AimTo investigate the neuroendocrine axis in patients with arhinia, hemiarhinia, and their associated malformations.MethodsData were manually extracted from medical records at Hospital de Reabilitação de Anomalias Cranio...AimTo investigate the neuroendocrine axis in patients with arhinia, hemiarhinia, and their associated malformations.MethodsData were manually extracted from medical records at Hospital de Reabilitação de Anomalias Craniofaciais, Universidade de São Paulo (HRAC-USP) using a newly developed protocol. They were explored through a descriptive and qualitative manner. This study was approved by the HRAC-USP Ethics Committee and is part of a large research project on this topic.ResultsNine patients were included in the final sample, of which three had isolated hemiarhinia, three had hemiarhinia with additional malformations, and three were diagnosed with Bosma arhinia-microphthalmia (BAM) syndrome. Among those with isolated hemiarhinia, two presented neuroendocrine alterations (primary hypothyroidism, micropenis with bilateral cryptorchidism, and weight and height deficits). Patients with hemiarhinia and associated malformations demonstrated no clinical or laboratory evidence of neuroendocrine dysfunction. In the BAM syndrome group, two patients exhibited central hypogonadotropic dysfunction, with undetectable luteinizing hormone and follicle-stimulating hormone levels. The third patient showed no abnormalities.ConclusionsArhinia, hemiarhinia, and their associated malformations encompass a broad spectrum of manifestations that can affect multiple organs and systems, particularly the neuroendocrine system. Despite their potential impact, screening for these conditions remains poorly conducted, which may lead to underdiagnosis or delayed recognition of associated complications. Consequently, a comprehensive evaluation is essential to ensure appropriate monitoring and management of affected patients.
Cleft Palate Craniofac J
· 2026 Mar · PMID 41451897
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ObjectiveTo compare cross-linguistic auditory-perceptual nasality ratings and language-specific nasalance scores in Spanish-English bilingual children and adolescents with cleft palate.DesignRetrospective case series.Set...ObjectiveTo compare cross-linguistic auditory-perceptual nasality ratings and language-specific nasalance scores in Spanish-English bilingual children and adolescents with cleft palate.DesignRetrospective case series.SettingAcademic, tertiary pediatric hospital.ParticipantsSpanish- and English-speaking patients with cleft palate who underwent a bilingual resonance and articulation evaluation.Interventions/ComparisonsAuditory-perceptual nasality ratings versus nasalance scores.Main Outcome MeasuresCross-linguistic ratings of hypernasality, hyponasality, and audible nasal emission; mean and maximum nasalance scores for oral- and nasal-loaded stimuli in each language.ResultsEleven patients' data were analyzed (six females and five males; mean age, 11.3 years). Oral-loaded mean nasalance strongly reflected perceptual hypernasality in both languages; hyponasality ratings showed no relationship to mean nasalance with nasal-loaded stimuli in either language; and audible nasal emissions were associated with maximum nasalance in Spanish but not English. Repeated-measures analyses revealed no significant effect of phoneme context, language, or their interaction on either mean or maximum nasalance.ConclusionsOral-loaded mean nasalance measures strongly reflect perceptual hypernasality in both languages. Audible nasal emissions are significantly associated with maximum nasalance scores in Spanish only, suggesting possible language-specific perceptual and acoustic patterns. Further research is needed to determine if these conclusions hold in a larger, prospective cohort.
ObjectiveTo characterize the audiological and sociodemographic profiles of children diagnosed with isolated Robin sequence (RS) and cleft palate (CP) during the first year of life. In addition, this study aimed to propos...ObjectiveTo characterize the audiological and sociodemographic profiles of children diagnosed with isolated Robin sequence (RS) and cleft palate (CP) during the first year of life. In addition, this study aimed to propose an audiological assessment protocol for early identification of hearing loss (HL) in this population.DesignRetrospective longitudinal study analyzing secondary data from medical records.SettingSpecial Care Unit at the Hospital for Rehabilitation of Craniofacial Anomalies.PatientsFifty-eight children aged 0 to 12 months diagnosed with isolated RS and CP.InterventionsNo interventions were performed.Main Outcome MeasuresAudiological assessments revealed a high prevalence of bilateral conductive HL ranging from mild to moderate severity.ResultsMost participants resided in São Paulo state and belonged to lower-upper socioeconomic backgrounds. The most frequent risk factor for early childhood HL was neonatal intensive care for more than 5 days. No statistically significant association was found between HL risk indicators and the presence of conductive HL.ConclusionsConductive HL was identified in 39.64% of children with isolated RS and CP, with a higher prevalence observed in females. The sample demonstrated no cases of permanent and disabling HL. We recommend implementing a standardized audiological assessment protocol for infants with isolated RS and CP during the first year of life, including tympanometry and air-conduction click-evoked auditory brainstem response (ABR), with bone conduction ABR and otoscopic examination performed when clinically indicated.
