Cleft Palate-Craniofacial Journal最新文献

ObjectiveThe triad of micrognathia, glossoptosis, and airway obstruction defines Robin sequence (RS). Syndromic RS accounts for about 60% of cases, but whether mandibular "catch-up" growth occurs in these cases remains unclear. The aim of the systematic review was to assess the current literature for evidence of catch-up growth in syndromic RS.DesignA PRISMA-guided systematic literature review was performed. PubMed was searched for studies describing cases with syndromic RS who underwent conservative (non-surgical) management.Patients, ParticipantsCase reports, case series, and chart reviews containing both the diagnosis of RS and at least one associated syndrome were included. Review articles, non-English publications, and studies lacking sufficient follow-up or clarity on mandibular morphology were excluded.Main Outcome MeasuresThe review assessed micrognathia, mandibular length, catch-up growth, and the necessity of airway enhancement.ResultsThirty-four studies met the criteria for inclusion. These studies included clinical information from a total of 202 cases and addressed 20 different syndromes. RS in the context of Stickler syndrome and 22q11.2 deletion syndrome were most frequently reported, with multiple studies suggesting potential for balanced maxillo-mandibular growth or apparent catch-up growth. However, definitive conclusions were limited by small study population, heterogeneous methodologies, and variable follow-up durations.ConclusionWhile additional data are needed, available literature suggests that certain syndromic RS groups may exhibit spontaneous mandibular growth, potentially reducing the need for invasive procedures such as distraction osteogenesis. Prospective research employing standardized cephalometric measures is necessary to identify which syndromic subpopulations and RS types benefit from conservative versus surgical management.

ObjectiveTo assess volumetric and morphological changes in the maxillary sinus of patients with cleft lip and palate (CLP) undergoing Le Fort I osteotomy using cone-beam computed tomography (CBCT) and 3D segmentation.DesignRetrospective observational pilot study.SettingA specialized CLP treatment center in the oral and maxillofacial surgery department.Patients/ParticipantsThirty adults with CLP (15 unilateral, 15 bilateral) who underwent Le Fort I osteotomy, with preoperative and ≥12-month postoperative CBCT scans available.MethodsThree-dimensional segmentation of the maxillary sinus was performed using ITK-SNAP software on pre- and postoperative CBCT scans to calculate volumetric changes.Outcome MeasureMaxillary sinus volume (mm³) before and after surgery.ResultsBoth groups exhibited postoperative reductions in sinus volume. In the unilateral group, mean bilateral volume decreased from 12 272 mm³ to 11 433 mm³; in the bilateral group, from 15 082 mm³ to 14 519 mm³. Although the unilateral group showed slightly greater reduction, differences were not statistically significant within or between groups (P > 0.05).ConclusionsLe Fort I osteotomy led to modest postoperative volumetric reduction in the maxillary sinus in both unilateral and bilateral CLP patients. Bilateral CLP cases consistently exhibited larger sinus volumes pre- and postoperatively compared to unilateral cases. These findings suggest cleft morphology influences sinus anatomy and that surgical intervention may alter its structure. Although volumetric changes were not significant, frequent morphological variations highlight the need for ongoing postoperative radiological monitoring.

An 18-year-old female with incomplete unilateral cleft lip and palate presented with skeletal class I malocclusion, severe crowding, anterior crossbite, unilateral posterior crossbite, congenitally missing maxillary left lateral incisor, and root dilacerations. The aims of the treatment were to achieve good occlusion and improve psychological well-being. Orthodontic treatment with a preadjusted edgewise MBT 0.022 bracket system and asymmetric Hyrax-type expander achieved proper dental alignment and unilateral transverse expansion without affecting the non-crossbite side. The Rosenberg Self-Esteem Scale increased from 13 to 27, indicating enhanced psychological well-being. The asymmetric Hyrax-type expander was effective for correcting unilateral posterior crossbite.

ObjectiveThis study examined the effects of intensive hybrid telepractice and in-person speech therapy on speech characteristics in Persian-speaking children.DesignSingle-subject experimental design (ABA type).SettingMobasher Speech Therapy Clinic, Hamadan, Iran.ParticipantsFour Persian-speaking children aged 3 to 12 years with CP ± L and at least one active cleft-type characteristic (CTC).InterventionThirty intensive hybrid sessions (three in-person and two telepractice sessions per week) over six weeks.Main Outcome MeasuresPercentage of active oral/nonoral CTCs, percentage of correct consonants (PCC) in sentence imitation, word imitation, and word naming, percentage of stimulable consonants, hypernasality and nasal air emission were evaluated using the cleft audit protocol for speech-augmented protocol. Visual analysis, the two-standard deviation (2SD) test, and effect size indices, percentage of nonoverlapping data (PND) and Improvement Rate Difference (IRD), were used to assess outcomes.ResultsAll participants demonstrated reductions in active CTCs, hypernasality, and nasal air emission, alongside increases in PCC and stimulable consonants. These gains were maintained during the follow-up. PND and IRD indices reached 100% for all participants across all variables, except for hypernasality and nasal air emission. Hypernasality showed moderate treatment effects (PND/IRD = 40-60%), while the PND and IRD values for nasal air emission were 80%. The 2SD test confirmed statistically significant improvements (α = 0.05) in interval-level variables such as PCC and active CTCs, supporting the robustness of treatment effects.ConclusionIntensive hybrid telepractice and in-person therapy can improve multiple speech characteristics in Persian-speaking children with CP ± L, supporting the value of integrated, accessible treatment approaches for this population.

