three of four individual speech parameters: hypernasality, passive cleft speech characteristics, and audible nasal emission. Overall, 13.5% of patients required further corrective speech surgery.

The buccinator mucomuscular flap reliably and effectively improves velar function in the management of VPD and has low complication rates. We therefore recommend the use of the buccinator flap in primary surgical management of persisting VPD.

Retrospective cohort study.

Major international tertiary care referral center for cleft palate repair.

One hundred thirty-eight patients at the Children's Hospital of Philadelphia who had palate repair performed between 2010 and 2013, excluding syndromic patients, patients undergoing palate revision, and patients with incomplete payment information.

None.

Fees and charges for procedures.

Surgeon payment was significantly higher for international adoptees (Δ = $2047.51 [$128.35 to $3966.66], P = .038). Medicaid-adjusted surgeon payments averaged $1006 more for adoptees ([-$394.19 to $2406.98], P = .158).

Hospital and anesthesiology costs for adoptee palate repair were highly variable but did not differ significantly from those for nonadoptees. Partly due to payer mix, surgeon reimbursement was somewhat higher for international adoptees. No difference in total payment was found.

To compare length of follow-up and cleft site dental management on bone graft ratings from two centers.

Blind retrospective analysis of cleft site radiographs and chart reviews for determination of cleft-site lateral incisor management.

A total of 78 consecutively grafted patients with complete clefts from two major cleft/craniofacial centers (43 from Center 1 and 35 from Center 2).

Secondary iliac crest alveolar bone grafting, at a mean age of 9 years 9 months (Center 1: 9 years 7 months; Center 2: 10 years 0 month).

The Americleft Standardized Way to Assess Grafts scale from 0 (failed graft) to 6 (ideal) was used to rate graft outcome at two time points (T1, T2). Average T1 was 11 years 1 month of age, 1 year 3 months postgraft. Average T2 was 17 years 11 months of age, 8 years 0 months postgraft. Six trained and calibrated raters scored each radiograph twice. Reliability was calculated at T1 and T2 using weighted kappa. A paired Wilcoxon signed rank test (P < .05) tested T1 and T2 differences for each center. A Kruskal-Wallis test was used to determine the significance of differences between centers at T1 and T2. Correlation tested whether T1 ratings predicted T2. Linear regression determined possible factors that might contribute to graft rating changes over time.

Reliability was good at T1 and T2 (interrater = .713 and .701, respectively; intrarater = .790 and .805, respectively). Center 1 scores were significantly better than those from Center 2 at both T1 (5.21 versus 3.29) and T2 (5.18 versus 3.44). There was no statistical difference between T1 and T2 scores for either center; although, there was a greater chance of bone graft score improving with completion of canine eruption and substitution for missing lateral incisors.

Short-term ratings of graft outcomes identified significant differences between centers that persisted over time. Dental cleft-site management influenced final graft outcome.

The aim of this study was to explore how 10-year-olds describe speech and communicative participation in children born with unilateral cleft lip and palate in their own words, whether they perceive signs of velopharyngeal insufficiency (VPI) and articulation errors of different degrees, and if so, which terminology they use.

Nineteen 10-year-olds participated in three focus group interviews where they listened to 10 to 12 speech samples with different types of cleft speech characteristics assessed by speech and language pathologists (SLPs) and described what they heard. The interviews were transcribed and analyzed with qualitative content analysis.

The analysis resulted in three interlinked categories encompassing different aspects of speech, personality, and social implications: descriptions of speech, thoughts on causes and consequences, and emotional reactions and associations. Each category contains four subcategories exemplified with quotes from the children's statements. More pronounced signs of VPI were perceived but referred to in terms relevant to 10-year-olds. Articulatory difficulties, even minor ones, were noted. Peers reflected on the risk to teasing and bullying and on how children with impaired speech might experience their situation. The SLPs and peers did not agree on minor signs of VPI, but they were unanimous in their analysis of clinically normal and more severely impaired speech.

Articulatory impairments may be more important to treat than minor signs of VPI based on what peers say.

To gain understanding of perspectives on peer relationships from children with congenital craniofacial anomalies (CFA).

This was qualitative research based in a phenomenological approach, using narratives that captured children's responses to open-ended and objective questions about peer relations and life with a CFA. Interviews were audio recorded and transcribed. Transcripts were coded according to thematic categories.

Children were patients at a reconstructive plastic surgery center in an urban hospital and medical school and were recruited from a regional support organization for families of children with CFA that was associated with the hospital.

Nine children with congenital CFA aged 9 to 14 years.

Main Outcome Measures: Thematic coding categories were developed from the narratives using an open coding strategy; these categories focused on aspects of children's interactions with peers and their appraisals of the role of their CFA in their lives.