ObjectiveTo evaluate maxillary growth differences when buccal fat pad graft (BFP) is utilized during primary cleft palate repair, and to evaluate the need for later surgical revision of the BFP due to non-eruption of max...ObjectiveTo evaluate maxillary growth differences when buccal fat pad graft (BFP) is utilized during primary cleft palate repair, and to evaluate the need for later surgical revision of the BFP due to non-eruption of maxillary permanent molars.DesignRetrospective cohort study.SettingInstitutional hospital and clinic.PatientsPatients with CP ± L who underwent primary palatoplasty with or without BFP.InterventionsMaxillary measurements of pre-orthodontic dental models.Main outcome measureMaxillary dimensions, disruption to maxillary permanent molar eruption.ResultsPatients treated with BFP exhibited a tendency toward an increased maxillary posterior width and a more favorable maxillomandibular posterior transverse relationship ( = 0.069 and 0.072, respectively). A similar percentage of patients required maxillary expansion between the non-BFP and BFP-treated group ( = 0.103). Secondary surgical revision was recommended for 70.6% of BFPs placed due to inhibition of eruption of the maxillary permanent molars. 33.3% of BFP revisions were not combined with any other surgical procedure during the general anesthesia event.ConclusionThe use of BFP adjunctive flap during primary palatoplasty likely allows for increased transverse growth of the posterior maxilla during childhood and likely reduces the maxillomandibular posterior arch width discrepancy. However, the use of this graft does not reduce the proportion of patients who require orthodontic maxillary expansion. Patients may experience disruption of maxillary permanent molar eruption, and 70.6% of BFPs placed require secondary surgical intervention under general anesthesia to reposition the flap in late childhood.
ObjectiveThis study aimed to evaluate the accuracy of different mixed dentition period analysis methods in individuals with cleft lip and palate (CLP).DesignRetrospective comparative study.SettingDepartment of Orthodonti...ObjectiveThis study aimed to evaluate the accuracy of different mixed dentition period analysis methods in individuals with cleft lip and palate (CLP).DesignRetrospective comparative study.SettingDepartment of Orthodontics, Erciyes University, Turkiye.Patients/ParticipantsA total of 70 individuals aged 13 to 16 years were included in the study: 35 with CLP and 35 noncleft controls. In all individuals included in the study, all permanent teeth had erupted except for the maxillary lateral incisors, which are frequently missing in individuals with CLP.InterventionsFor formulas suggested by 8 different prediction methods (Tanaka-Johnston, Moyers 50%, Moyers 75%, Boboc, Cattaneo, Camilo, Barnabe, and Melgaço), the MD and buccolingual dimensions of incisors and molars, as well as the mesiodistal dimensions of erupted canines and premolars, were measured on the 3-dimensional dental models.Main Outcome MeasuresComparison of mean difference between actual and predicted permanent canine and premolar widths, using the control sample as a benchmark to judge model suitability in the CLP population.ResultsAll regression methods showed significant differences between predicted and actual values in the CLP group ( < .05). Boboc and Cattaneo had the fewest errors, while Tanaka-Johnston and Barnabé showed the most significant overestimation. In controls, Cattaneo was most accurate in the maxilla and Tanaka-Johnston in the mandible.ConclusionsThe Boboc and Cattaneo methods provided the most accurate predictions of permanent canine and premolar widths in patients with CLP, while the Tanaka-Johnston and Barnabé methods consistently overestimated values.
ObjectiveTo evaluate and compare the morphology of the pterygomaxillary suture (PMS) in unilateral cleft lip and palate (UCLP), bilateral cleft lip and palate (BCLP), and non-cleft (NC) individuals using cone-beam comput...ObjectiveTo evaluate and compare the morphology of the pterygomaxillary suture (PMS) in unilateral cleft lip and palate (UCLP), bilateral cleft lip and palate (BCLP), and non-cleft (NC) individuals using cone-beam computed tomography (CBCT), focusing on side-specific differences relevant to Le Fort I osteotomy planning.DesignRetrospective cross-sectional study.SettingOral and maxillofacial radiology clinic of a university-affiliated center, where CBCT data were originally obtained for diagnostic purposes.PatientsOne hundred and fifty-six subjects-57 UCLP, 21 BCLP, and 78 NC-matched for age and sex ( > .25). Inclusion required complete records and high-quality CBCT scans; patients with syndromic anomalies or prior orthognathic surgery were excluded.InterventionsCBCT scans were retrospectively analyzed using standardized NNT software (v 6.2). Anatomical landmarks were referenced to the midsagittal plane for reproducibility.Main Outcome MeasuresSeven PMS parameters-thickness, width, length, angulation, lateral pterygoid plate (LPP) length, medial pterygoid plate (MPP) length, and distance to the greater palatine foramen (GPF)-were compared using ANOVA, paired-sample -tests, and Tukey post-hoc analyses (α = 0.05).ResultsPMS angulation was higher in UCLP (79.82 ± 7.96°) and BCLP (79.84 ± 9.11°) compared with NC (75.90 ± 8.07; < .001). In UCLP, the cleft side showed greater angulation and shorter LPP ( = .027; = .001). Other parameters did not differ significantly.ConclusionsCLP patients show increased PMS angulation and cleft-side LPP shortening compared with controls. These variations may be relevant to surgical planning; however, the present study did not assess surgical outcomes. Future research should determine whether such differences influence intraoperative complexity.
