Objective To investigate operative experiences and perspectives of surgeons presented with cleft lip (CL) and/or palate (CP) in children with life-limiting and terminal illnesses.Design Survey study.Setting Electronic.Patients, Participants American Cleft Palate Craniofacial Association surgeon members.Interventions None.Main Outcome Measures Proportion of surgeons who performed cleft lip and/or palate (CL/P) repair, likelihood to operate again, and factors impacting operative decision.Results Response rate was 20.5% (121/589) including 113 surgeons treating CL/P across plastic surgery (63.7%), otolaryngology (23.9%), and oral and maxillofacial surgery (12.4%). More completed CL than CP repairs (59.3% vs 21.2%) for patients with example conditions of holoprosencephaly, cardiac abnormalities, trisomy 13, and trisomy 18. Leading CL repair motivations were "parent/caregiver request" (89.6%, 60/67), "appearance" (62.7%, 42/67), and "feeding" (28.4%, 19/67). Leading CP repair motivations were "parent/caregiver request" (66.7%, 16/24), "feeding" (62.5%, 15/24), and "communication" (54.2%, 13/24). Surgeons who had not attempted CL repair described lacking opportunity (34.8%, 16/46) or unfavorable risk-benefit ratios (37.0%, 17/46) as reasons. A greater proportion of those who had not attempted CP repair cited unfavorable risk-benefit ratios (59.6%, 53/89) versus lacking opportunity (21/89, 23.6%). 100% and 95.8% who repaired CL and CP endorsed they would again.Conclusions Surgeons more commonly repaired CL than CP in children with life-limiting and terminal illnesses. Nearly all would perform these surgeries again. Respondents who did not repair CP often stated risks outweighed benefits. Studies exploring outcomes of CL/P repair in patients with life limiting and terminal illnesses are scarce. More data are needed to help guide these difficult decisions.
Objective To evaluate the safety of immediate postoperative single-dose ketorolac after primary palatoplasty (PP) and assess its efficacy in reducing opioid use and improving other recovery metrics. Design Prospective cohort with historical controls. Setting Tertiary pediatric hospital. Patients, Participants Two hundred forty-nine patients who underwent PP between 2009 and 2023. Interventions Following institutional implementation of routine ketorolac use after PP, 124 patients who received an immediate postoperative single dose of IV ketorolac (median 0.5 mg/kg) were compared with 125 patients who did not receive ketorolac. Main Outcome Measures Safety outcomes included significant bleeding, supplemental oxygen requirements, and 30-day postoperative complications. Efficacy outcomes were assessed using the Face, Legs, Activity, Cry, Consolability (FLACC) scale, postoperative opioid use, time to first oral intake, and antiemetic use. Results No significant differences were observed between groups regarding the minimal rates of postoperative bleeding, need for supplemental oxygen, or other adverse events. Postoperative FLACC scale was significantly lower in the ketorolac group during the first postoperative hour (adjusted difference: 0.54, 95% CI: 0.04-1.03, P = .033) and hours 1 to 3 (adjusted difference: 0.44, 95% CI: 0.02-0.85, P = .039). The ketorolac group also had lower total opioid use during hospitalization, earlier initiation of oral intake, and shorter length of stay, though these differences were not significant after multivariable adjustment. Conclusions Immediate postoperative single-dose ketorolac after PP is associated with improved early postoperative pain control without increasing complication rates. Other observed potential benefits deserve further attention.
Objective Explore barriers and prioritize supports that could increase breast milk feeding (BMF) rates. Design Survey study. Setting Tertiary children's hospital. Patients, Participants 204 mothers [102 with child with cleft palate (CP), 102 with child with intact palate], ages 0-3 years old. Interventions Survey questions about older sibling feeding history, maternal education, and breastfeeding/breast milk pumping experience. Main Outcome Measure(s) Differences in survey responses between groups; associations between socioeconomic (SES) proxies and BMF. Results Median child age at survey completion was 17.7 months (range 9 days-3.9 years). Direct breastfeeding was less common in the group with CP (46.1% vs. 73.5%, P < .001), who were also more frequently advised against breastfeeding (34.0% vs. 10.9%, P < .001) and more often formula fed (94.1% vs. 85.3%, P = .04). Despite this, the control and CP groups had equal initiation of BMF (77.5%), with no significant difference in median duration (1.0 vs. 1.5 months). Sustained BMF to 6 months was seen in 23.1% of CP group and 28.9% of controls. In the CP group, postnatal counseling (odds ratio [OR] 21.8, P < .001), receiving a breast pump (OR 40.8, P < .001), family support (OR 7.44, P < .001), prior experience with BMF (OR 11.4, P < .001), and maternal education (OR 4.30, P = .006) increased the odds of BMF. Proxies of higher SES were associated with longer BMF in the CP group but not controls (all P < .02). Conclusions Targeted supports for mothers of children with CP such as integrating early feeding specialists and education on pump retrieval are vital to decrease the barriers to sustained BMF.