Children reported satisfaction with most aspects of their peer relationships and expressed confidence in their ability to manage challenges. They acknowledged some difficulties with living with a CFA but tended to hold a balanced perspective on the impact of a CFA on their lives, and they expressed optimism about their future lives.

This sample of children with CFA exhibited much resilience. Although they may not be representative of all children with CFA, they provide examples that can be used to generate hypotheses for future research.

To determine the association of single-nucleotide polymorphisms (SNPs) in genes related to craniofacial development, which were previously identified as susceptibility signals for nonsyndromic oral clefts, in Brazilians with nonsyndromic cleft lip and/or palate (NSCL/P).

The SNPs rs748044 (TNP1), rs1106514 (MSX1), rs28372960, rs15251 and rs2569062 (TCOF1), rs7829058 (FGFR1), rs1793949 (COL2A1), rs11653738 (WNT3), and rs242082 (TIMP3) were assessed in a family-based transmission disequilibrium test (TDT) and a structured case-control analysis based on the individual ancestry proportions.

The SNPs were initially analyzed by TDT, and polymorphisms showing a trend toward excess transmission were subsequently studied in an independent case-control sample.

The study sample consisted of 189 case-parent trios of nonsyndromic cleft lip with or without cleft palate (NSCL±P), 107 case-parent trios of nonsyndromic cleft palate (NSCP), 318 isolated samples of NSCL±P, 189 isolated samples of NSCP, and 599 healthy controls.

Association of alleles with NSCL/P pathogenesis.

Preferential transmission of SNPs rs28372960 and rs7829058 in NSCL±P trios and rs11653738 in NSCP trios (P = .04) were observed, although the structured case-control analysis did not confirm these associations. The haplotype T-C-C formed by TCOF1 SNPs rs28372960, rs15251, and rs2569062 was more frequently transmitted from healthy parents to NSCL±P offspring, but the P value (P = .01) did not withstand Bonferroni correction for multiple tests.

With the modest associations, our results do not support the hypothesis that TNP1, MSX1, TCOF1, FGFR1, COL2A1, WNT3, and TIMP3 variants are risk factors for nonsyndromic oral clefts in the Brazilian population.

With the recent advances of surgical adjuncts including presurgical naso-alveolar molding and postoperative nasal stenting, information on the anthropometric evaluation of cleft lip nasal symmetry after primary rhinoplasty is lacking.

Twenty-nine nonsyndromic patients with complete unilateral cleft lip and palate undergoing modified rotation advancement cheiloplasty with synchronous primary rhinoplasty in our center were prospectively recruited in our study. All of them received our center's perioperative management protocol, including presurgical naso-alveolar molding and postoperative nasal stenting. Direct anthropometric measurements of their nasal configuration were documented when they were anesthetized for primary lip repair at 3 months of age and for the primary palate repair at 12 months of age. Their nasal configurations were analyzed before primary rhinoplasty and at 9 months after rhinoplasty.

There is a statistically significant change in nasal symmetry at 9 months after the primary rhinoplasty. There is no significant correlation between the nasal configuration before and at 9 months after the primary rhinoplasty.

With significant relapse of nasal deformity at 9 months after the primary rhinoplasty despite the use of presurgical naso-alveolar molding and postoperative nasal stenting, overcorrection of the nasal configuration at primary rhinoplasty should be considered for optimal long-term nasal symmetrical outcome.

Posterior pharyngeal flap (PPF) surgery is effective for treating velopharyngeal insufficiency but has historically been associated with risk of airway compromise. This study aims to identify risk factors for complications from and readmission after PPF using a national database.

Patients who underwent PPF surgery were selected from the 2012 American College of Surgeons National Surgical Quality Improvement Program Pediatric (NSQIP-PEDS) database. Patient characteristics, comorbidities, and complication/readmission data were analyzed.

Among 225 study patients, 12 (5.3%) suffered perioperative complications. The most common complications were pulmonary in nature (5 patients, 2.2%), including prolonged postoperative mechanical ventilation (3 patients, 1.3%). Underlying asthma (P = .024) or any cardiac risk factor (P = .047) conveyed significant complication risk. Further, severe cardiac risk factors were associated with postoperative bleeding (P = .024). Readmission (4 patients, 1.7%) and reoperation (3 patients, 1.3%) occurred at mean intervals of 9 and 10 days after the original procedure. Seventy-nine patients (35%) were discharged postoperatively on an outpatient basis, and this subgroup included only one patient (1.2%) with a complication (P = .038).

The overall perioperative complication rate for PPF surgery is low at 5.3%. Patients with underlying cardiac risk factors, severe American Society of Anesthesiologists Physical Status class, and asthma should prompt greater attention given their heightened risk profiles.