ObjectiveTo evaluate the prevalence, access to care, and health outcomes of orofacial clefts (OFCs) among American Indian and Alaska Native (AI/AN) populations through a systematic review and meta-analysis.DesignSystematic review and meta-analysis performed in accordance with PRISMA 2020 guidelines and registered with PROSPERO (CRD420251035364).SettingUS-based population registries, hospital databases, and institutional or community-level retrospective studies involving AI/AN populations.Patients and ParticipantsAI/AN individuals with OFCs compared with non-Hispanic White patients.InterventionsPrimary cleft lip and palate repair, secondary cleft-related procedures, and multidisciplinary cleft care.Main Outcome Measure(s)Prevalence of OFCs, timing of cleft surgery, discharge disposition, access to specialists, and qualitative determinants of disparities.ResultsEighteen studies including more than 1985 AI/AN patients were identified. Meta-analysis of 5 studies estimated a pooled OFC prevalence of 15 per 10 000 live births (95% confidence interval: 5-49), with substantial heterogeneity (I² = 99.8%). Individual studies reported significantly higher OFC prevalence in AI/AN populations compared to non-Hispanic Whites (odds ratio range: 1.44-2.68). Geographic maldistribution of craniofacial-trained surgeons, increased odds of nonhome discharge, and delayed cleft palate repair were consistently observed barriers. Qualitative analyses highlighted structural inequities, perceived racism, and lack of culturally responsive care as major contributors to disparities.ConclusionsAI/AN populations face a disproportionately high burden of OFCs alongside structural barriers to timely, culturally competent care. Addressing these disparities requires community-engaged, multidisciplinary interventions that improve geographic access and integrate culturally responsive approaches to care.

ObjectiveNo studies have examined the effectiveness of platelet-rich plasma (PRP) alone and platelet-rich fibrin (PRF) without alveolar bone grafting or other bone substitutes for alveolar cleft repair during infancy. This study evaluated the effectiveness of autologous PRP/PRF on alveolar regeneration in infantile unilateral cleft lip and alveolus.DesignRetrospective study.SettingPatients, Participants: A total of 56 patients with alveolar clefts were classified into the no PRP/PRF (which received gingivoperiosteoplasty [GPP] without PRP/PRF), PRP (which received GPP with PRP), and PRF (which received GPP with PRF) groups.InterventionsRegarding preparation, 5 mL of blood was withdrawn, and 1 mL each of PRP and PRF was prepared via centrifugal separation. PRP was implanted in the alveolar cleft after loading it into a commercially available gelatin sponge, while PRF was implanted directly.Main Outcome MeasuresPostoperative bone formation was assessed using computed tomography and radiography for qualitative analysis.ResultsThe new alveolar bone volume was higher in the PRF group than in the other groups; however, no differences were observed between the PRP, PRF, and control groups.ConclusionThis study was performed without complication. However, no significant difference in bone formation was found between the PRP, PRF, and control groups.

ObjectiveInvestigate the pre- and postoperative changes in patients with submucous cleft palate (SMCP) treated by intravelar veloplasty (IVV) with a buccinator musculomucosal flap (BMMF).DesignRetrospective case series.SettingTertiary, cleft team.Patients/ParticipantsChildren with SMCP who underwent IVV with a BMMF at Nagano Children's Hospital.Interventions: IVV with a BMMF between December 2018 and January 2022.Main outcome measuresPerceptual Japanese speech evaluation (hypernasality [HN], nasal emission, and intelligibility [IN]), amount of velopharyngeal (VP) gap, degree of soft palate elevation, amount of soft palate lengthening, VP closure pattern, additional surgery, and postoperative complications.ResultsEleven patients (four syndromic) were analyzed pre- and almost of 1 year postoperatively. Preoperative mean HN and IN scores were significantly improved (P = .0232 and P = .0313, respectively). VP gap was closed after surgery in most cases with initial VP gap <4.5 mm and good soft palate elevation. Mean soft palate lengthening with the BMMF method was 3.9 ± 1.2 mm, which was a significant increase (P = .00589). Four patients required additional surgery. We encountered no severe postoperative complications or fistula formation.ConclusionsIVV with a BMMF is considered applicable in patients with VP gap of up to 4.0 to 4.5 mm and good soft palate elevation. Our technique appears safe, with low risks of severe postoperative complications and fistula formation. Due to the limited cohort size, more cases are needed to elucidate the precise indications for the BMMF method.