A 6-day-old girl presented with bilateral macrostomia, micrognathia, and a complex cleft palate consisting of a midline submucous cleft, left lateral submucous cleft, and a right lateral cleft with partial soft-palate agenesis. Owing to Pierre Robin Sequence, surgery was deferred until airway stability. Bilateral macrostomia repair was performed at 12 months, followed by one-stage correction of all palatal clefts at 17 months using a right buccal myomucosal flap for oral lining. Rare clefts variably labeled as oblique clefts or soft-palate agenesis should be uniformly termed “lateral cleft palate.”
ObjectiveTo evaluate the validity, reliability, and usability of a newly digitized GOSLON Yardstick model set for unilateral cleft lip and palate (UCLP) outcome assessment. It was hypothesized that digital ratings would demonstrate reliability comparable to traditional plaster models and support consistent outcome comparisons across centers.DesignRetrospective secondary analysis of dental casts. Raters were blinded to patient and institutional identity. Calibration sessions were held prior to assessment. Reliability, workload, and usability were evaluated using validated instruments.SettingOne craniofacial center.ParticipantsThirty-eight Caucasian UCLP patients in mixed dentition, with primary surgical care at a single institution. Seven orthodontic residents (novices) and 5 craniofacial orthodontists (experts) completed all ratings.InterventionsPlaster casts were digitized using a 3Shape TRIOS scanner and uploaded to Sketchfab. Novices rated both formats across 2 sessions post-calibration. Experts completed 2 digital-only sessions after virtual calibration.Main Outcome MeasuresIntra- and inter-rater reliability (Cohen's kappa), GOSLON score distribution, task workload (NASA-TLX), and system usability (SUS).ResultsDigital GOSLON ratings showed moderate-to-high reliability (expert kappa: 0.82-0.86; novice: 0.75-0.81). No significant differences were found between plaster and digital scores for novices. Experts assigned more GOSLON 3 scores; novices assigned more GOSLON 4 (P < .001). Experts preferred digital models; novices preferred plaster. Workload and usability scores were acceptable across both groups.ConclusionsEven though calibration remains critical for novice raters, the Digital GOSLON is a reliable tool for outcomes assessment and may facilitate inter-center comparisons.
ObjectiveThis scoping review aimed to identify patient-reported outcome (PRO) and proxy-report outcome (ProxRO) measures administered to children under 8 years of age with cleft lip and/or palate (CL/P) and to describe the age at which proxy or self-report measures were used.DesignScoping ReviewSettingWhen children are unable to self-report, PROs and ProxROs can be used. The age at which proxy-report is appropriate has not been described in patients with CL/P.Patients, ParticipantsChildren born with CL/P.InterventionsOvid MEDLINE, EMBASE, PsycINFO, the Cochrane Register, and Web of Science were searched from inception until July 2024. Title and abstract screening, full text review, and data extraction were done independently in duplicate. Measures were categorized as PROs, ProxROs, or "MultiROs," where both caregivers and children responded. A narrative synthesis was performed to describe the identified measures.Main Outcome MeasuresPRO or ProxRO measures in children with CL/P less than 8 years old.ResultsOf 7001 publications identified, 57 studies met the inclusion criteria. Fifty-six measures were identified, of which 43 had at least 1 published psychometric property. Thirteen studies used ad hoc measures. Most measures were not condition-specific to CL/P. ProxROs were more commonly utilized than PROs. Parent-proxy tools were used from birth to 8 years, while self-report was used in patients as young as 3 years old.ConclusionsThere is a paucity of cleft-specific ProxRO measures that assess outcomes of cleft care in patients under 8 years of age. Work to develop and validate ProxRO measures for CL/P is needed.