This study was undertaken to evaluate intraoral 3D scans for assessing dental arch relationships and obtain patient/parent perceptions of impressions and intraoral 3D scanning.

Forty-three subjects with nonsyndromic unilateral cleft lip and palate (UCLP) had impressions taken for plaster models. These and the teeth were scanned using the R700 Orthodontic Study Model Scanner and Trios® Digital Impressions Scanner (3Shape A/S, Copenhagen, Denmark) to create indirect and direct digital models. All model formats were scored by three observers on two occasions using the GOSLON and modified Huddart Bodenham (MHB) indices. Participants and parents scored their perceptions of impressions and scanning from 1 (very good) to 5 (very bad). Intra- and interexaminer reliability were tested using GOSLON and MHB data (Cronbach's Alpha >0.9). Bland and Altman plots were created for MHB data, with each model medium (one-sample t tests, P < .05) and questionnaire data (Wilcoxon signed ranks P < .05) tested.

Intra- and interexaminer reliability (>0.9) were good for all formats with the direct digital models having the lowest interexaminer differences. Participants had higher ratings for scanning comfort (84.8%) than impressions (44.2%) (P < .05) and for scanning time (56.6%) than impressions (51.2%) (P > .05). None disliked scanning, but 16.3% disliked impressions. Data for parents and children positively correlated (P < .05).

Reliability of scoring dental arch relationships using intraoral 3D scans was superior to indirect digital and to plaster models; Subjects with UCLP preferred intra-oral 3D scanning to dental impressions, mirrored by parents/carers; This study supports the replacement of conventional impressions with intra-oral 3D scans in longitudinal evaluations of the outcomes of cleft care.

To determine the repeatability and reproducibility of using three different viewing media to assess the outcomes of the dental arch relationships of patients with unilateral cleft lip and palate (UCLP) using the GOSLON Yardstick.

The GOSLON Yardstick was used to rate the dental arch relationships of 29 patients with UCLP. Three experienced calibrated orthodontists rated the plaster study models, digital study models, and stereoscopic projected three-dimensional (3D) study models separately. There was a minimum of a 1-week interval between each rating session. All three rating sessions were repeated 1 month later. A linear weighted kappa statistic was performed to assess intrarater repeatability and inter-rater reproducibility, as well as the comparison between different viewing media using Kendall's Coefficient of Concordance (Kendall's W) statistic.

Intra-rater repeatability was very good for all three viewing media (kappa = 0.83-0.92). Inter-rater reproducibility was good to very good across the three viewing media (kappa = 0.63-0.88). Agreements between plaster study models and digital study models or stereoscopic projected 3D study models were good to very good (kappa = 0.78-0.97 and kappa = 0.72-0.97, respectively), and a Kendall's W ranging from 0.86 to 0.92 (P < .001).

Stereoscopic projected 3D is an alternative method to assess the outcomes of dental arch relationships in patients with cleft lip and palate using the GOSLON Yardstick. It could also be used for viewing patient records, as it recovers the full 3D information captured at the time of the clinical examination.

Tongue-lip adhesion (TLA) is commonly used to relieve obstructive sleep apnea (OSA) in infants with Robin sequence (RS), but few studies have evaluated its efficacy with objective measures. The purpose of this study was to measure TLA outcomes using polysomnography. Our hypothesis was that TLA relieves OSA in most infants.

This is a retrospective study of infants with RS who underwent TLA from 2011 to 2014 and had at least a postoperative polysomnogram. Predictor variables included demographic and birth characteristics, surgeon, syndromic diagnosis, GILLS score, preoperative OSA severity, and clinical course. A successful outcome was defined as minimal OSA (apneahypopnea index score < 5) on postoperative polysomnogram and no need for additional airway intervention. Descriptive, bivariate, and regression statistics were computed, and statistical significance was set at P < .05.

Eighteen subjects who had TLA at a mean age of 28 ± 4.7 days were included. Thirteen (72.2%) had a confirmed or suspected syndrome, and the mean GILLS score was 3 ± 0.3. All parameters trended toward improvement from the preoperative to postoperative polysomnograms, and improvement in OSA severity, oxygen saturation nadir, and arousals per hour was statistically significant (P < .02). This effect was significant across categories of surgeon, syndrome, and GILLS score. Nine subjects (50%) met the criteria for a successful outcome. Bivariate and regression analyses did not demonstrate a significant relationship between success and any predictor variable.

TLA improved airway obstruction in all infants with RS but resolved OSA in only nine patients, and success was unpredictable.

The purpose of this study was to present our experience with the closure of challenging palatal fistulae using the deepithelialized dorsal anterior tongue flap. We highlight the efficacy of suturing the tip of the tongue flap to the nasal septum for prevention of flap detachment.

