Cleft Palate-Craniofacial Journal最新文献

{"title":"Histological and Molecular Manifestations of Cleft Myopathy.","authors":"Yixuan Huang, Pinting He, Jinggui Li, Jinfeng Dou, Bing Shi, Jingtao Li, Qian Zheng, Xu Cheng","doi":"10.1177/10556656251313602","DOIUrl":"https://doi.org/10.1177/10556656251313602","url":null,"abstract":"<p><strong>Objective: </strong>Apart from rupture and displacement of muscle fibers, structural defects exist in cleft muscles but have not been adequately investigated. This study aimed to examine the histological and molecular features of the cleft muscles.</p><p><strong>Design: </strong>Orbicularis oris (OO) and tensor fasciae latae (TFL) muscle samples were obtained from patients with cleft lip and alveolar. The non-cleft OO muscles were obtained from patients with facial trauma. Myofiber histology, stem cell composition, and molecular signatures were compared among the cleft OO muscle, non-cleft OO muscle, and TFL muscle.</p><p><strong>Main outcome measures: </strong>Histological analysis of the fibrotic area, myofiber size, fiber type composition, and dystrophin expression pattern was performed to characterize the pathological manifestations in cleft muscles. Immunofluorescent staining of Pax7⁺ muscle satellite cells (MuSCs) and PDGFRα⁺ fibro-adipogenic progenitors (FAPs) and transcriptional profiling of MuSCs were carried out to explore the stem cell number and behavior in the cleft muscle.</p><p><strong>Results: </strong>Cleft muscles had an increased fibrotic area, variation in fiber size, and proportion of human fast myofiber. The defect in dystrophin expression was considerable in the non-cleft OO muscle and was even higher in the cleft OO muscle. MuSCs from cleft muscles showed a trend of increased Dux4 signature gene expression and repressed Pax7 target gene expression.</p><p><strong>Conclusion: </strong>Cleft myopathy resembles facial muscle-specific muscular dystrophy. The characterization of structural deformity inherited in cleft muscles could pave the way for a deeper understanding of orofacial cleft pathology.</p>","PeriodicalId":49220,"journal":{"name":"Cleft Palate-Craniofacial Journal","volume":" ","pages":"10556656251313602"},"PeriodicalIF":1.1,"publicationDate":"2025-01-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143014743","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Exparel and Outpatient Alveolar Bone Grafting: A Feasibility Study.","authors":"John N Muller, Lucas R Perez Rivera, David A Staffenberg, Roberto L Flores","doi":"10.1177/10556656251313848","DOIUrl":"https://doi.org/10.1177/10556656251313848","url":null,"abstract":"<p><strong>Objectives: </strong>To examine the feasibility of outpatient alveolar bone grafting (ABG) using Exparel (bupivacaine liposome injectable suspension) for donor site analgesia.</p><p><strong>Design: </strong>Retrospective, observational study.</p><p><strong>Setting: </strong>Single institution, 39-month retrospective review.</p><p><strong>Patients/participants: </strong>In total, 21 patients with cleft alveolus were included in this study.</p><p><strong>Intervention: </strong>In autologous outpatient ABG procedures using a mixed open harvest and Acumed trephine method to retrieve corticocancellous bone from the iliac crest, Exparel was injected in the donor site fascia.</p><p><strong>Main outcome measurements: </strong>Postoperative data including length of stay, post anesthesia care unit narcotics delivered, and maximum pain scores were recorded. Successful outcomes were assessed by exploring 30-day complications, 30-day emergency department visits, 30-day readmissions, and ambulation at follow-up.</p><p><strong>Results: </strong>The average hospital length of stay was 360.8 min. Four patients required overnight stay secondary to pain. The average doses of pain medications administered postoperatively were 198.4 mg acetaminophen, 9.3 mg ketorolac, 3.0 µg fentanyl, and 1.5 mg oxycodone. No patient reported 30-day complications secondary to donor site pain, 30-day emergency department visits, or 30-day readmissions, and there was only one case of wound dehiscence. All patients exhibited proper ambulation at follow-up and healed well from ABG.</p><p><strong>Conclusions: </strong>The use of Exparel for donor site analgesia in outpatient ABG procedures using corticocancellous grafts can result in adequate pain management and ambulation without an increased risk of complications and readmissions.</p>","PeriodicalId":49220,"journal":{"name":"Cleft Palate-Craniofacial Journal","volume":" ","pages":"10556656251313848"},"PeriodicalIF":1.1,"publicationDate":"2025-01-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143014723","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Exploring the Needs of Young People Born With Cleft Lip and/or Palate Approaching End of Routine Care, in the UK.","authors":"Julie Davies, Elaine Davidson, Sam Harding, Yvonne Wren, Lucy Southby","doi":"10.1177/10556656241312494","DOIUrl":"https://doi.org/10.1177/10556656241312494","url":null,"abstract":"<p><strong>Objective: </strong>The aim of this study was to understand the needs of young people, aged 16 to 20 years, born with cleft lip and/or palate (CL/P) approaching end of routine care, in the United Kingdom (UK).