ObjectiveTo evaluate skeletal outcomes of the Alt-RAMEC protocol with facemask therapy compared with conventional rapid maxillary expansion (RME) in patients with cleft lip and/or palate.DesignSystematic review and meta-analysis.SettingData from international databases (PubMed, Embase, Scopus, Web of Science, CENTRAL, clinical trials registers, and Google Scholar) were analyzed.Patients/ParticipantsFour studies involving approximately 136 patients with cleft lip and/or palate met inclusion criteria.InterventionsAlternate RME and Constriction (Alt-RAMEC) followed by facemask protraction versus conventional expansion protocols.Main Outcome Measure(s)Cephalometric skeletal parameters (SNA, SNB, and ANB) and maxillary advancement.ResultsAlt-RAMEC with facemask produced a greater increase in SNA compared with controls (SMD 1.04; 95% CI 0.60-1.49; I² = 0%), while SNB changes were non-significant. ANB changes were variable across studies (I² = 96%). Certainty of evidence was moderate for SNA and low for SNB.ConclusionsAlt-RAMEC combined with facemask therapy may yield greater anterior maxillary displacement in cleft lip and palate patients than conventional RME protocols, though evidence remains limited. Standardized multicenter studies with long-term follow-up are needed.
ObjectiveTo evaluate the incidence and characteristics of secondary synostosis following spring-mediated cranioplasty (SMC) for nonsyndromic sagittal craniosynostosis.DesignRetrospective cohort study.SettingSingle tertiary pediatric hospital.Patients/ParticipantsTen patients with isolated sagittal craniosynostosis who underwent primary SMC between 2021 and 2023. Patients with syndromic diagnoses or prior cranial surgery were excluded.InterventionsSMC was performed using 2 to 3 stainless-steel springs following sagittal strip craniectomy. Springs were typically removed 3 to 4 months postoperatively. Follow-up included review of clinical photographs, radiographs, and operative records.Main Outcome MeasuresChange in cephalic index (CI) from preoperative to postoperative assessment and occurrence of secondary suture fusion, particularly coronal synostosis.ResultsMean age at surgery was 4 months (range, 3-6 months). Mean CI improved from 68.5 to 79 following expansion. Three patients (30%) developed left unicoronal synostosis (UCS) during the perioperative period prior to spring removal. One patient required secondary coronal suturectomy for significant frontal asymmetry, while 2 were observed without intervention.ConclusionsSecondary synostosis, particularly UCS, may represent an underrecognized sequela of SMC. Altered biomechanical forces or asymmetric cranial remodeling during distraction may contribute to this finding. Larger, multicenter studies with longitudinal imaging are warranted to determine incidence, risk factors, and preventive strategies.
ObjectiveThis study compares perioperative outcomes of three operative approaches for metopic craniosynostosis: spring-assisted cranioplasty (SAC), strip craniectomy (SC), and fronto-orbital advancement (FOA).DesignRetrospective cohort study.SettingSingle-institution.PatientsPatients with metopic craniosynostosis treated 2021 to 2024.InterventionsSC, FOA, SAC.Main Outcome Measure(s)Perioperative data including blood loss, anesthesia duration, operative duration, hospital length of stay. For SAC, metrics for placement and removal were combined.ResultsSix patients underwent SAC, seven SC, and seven FOA at 4.77 (±1.22), 3.42 (±.46), and 11.38 (±3.65) months, respectively. FOA exhibited increased blood loss (median [IQR]; 200 mL [162.5, 250]), anesthesia time (328.86 min ±49.65) and operative time (230.86 min ±45.38) compared to SC (40 [20, 57.5]; P < .005; 153.29 ± 32.2; P < .001; 70.43 ± 20.11; P < .001) and SAC (50 [40,75]; P < .012; 254.17 ± 32.81; P < .012; 131 ± 24.5; P < .0010).ConclusionsSAC for metopic craniosynostosis has lower blood loss, shorter operative time, and shorter anesthesia time in comparison to FOA. Total hospital stay duration required for SAC (including spring placement and removal procedures) is similar to FOA and greater than SC. Perioperative metrics for SAC are favorable or comparable relative to current standard-of-care procedures.