ObjectivesTo explore individual speech-language pathologists' (SLPs) experiences in the clinical practice of cleft palate speech/velopharyngeal dysfunction (VPD), and to identify the need for and type of (further) traini...ObjectivesTo explore individual speech-language pathologists' (SLPs) experiences in the clinical practice of cleft palate speech/velopharyngeal dysfunction (VPD), and to identify the need for and type of (further) training necessary.DesignDescriptive and exploratory survey using QualtricsXM online platform, with subsequent in-person/Zoom Workplace platform interviews.SettingUniversity research and clinical teamParticipantsMembers of the Hong Kong Association of Speech Therapists and the Macao Association of Speech TherapistsInterventionsThe survey consisted of 39 items across 7 sections, for example, continuing professional development and assessment and treatment practices of cleft palate speech. Skip logic was applied to enhance survey efficiency and experience. Interviews were conducted in various modes.Main Outcome Measure(s)The Checklist for Reporting Results of Internet E-Surveys (CHERRIES) was used. Quantitative measures include proportions and percentages; qualitative data were summarized from the survey and subsequent interviews.Results90% reported receiving teaching in cleft palate speech/VPD ranging from 0 to 96 h ( = 12.7, SD = 15.58). Over half of respondents felt not (very) confident when undertaking assessment (61%) and treatment (54%). Lack of exposure to real clients was a key reason for low confidence levels. Almost all respondents expressed a strong need for continuing professional development in the area.ConclusionsStudy findings highlight the need for further training in cleft palate speech/VPD for SLPs in the region and emphasize the importance of continuing professional development to enhance skills and confidence in assessment and treatment, ultimately improving care and outcomes for individuals with cleft palate with or without lip.
ObjectiveTo systematically review advances in surgical techniques for secondary cleft palate repair, emphasizing their impact on velopharyngeal function, speech outcomes, and the methodological validity of speech assessm...ObjectiveTo systematically review advances in surgical techniques for secondary cleft palate repair, emphasizing their impact on velopharyngeal function, speech outcomes, and the methodological validity of speech assessments used in published studiesDesignFollowing PRISMA 2021 guidelines, six electronic databases were searched for articles from January 2012 to February 2025 using MeSH terms related to secondary cleft palate repair, velopharyngeal insufficiency, palatoplasty, and speech outcomes. Eligible studies included clinical reports with ≥10 patients undergoing secondary repair. Data on surgical methods, outcomes, and complications were extracted and qualitatively synthesized due to heterogeneity across studies.SettingAll published clinical studies evaluating secondary cleft palate repair outcomes.Patients/ParticipantsIndividuals presenting with residual velopharyngeal insufficiency, recurrent fistula, or speech dysfunction following primary palatoplasty.Main Outcome MeasuresSpeech resonance and intelligibility, velopharyngeal closure rate, fistula recurrence, donor-site morbidity, and obstructive sleep apnea risk.ResultsFourteen studies met the inclusion criteria. Palate-based re-repair with Furlow double-opposing Z-plasty and buccal myomucosal flaps improved resonance and closure in small to moderate gaps. Pharyngeal flap and sphincter pharyngoplasty achieved satisfactory closure in larger defects but increased the risk of airway obstruction. However, most studies lacked validated speech protocols or controlled for articulatory errors and fistula effects, limiting confidence in the interpretation of outcomes.ConclusionsWhile secondary repairs often improve resonance and velopharyngeal competence, evidence remains constrained by heterogeneity and non-validated assessment methods. Future multicenter research integrating standardized, speech pathologist-verified protocols is essential to establish evidence-based algorithms for secondary cleft palate repair.