Prospective analysis of cleft patients with anterior palatal fistulae repaired by deepithelialized dorsal anterior tongue flap.

Institutional center.

A total of 30 patients with anterior palatal fistulae in the setting of previous cleft palate or fistula repair.

Deepithelialized dorsal anterior tongue flap for treatment of anterior palatal fistulae.

Patients had repair using deepithelialized dorsal anterior tongue flap between 2011 and 2014. Patients’ photographs and clinical records were collected. The technique of flap harvesting and method of securing it in its position are described in this study.

Patients were followed up over a mean period of 13.8 months to check flap viability, competent repair, and donor site function and aesthetics. All patients showed uneventful healing without complications.

This technique offered a secure method of palatal fistulae reconstruction. Further research is needed to show this technique's advantages and drawbacks.

Recently, platelet-rich fibrin (PRF) was described as a second-generation platelet concentrate. PRF is known as a rich source of autologous cytokines and growth factors and is universally used for tissue regeneration in clinical medicine.

The aim of the current study was to evaluate the effect of PRF on the quality and quantity of bone formation in unilateral maxillary alveolar cleft reconstruction.

Twenty-four patients with unilateral alveolar cleft underwent bone reconstruction. Patients were randomly divided into two groups. Group A consisted of patients grafted with PRF combined with autogenous anterior iliac crest bone graft. Group B patients were grafted using autogenous bone graft alone (control group). Computed tomography was performed 6 months postoperatively to assess the quality and quantity of the newly formed bone.

The percentage of newly formed bone (quantity) in group A ranged from 79.74% to 88.4%, with a mean percentage of 82.6% ± 3.9%. In group B, the percentage of bone formation ranged from 60.3% to 76.4%, with a mean percentage of 68.38% ± 6.67%. There was a statistically significant increase in the percentage of newly formed bone in group A. The mean bone density (quality) of the newly formed bone was lower in group A than group B, but the difference was not statistically significant (P < .05).

PRF in combination with autogenous bone was beneficial in improving the volume of newly formed bone in alveolar cleft reconstruction and does not enhance bone density.

To describe the temporal pattern of otitis media with effusion (OME) resolution for a cohort of nonsyndromic cleft palate children enrolled before palatoplasty and followed through 5 years of age.

This is a prospective, longitudinal study of the time course for OME resolution in infants and children with palatal clefts.

Cleft Palate Craniofacial Center of a tertiary care pediatric hospital.

This study included 52 children with cleft palate (29 boys, 45 white, Veau 1 through 4) who had a Furlow-type palatoplasty between 10 and 24 months of age performed by one of six surgeons.

Standard cleft palate management was supplemented with study visits to the research clinic pre- and postpalatoplasty and then yearly to 6 years of age for assessments of middle ear status by interval history, otoscopy, and tympanometry.

The main outcome measure was age at otitis media resolution defined as the age in years at the first in a sequence of “disease-free” diagnoses not interrupted or followed by any other diagnosis.

The cumulative percent OME resolution for ears/children at ages <1, 1, 2, 3, 4, 5 years was 4.1/4.4, 14.3/10.9, 31.6/21.7, 45.9/37.0, 56.1/50.0, and 70.4/60.9%. OME resolution followed a simple linear time curve with slopes of 13.5% (confidence interval [CI] = 12.2% to 14.8%, r² = .99) and 11.9% (CI = 10.1% to 13.6%, r² = .99) resolutions per year for ears and children, respectively.

There is a natural, age-related pattern of resolution for persistent OME that affects most infants and young children with cleft palate that is not affected by palatoplasty.

To explore ultrasonographic evaluation of primary alveolar repair in cleft lip/palate patients and develop a grading system to assess outcomes of graft success.

Sixteen patients with an average age of 2 years 1 month had sonograms performed at various points in their treatment to determine the feasibility of ultrasound in visualizing alveolar bone defects and changes over time postgrafting. A total of 23 sonograms were performed: 21 at an average of 12 months postoperatively and 2 at an average of 1 month preoperatively.

A 10-point grading system was developed assessing three categories: locations of lateral bone bridging across the cleft, quantification of residual defects with air or fluid channels, and locations of calcification. Three operators graded 10 sonograms to assess interobserver reliability, and the scores were also validated against dental radiographs in patients old enough for radiographic imaging.

Linear weighted kappa statistics revealed substantial interobserver agreement for total scores, with an average kappa value of .708. In limited patients with radiographs, a total score of 9/10 correlated with a Chelsea score of 6.5/8 and category A.

Sonographic evaluation, coupled with this novel grading system, shows potential for early assessment of outcomes of graft success when evaluating new techniques of primary alveolar grafting.