Objective This systematic review and meta-analysis aim to evaluate the prevalence of dental anomalies in primary versus permanent dentition among individuals with non-syndromic cleft lip and palate (CLP) and to elucidate the differences in these anomalies between the two dentitions. Methods A comprehensive literature search was conducted across PubMed, LILACS, Web of Science, EMBASE, and Scopus. Studies included were those assessing dental anomalies in patients with CLP, with data on both primary and permanent dentition. The review adhered to PRISMA guidelines and included data extraction, risk of bias assessment, and meta-analysis. The Grading of Recommendations, Assessment, Development, and Evaluations (GRADE) approach was utilized to evaluate the quality of evidence. Results Seven retrospective cohort studies met the inclusion criteria. The pooled prevalence of hyperdontia was higher in primary dentition (25%) compared to permanent dentition (12%), while hypodontia was more prevalent in permanent dentition (32%) than in primary dentition (12%). Patients with BCLP exhibited a greater prevalence of hypodontia in permanent dentition. Significant heterogeneity was observed across studies in terms of methodologies and sample sizes. Conclusion Dental anomalies in patients with CLP differ between primary and permanent dentition, with hyperdontia being more prevalent in primary dentition and hypodontia in permanent dentition. Variations in prevalence and types of anomalies between patients with UCLP and BCLP highlight the need for standardized diagnostic protocols. Future research should address methodological inconsistencies to improve the robustness of findings.
Objective To investigate whether the use of buccal fat pad flap during cleft palate repair reduces the risk of post-operative palatal fistula formation. Design Retrospective analysis of cleft palate repairs undertaken by a single surgeon over the period 2013–2025. Setting Tertiary care institution in London. Patients Two hundred and seventy-seven patients (146 female, 131 male) who underwent cleft palate repair between 2013 and 2025 were included in the study. One hundred and seventy-three patients had an isolated cleft palate, 78 patients had a unilateral cleft lip and palate, and 26 patients had a bilateral cleft lip and palate. The range of cleft width treated was 3–22 mm. The standard surgical technique employed a Sommerlad style intravelar veloplasty with von Langenbeck releases where necessary. Interventions The utilization of pedicled buccal fat pad flap in primary cleft palate repairs. Main Outcome Measure(s) Development of post-operative palatal fistula. Results Among 277 patients, 21 (7.6%) patients developed a post-operative fistula, most commonly in Zone IV (52%). The fistula rate was 3.7% with buccal fat pad flap use compared with 8.5% without ( P = 0.39). Bayesian logistic regression showed a two-thirds reduction in the odds of fistula formation (odds ratio [OR] = 0.33; 95% credible interval [CrI]: 0.15–0.74), suggesting a high probability of a protective effect. Conclusions This study demonstrates that the use of buccal fat pad flaps in primary cleft palate repair is an effective adjunct associated with a reduced incidence of post-operative oronasal fistula.
ObjectiveTo compare outcomes of cranial vault remodeling via fronto-orbital advancement and remodeling (FOAR) versus spring cranioplasty in patients with nonsyndromic unicoronal craniosynostosis (UCS), focusing on surgical parameters and craniofacial symmetry improvements.DesignRetrospective review comparing FOAR and spring-assisted cranioplasty outcomes using preoperative and postoperative CT data. Statistical analysis was performed using SPSS Statistics 30.0.SettingInstitutional tertiary care center specializing in craniofacial surgery.Patients, ParticipantsNonsyndromic patients with UCS undergoing FOAR (n = 7) or spring cranioplasty (n = 11), with complete pre- and postoperative imaging and surgical data available.InterventionsFOAR involved traditional fronto-orbital advancement and remodeling. Spring cranioplasty consisted of spring placement followed by spring removal surgery, designed to remodel cranial vault less invasively.Main Outcome Measure(s)Retrospective review of surgical time, estimated blood loss (EBL), packed red blood cells (PRBC) transfused, intracranial volume (ICV), facial twist (FT), skull-base twist relative to palate (SBT-P) and nasion (SBT-N), and orbital morphology symmetry.ResultsSpring cranioplasty, when considering both placement and removal, significantly reduced operative time (157.9 ± 28.6 vs 264.7 ± 48.3 min), EBL (5.5 ± 1.6 vs 26.5 ± 9.2 mL/kg), and PRBC transfusion (2 ± 4.2 vs 27.4 ± 5.9 mL/kg) compared with FOAR (P < .001). Postoperatively, patients who underwent spring cranioplasty demonstrated significant improvements in FT, SBT-P, and SBT-N (P = .003), whereas FOAR did not achieve significant changes. ICV did not differ significantly between groups. Spring cranioplasty patients demonstrated superior orbital symmetry improvement.ConclusionsSpring cranioplasty offers a less invasive alternative to FOAR for UCS, with shorter surgery, less blood loss, and better craniofacial symmetry correction without compromising ICV expansion.