ObjectiveThe goal of this study is to provide an overview of the current clinical practices of cleft teams affiliated to the Dutch Association for Cleft Palate (CP) and Craniofacial anomalies in the Netherlands and in Belgium, with regards to the placement of ventilation tubes in young children with CP.DesignCross-sectional survey.SettingMulticenter study, Oral Cleft Referral Centers of the Netherlands and Belgium.Patients, ParticipantsEar-nose-throat (ENT) surgeons, plastic surgeons, and language speech pathologists.InterventionsOnline survey.Main Outcome Measure(s)The survey questions covered the following topics: audiology assessment before palatoplasty, ventilation tube insertion (VTI) timing, postoperative follow-up visits.ResultsResponse rate per center was 100% (11/11 cleft centers), for a total of 21 cleft specialists (44%). Most centers (n = 7/11) establish VTI indication before palatoplasty by means of audiology assessment combined with ENT surgeon examination. Most centers combine VTI with palatoplasty (n = 9/11), and schedule a routine follow-up 2 to 3 months after surgery (n = 7/11).ConclusionsAlthough no standardized national or international protocols exist for the diagnosis and treatment of otitis media with effusion in children with CP, most Dutch and both Belgian cleft centers have independently developed local protocols that are largely aligned. Formalizing these into a unified written protocol represents an important next step toward optimizing and standardizing care for this patient population.

ObjectiveTo assess the impact of neighborhood socioeconomic deprivation on the timing of cleft surgery and outcomes, including postoperative palatal fistula and velopharyngeal insufficiency (VPI).DesignRetrospective cohort study.SettingSingle pediatric tertiary care center.Patients, ParticipantsA total of 702 patients who underwent cleft repair between 1995 and 2024, categorized by Area Deprivation Index (ADI) into low, medium, and high socioeconomic deprivation.InterventionsPrimary cleft lip (CL) and cleft palate (CP) repair, palatal fistula repair, and VPI surgery.Main Outcome Measure(s)Age at CL and CP repair, surgical delay (CL repair >6 months, CP repair >12 months), palatal fistula rate, time to fistula repair, and VPI surgery incidence.ResultsPatients in high-ADI areas experienced later CL (P < 0.05) and CP repairs (low ADI: 1.1 years; medium: 1.7 years; high: 1.8 years; P < 0.05). Logistic regression identified high ADI and female sex as independent predictors of CL delay (P < 0.001), while public insurance, race, greater hospital distance, and prior CL delay contributed to CP repair delays (P < 0.001). Palatal fistula rates (14.9%) were not linked to ADI, but high-ADI patients had longer times to repair (P < 0.05). Patients undergoing VPI surgery lived closer to the hospital than those not requiring repair (P < 0.05).ConclusionsHigher ADI and female sex were associated with CL repair delays, while CP repair delays correlated with public insurance, race, hospital distance, and prior CL delay. Targeted interventions are needed to improve timely cleft care.

ObjectiveExamine the percent consonants correct (PCC) produced by 3- and 4-year-old children with and without cleft palate using the American English Phrase Sample (AEPS) from the Cleft Audit Protocol for Speech-Augmented-Americleft Modification (CAPS-A-AM) protocol.DesignProspective, cross-sectional study.SettingPublic research university.ParticipantsSixty preschool age children were included: 30 with cleft palate and 30 without cleft palate. Each group was comprised of 15 children aged 3;0 to 3;11 (years; months) and 15 children aged 4;0 to 4;11.InterventionsImitation of the AEPS.Main Outcome MeasuresTotal PCC and PCC by manner of production were calculated from the AEPS. Group differences were analyzed using the Mann-Whitney U test; data are reported as median and interquartle range (IQR).ResultsThe median total PCC for 3-year-olds with cleft palate was 38% (IQR = 24-58), which was significantly lower (p = 0.0001) than the 3-year-olds without a cleft (70%; IQR = 60-77). Similarly, 4-year-olds with cleft palate produced a median PCC of 40% (IQR = 9-67), which was significantly lower (p < 0.0001) than 4-year-olds without a cleft (86%; IQR = 84-96). Only 20% (n = 3) of 3-year-olds and 7% (n = 1) of 4-year-olds with cleft palate produced a total PCC within of the IQR of their noncleft peers' total PCC. Across both age groups, children with cleft palate produced stops, fricatives, affricates, liquids, and consonant clusters with significantly lower accuracy than their same-age peers.ConclusionsPCC scores derived from transcription of the AEPS may offer speech language pathologists a practical way to assess consonant accuracy using an imitation stimulus already integrated into clinical protocols.