Sullivan LE, Alter NE, Hiller AR
… +4 more, Braun SA, Galdyn IA, Golinko MS, Pontell ME
Cleft Palate Craniofac J
· 2026 Mar · PMID 41417938
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ObjectiveTo investigate operative experiences and perspectives of surgeons presented with cleft lip (CL) and/or palate (CP) in children with life-limiting and terminal illnesses.DesignSurvey study.SettingElectronic.Patie...ObjectiveTo investigate operative experiences and perspectives of surgeons presented with cleft lip (CL) and/or palate (CP) in children with life-limiting and terminal illnesses.DesignSurvey study.SettingElectronic.Patients, ParticipantsAmerican Cleft Palate Craniofacial Association surgeon members.InterventionsNone.Main Outcome MeasuresProportion of surgeons who performed cleft lip and/or palate (CL/P) repair, likelihood to operate again, and factors impacting operative decision.ResultsResponse 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.ConclusionsSurgeons 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.
ObjectiveExplore barriers and prioritize supports that could increase breast milk feeding (BMF) rates.DesignSurvey study.SettingTertiary children's hospital.Patients, Participants204 mothers [102 with child with cleft pa...ObjectiveExplore barriers and prioritize supports that could increase breast milk feeding (BMF) rates.DesignSurvey study.SettingTertiary children's hospital.Patients, Participants204 mothers [102 with child with cleft palate (CP), 102 with child with intact palate], ages 0-3 years old.InterventionsSurvey 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.ResultsMedian 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%, < .001), who were also more frequently advised against breastfeeding (34.0% vs. 10.9%, < .001) and more often formula fed (94.1% vs. 85.3%, = .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, < .001), receiving a breast pump (OR 40.8, < .001), family support (OR 7.44, < .001), prior experience with BMF (OR 11.4, < .001), and maternal education (OR 4.30, = .006) increased the odds of BMF. Proxies of higher SES were associated with longer BMF in the CP group but not controls (all < .02).ConclusionsTargeted 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.
ObjectiveTo 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.DesignProspective cohor...ObjectiveTo 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.DesignProspective cohort with historical controls.SettingTertiary pediatric hospital.Patients, ParticipantsTwo hundred forty-nine patients who underwent PP between 2009 and 2023.InterventionsFollowing 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 MeasuresSafety 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.ResultsNo 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, = .033) and hours 1 to 3 (adjusted difference: 0.44, 95% CI: 0.02-0.85, = .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.ConclusionsImmediate 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.
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 ag...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...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 ( < .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 t...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.
IntroductionThe vomer flap is a versatile surgical technique used in cleft palate repair, primarily to reconstruct the nasal layer of the hard palate. Its implementation has gained renewed interest due to its anatomical...IntroductionThe vomer flap is a versatile surgical technique used in cleft palate repair, primarily to reconstruct the nasal layer of the hard palate. Its implementation has gained renewed interest due to its anatomical advantages and potential for reducing postoperative complications, especially oronasal fistulas.ObjectiveThis narrative review aims to evaluate the historical evolution, technical variations, clinical indications, outcomes, and limitations of the vomer flap in both unilateral and bilateral cleft palate repair.MethodsA systematic literature search was conducted across PubMed, Embase, and Cochrane Library databases, including studies published from 1981 to April 2024. Articles were selected based on relevance to vomer flap use in cleft surgery, focusing on surgical outcomes, functional results, complication rates, and long-term effects on maxillary growth.ResultsThree main techniques were identified for unilateral clefts: cephalically based, caudally based, and open-book vomer flaps. For bilateral clefts, cephalically based and open-book configurations demonstrated superior anatomical adaptation and lower fistula rates compared to caudally based flaps. The vomer flap is particularly advantageous in early repairs and wide clefts, with evidence suggesting a minimal adverse effect on maxillary development. However, high variability in technique and outcome reporting limits comparability.ConclusionThe vomer flap remains a valuable tool in cleft palate repair, offering a balance between surgical efficacy and preservation of growth potential. It's appropriate application, based on cleft morphology and surgical objectives, can enhance functional and aesthetic outcomes. Further prospective studies are needed to standardize technique and validate long-term benefits.
ObjectiveTo evaluate the incidence and characteristics of secondary synostosis following spring-mediated cranioplasty (SMC) for nonsyndromic sagittal craniosynostosis.DesignRetrospective cohort study.SettingSingle tertia...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.
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-...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; ² = 0%), while SNB changes were non-significant. ANB changes were variable across studies (² = 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.
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...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.
ObjectiveThis study compares perioperative outcomes of three operative approaches for metopic craniosynostosis: spring-assisted cranioplasty (SAC), strip craniectomy (SC), and fronto-orbital advancement (FOA).DesignRetro...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]; < .005; 153.29 ± 32.2; < .001; 70.43 ± 20.11; < .001) and SAC (50 [40,75]; < .012; 254.17 ± 32.81; < .012; 131 ± 24.5; < .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.