Objective The purpose of this study was to determine factors predicting communication-related quality of life in children with cleft palate with or without cleft lip (CP ± L) or congenital velopharyngeal insufficiency (VPI). Design Cross-sectional design. Setting Outpatient pediatric craniofacial anomalies clinic. Patients Two-hundred and seventy-one children <18 years of age, diagnosed with CP ± L or congenital VPI. Main Outcome Measure Velopharyngeal Insufficiency Quality of Life (VELO) scores as well as articulation, speech intelligibility, resonance, and voice outcomes were extracted from the electronic medical record in order to characterize communication-related quality of life. Total VELO score, as well as speech limitations, situational difficulty, emotional impact, perception by others and swallow subscores, were collected. Demographic factors, economic deprivation, medical history, and history of palatoplasty/pharyngoplasty were considered. Linear regression identified predictors of VELO total and subscores. Results Poorer speech intelligibility and increased hypernasality significantly predicted lower (worse) total VELO scores ( P < .01), as well as more severe speech limitations ( P < .01), situational difficulty ( P < .01), emotional impact ( P < .01), and perception by others ( P < .01). The presence of glottal stop substitutions also predicted more severe emotional impact ( P < .01). A comorbid behavioral health condition predicted significantly lower total VELO scores ( P < .01), and children living in neighborhoods experiencing greater socioeconomic deprivation presented with worse swallow section scores ( P < .01). Conclusions Hypernasality and decreased speech intelligibility primarily influenced the communication-related quality of life in children with velopharyngeal dysfunction. Additional research is warranted to determine how intervention can best mitigate VPI-related challenges in order to improve social interaction and quality of life.
Objectives (1) Provide our narrative experience treating patients with Robin Sequence (RS) with our orthodontic airway plate (OAP) protocol and (2) report initial patient outcomes. A secondary objective is to share OAP clinician survey results.Design (1) Narrative experience of a care pathway for neonates with RS and upper airway obstruction (UAO). (2) Retrospective review of initial series of patients with RS treated with this pathway. A clinician survey was also administered.Setting Neonatal intensive care unit and acute care units of a tertiary children's hospital, with continuity outpatient multidisciplinary craniofacial clinic care.Patients and Participants Neonates with RS and severe base of tongue UAO not stabilized with positioning, and without mechanical ventilation, profound dysphagia, or microstomia.Interventions Adapt an OAP clinical pathwayMain Outcome Measure(s) Lessons learned, clinical course, and respiratory metrics.Results In August 2023, our institution launched an OAP treatment pathway. Lessons learned highlight the importance of multidisciplinary communication, patient selection, and patience. Nine patients completed OAP therapy. oAHI values improved from mean 71/h (range 16-189, SD 55.1) to mean oAHI 7.9/h (range 1.1-12.7, SD 3.8). Pretreatment, 7 patients were on high-flow nasal cannula, one each on low-flow nasal cannula and continuous positive airway pressure. All OAP graduates advanced to room air. Clinician survey results suggested high OAP care preparedness and satisfaction.Conclusions The OAP can be introduced in new setting and delivered as a safe and effective nonsurgical intervention for UAO in RS. Interdisciplinary collaboration is key to treatment success. Infants undergoing OAP treatment can safely discharge home and experience sustained respiratory improvements.
