Cleft Palate-Craniofacial Journal最新文献

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.

ObjectiveThis 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.MethodsA 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.ResultsSeven 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.ConclusionDental 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.

ObjectiveTo investigate whether the use of buccal fat pad flap during cleft palate repair reduces the risk of post-operative palatal fistula formation.DesignRetrospective analysis of cleft palate repairs undertaken by a single surgeon over the period 2013-2025.SettingTertiary care institution in London.PatientsTwo 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.InterventionsThe utilization of pedicled buccal fat pad flap in primary cleft palate repairs.Main Outcome Measure(s)Development of post-operative palatal fistula.ResultsAmong 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.ConclusionsThis 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.

ObjectiveEvidence 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.DesignParticipants 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.SettingInterviews took place on Microsoft Teams video call, with photos shared on-screen throughout, and lasted an average of 94 min.ParticipantsFourteen women born with cleft aged 20 to 72 living in the United States or United Kingdom took part.ResultsSix 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.ConclusionsViewing 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 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.

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.SettingNeonatal intensive care unit and acute care units of a tertiary children's hospital, with continuity outpatient multidisciplinary craniofacial clinic care.Patients and ParticipantsNeonates with RS and severe base of tongue UAO not stabilized with positioning, and without mechanical ventilation, profound dysphagia, or microstomia.InterventionsAdapt an OAP clinical pathwayMain Outcome Measure(s)Lessons learned, clinical course, and respiratory metrics.ResultsIn 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.ConclusionsThe 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.

ObjectiveTo identify predictors of speech-correcting surgery after primary palatoplasty in patients with cleft palate with or without cleft lip (CP ± L) and to develop pre- and postoperative prediction models.DesignRetrospective cohort study.SettingWilhelmina Children's Hospital, Utrecht, the Netherlands.Patients, ParticipantsA total of 239 patients with CP ± L who underwent primary palatoplasty between 2008 and 2017 and completed standardized speech assessment at age 5.InterventionsStraight-line palatoplasty with intravelar veloplasty (Sommerlad) within the first year of life.Main Outcome Measure(s)Likelihood of speech-correcting surgery after primary palatoplasty. Potential predictors included cleft type, cleft width, age at palatoplasty, associated syndromes, and postoperative complications such as palatal dehiscence and oronasal fistula. Logistic regression models were developed using pre- and postoperative variables. Model performance was assessed by AUROC, calibration, Brier score, and R².ResultsOf 239 patients, 49% required speech-correcting surgery. In the preoperative model, cleft width and presence of syndromes were significant predictors, showing moderate discrimination (AUROC: 0.694; 95% CI: 0.620-0.759) and good calibration. Adding oronasal fistula in the postoperative model minimally improved performance (AUROC: 0.697; 95% CI: 0.621-0.764).ConclusionsA clinically applicable model was developed to predict the likelihood of speech-correcting surgery following primary palatoplasty. Wide clefts, the presence of syndromes, and oronasal fistula were identified as key predictors.

ObjectiveTo 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.DesignRetrospective study with anonymized lateral cephalometric radiographs.SettingDepartment of Orthodontics, Faculty of Dentistry.Patients, ParticipantsThe study included 188 patients with CLP (mean age, 9.18 ± 4.73 years).InterventionsManual 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.ResultsIntraobserver 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.ConclusionsAI-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.