Objective To evaluate the accuracy of the artificial intelligence (AI)-powered orthodontic imaging system OrthoDx™ for cephalometric analysis in patients with cleft lip and/or palate (CLP), compared with manual semi-automated measurements obtained using Dolphin Imaging software. Design Retrospective study with anonymized lateral cephalometric radiographs. Setting Department of Orthodontics, Faculty of Dentistry. Patients, Participants The study included 188 patients with CLP (mean age, 9.18 ± 4.73 years). Interventions Manual cephalometric analysis was performed using Dolphin Imaging software used as reference, while AI-based and examiner-corrected analyses were conducted using OrthoDx™. Seventeen angular and eight linear cephalometric parameters were analyzed. Main Outcome Measure(s) Primary outcome was the agreement between manual, AI, and examiner-corrected AI cephalometric measurements, assessed using intraclass correlation coefficients (ICCs), one-sample t-tests, and Bland-Altman analyses. Primary outcome measures were defined prior to data collection. Results Intraobserver reliability for the manual method showed good to excellent reliability with no significant differences between repeated measurements. SNA, saddle, articular, and U1-FH angles differed significantly between manual and AI methods but not after examiner correction. Significant differences were observed between manual and AI, and between manual and corrected AI, for several other parameters. ICCs ranged from moderate (0.70-0.75) to excellent (>0.90), indicating variable agreement across parameters. Conclusions AI-based cephalometric analysis using OrthoDx™ demonstrated limited accuracy in patients with CLP. Examiner intervention reduced the variability of certain cephalometric measurements, making the results closer to the manual group, supporting the role of clinician-supervised AI as a complementary rather than replacement tool.
Objective To propose recommendations for ethical, participant-centered clinical data sharing in craniofacial research.Design Series of deliberative multidisciplinary expert working group meetings to develop recommendations.Setting Two 1-h virtual meetings; one all-day hybrid meeting.Patients, Participants Working group (n = 16) comprised individuals with expertise in craniofacial research, bioethics, and patient/caregiver advocacy, as well as lived experience of craniofacial conditions.Interventions The working group first reviewed prior empirical data about research participant attitudes about data sharing then drafted initial recommendations that were built on the data and the group's collective expertise. Recommendations were iteratively refined until the group agreed upon their final presentation.Main outcome measures Working group endorsement of recommendations.Results The working group produced 16 recommendations that addressed considerations for primary and secondary researchers, data repositories, and the craniofacial research community across 5 domains. These domains address: (1) research team communication with participants, (2) data collection and protections, (3) data governance, (4) education for researchers, and (5) remaining research gaps. Recommendations highlight the importance of prioritizing the experiences of those with lived experience of craniofacial conditions in decision-making about data sharing, navigating varied perspectives on privacy protections for facial images, and striving to implement trustworthy data sharing and governance practices.Conclusions This summary of recommendations offers guidance for the craniofacial research community to advance participant-centered clinical data sharing practices. Ethical data sharing that accounts for participants' experiences and values has potential to advance scientific research and improve outcomes for individuals with craniofacial conditions, their families, and their communities.
Objective Evidence suggests that cleft lip and/or palate can have significant psychosocial impacts on adults' lives beyond the end of the standard treatment pathway. Although some studies indicate gender differences, no research has specifically explored the experiences of women with cleft. This study aimed to explore the stories told by women about their adult experiences of cleft, for discussion informed by intersectionality theory.Design Participants were interviewed using a narrative, photo elicitation approach. Each participant brought photos they felt represented their story to an unstructured interview, which was recorded and transcribed verbatim. Data were subject to narrative analysis.Setting Interviews took place on Microsoft Teams video call, with photos shared on-screen throughout, and lasted an average of 94 min.Participants Fourteen women born with cleft aged 20 to 72 living in the United States or United Kingdom took part.Results Six overarching discourses were identified and presented alongside 14 individual narrative synopses, depicting converging stories told by each participant throughout their interview. These were (1) enduring, (2) hiding, (3) striving, (4) healing, (5) reclaiming, and (6) reconciling.Conclusions Viewing cleft through an intersectional lens offers important insights into the lived and living experiences of adults navigating treatment and the ongoing psychosocial impact of cleft. The findings suggest that expectations and pressures placed on women by society are compounded by, rather than exist in parallel with, the ongoing impact of cleft.
ObjectiveTo describe characteristic CLEFT-Q response profiles and patterns in patients with cleft palate and/or lip (CP ± L).DesignRetrospective analysis using latent profile analysis (LPA) to categorize patient-reported outcome responses into distinct profiles.SettingTertiary care pediatric hospital with multidisciplinary cleft team.Patients, ParticipantsPatients aged 8-29 years with CP ± L completing CLEFT-Q questionnaires from September 2021 to June 2025 (n = 596). Total of 923 responses analyzed after excluding 94 responses from patients under 8 years.InterventionsAdministration of the 119-item CLEFT-Q questionnaire prior to clinic visits, measuring appearance, function, and quality of life.Main Outcome Measure(s)Six latent profiles identified through LPA based on CLEFT-Q scores. Secondary outcomes included profile stability and surgical effects.ResultsSix profiles of CLEFT-Q responses emerged among subjects: Universally Affected-Moderate (UA1, n = 228, 24.7%), Universally Affected-Severe (UA2, n = 119, 12.9%), Facial Appearance-Moderate (FA1, n = 183, 19.8%), Facial Appearance-Severe (FA2, n = 56, 6.1%), Social/School/Speech distress (SSS, n = 95, 10.3%), and Minimally Distressed (MD, n = 242, 26.2%). Patients in severe profiles (UA2 and FA2) were older (P < .001) and more likely to have complete cleft lip (P < .001), while patients in the MD profile were younger and more likely to have isolated cleft palate (P < .001). Among 279 patients with longitudinal CLEFT-Q data, 47.5% remained in the same profile at subsequent CLEFT-Q response, with longer follow-up intervals predicting profile change (P = .049).ConclusionsLatent profile analysis categorizes CLEFT-Q responses into six clinically meaningful profiles correlating with demographics and treatment outcomes. This approach may guide personalized cleft care, though additional studies are needed for validation.
ObjectiveExamine factors that impact ratings of speech acceptability and parent-reported speech intelligibility using the Intelligibility in Context Scale (ICS).DesignProspective, cross-sectional study.SettingPublic research university.PatientsThirty children 4 to 7 years old with a repaired cleft palate.VariablesPerceptual ratings derived from the Cleft Audit Protocol for Speech-Augmented-Americleft Modification, ICS, percent consonants correct (PCC), age, and sex.Main Outcome Measure(s)Factors associated with speech accepatability were assessed using Spearman's rho (ρ) and chi-square tests. Factors that predicted the average score on the ICS were assessed using linear regression.ResultsHypernasality ratings and PCC were associated with ratings of speech acceptability. There was a moderate, positive correlation between ratings of hypernasality and ratings of speech acceptability [ρ = .51, P < .01]. As hypernasality ratings increased, speech was rated as less acceptable. There was a moderate, negative correlation between PCC and ratings of speech acceptability [ρ = -.67, P < .01]. Children who produced more consonants correct were rated as having more acceptable speech. PCC predicted (P = .010) the average total score on the ICS. As PCC increased, the average score on the ICS also increased. While PCC predicted ICS scores, hypernasality ratings significantly predicted PCC (P = .023) and percent of compensatory misarticulation errors used (P = .001).ConclusionsSpeech-language pathologists need to develop individualized treatment plans that may include (1) recommending speech therapy to increase consonant accuracy that would improve both speech acceptability and intelligibility and (2) identifying children who require surgical intervention to address moderate-to-severe hypernasality, which may be impacting consonant production.
Objective Assess the patient compliance rate for nasopharyngoscopy as documented in clinical reports.Design Cross-sectional.Setting Thirteen cleft teams in North America.Patients Patients aged 3 to 21 years old with a repaired cleft palate.Interventions Nasopharyngoscopy.Main Outcome Measure Patient compliance rate for nasopharyngoscopy.Results Patient compliance was documented in 128 of 158 reports (81%). Of the 128 reports, patient compliance was reported as "good/excellent" in 65% (n = 83), "marginal/fair" across 10% (n = 13), "poor" in 16% (n = 20), and "other" in 9% (n = 12). Patients with "poor" compliance had lower rates of documented imaging ratings, however 55% (n = 11) of reports included a surgical recommendation.Conclusions Among children completing nasopharyngoscopy for velopharyngeal insufficiency surgery planning, at least one-fourth of reports indicate significant compliance issues that limits velopharyngeal port imaging. These findings suggest that some cleft teams and surgeons may be proceeding with surgical management of velopharyngeal insufficiency without adequate visualization or ratings of velopharyngeal anatomy.