</p><p><strong>Design: </strong>Nominal group technique was used during 2 online focus groups to identify priorities for cleft care at transition to adult care. Focus group discussions were recorded, transcribed, and analyzed using reflexive thematic analysis.</p><p><strong>Participants: </strong>Ten young people born with CL/P, 8 aged 18 to 20 years and 2 aged 16 to 17 years were recruited via the UK-based cleft charity, the Cleft Lip and Palate Association (CLAPA).</p><p><strong>Results: </strong>Three themes were identified from the analysis: Theme 1-Psychological well-being is not a constant. Theme 2-Asking and listening-the cleft clinic appointment. Theme 3-Approaching end of routine care and re-accessing cleft services.</p><p><strong>Conclusions: </strong>Findings suggest that young people have ongoing needs in older adolescence. Psychological well-being, taking control of their own treatment and re-accessing the cleft team were highlighted as particular issues. Whilst further research with a larger and more representative sample is needed, these results support the need for continued access to and provision from the cleft team at this age.</p>","PeriodicalId":49220,"journal":{"name":"Cleft Palate-Craniofacial Journal","volume":" ","pages":"10556656241312494"},"PeriodicalIF":1.1,"publicationDate":"2025-01-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143014688","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Influence of Social Media in Multidisciplinary Cleft Care in Indonesia.","authors":"Kristaninta Bangun, Vika Tania, Prasetyanugraheni Kreshanti, Jessica Halim","doi":"10.1177/10556656241311739","DOIUrl":"https://doi.org/10.1177/10556656241311739","url":null,"abstract":"<p><p>Social media has played a pivotal role in relaying healthcare-related information, especially in efforts to improve cleft management. Cleft management involves a multidisciplinary team throughout the years of comprehensive intervention, from birth to adulthood.</p><p><p>To investigate the behaviors of parents and patients with clefts regarding social media use in improving cleft-related outcomes, knowledge, and awareness.</p><p><p>This questionnaire-based descriptive study was conducted online and involved both closed- and short-ended questions.</p><p><p>Parents and patients with cleft lip and palate provided consent to participate in the study.</p><p><p>Patient demographics, history, and timing of cleft treatment, barriers to care, and social media use were explored.</p><p><p>Fifty-six participants were enrolled in this study. The mean ages of the mothers and fathers of children with cleft were 34 (range, 24-57) and 36 (range, 26-59) years, respectively, and more than one-half were university graduates. <i>WhatsApp</i>, <i>Instagram</i>, and <i>YouTube</i> were used by 100%, 89%, and 89% of parents, respectively, 82% of whom were part of a cleft community group. Seventy-two percent underwent labioplasty, and 75% underwent palatoplasty at the ideal time. Parents exhibited low perceived barriers to care, with 33% and 37% reporting limitations on knowledge and accommodation, respectively.</p><p><p>Social media is the key to bridging the gap between the large number of cases and facility limitations and specialized human resources. Cleft centers should integrate combined platforms (<i>WhatsApp, YouTube, and Instagram</i>) for marketing and education purposes. Social media and community groups facilitate sharing, engagement, and empowerment of patients and caregivers to improve cleft-related outcomes.</p>","PeriodicalId":49220,"journal":{"name":"Cleft Palate-Craniofacial Journal","volume":" ","pages":"10556656241311739"},"PeriodicalIF":1.1,"publicationDate":"2025-01-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143014809","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Familial Oculoauriculovertebral Spectrum: A Genomic Investigation of Autosomal Dominant Inheritance.","authors":"Aline L Petrin, Ligiane Alves Machado-Paula, Austin Hinkle, Luke Hovey, Waheed Awotoye, Michael Chimenti, Benjamin Darbro, Lucilene A Ribeiro-Bicudo, Shareef M Dabdoub, Tabitha Peter, Patrick Breheny, Jeffrey C Murray, Eric Van Otterloo, Shankar Rengasamy Venugopalan, Lina M Moreno-Uribe","doi":"10.1177/10556656241306202","DOIUrl":"10.1177/10556656241306202","url":null,"abstract":"<p><strong>Objective: </strong>Oculoauriculovertebral spectrum (OAVS) encompasses abnormalities on derivatives from the first and second pharyngeal arches including macrostomia, hemifacial microsomia, micrognathia, preauricular tags, ocular, and vertebral anomalies. We present genetic findings on a 3-generation family affected with macrostomia, preauricular tags and ptosis following an autosomal dominant pattern.</p><p><strong>Design: </strong>We generated whole-genome sequencing data for the proband, affected father, and unaffected paternal grandmother followed by Sanger sequencing on 23 family members for the top candidate gene mutations. We performed parent and sibling-based transmission disequilibrium tests (TDTs) and burden analysis via a penalized linear mixed model, for segregation and mutation burden, respectively. Next, via bioinformatic tools we predicted protein function, mutation pathogenicity, and pathway enrichment to investigate the biological relevance of mutations identified.</p><p><strong>Results: </strong>Rare missense mutations in <i>SIX1</i>, <i>KDR/VEGFR2</i>, and <i>PDGFRA</i> showed the best segregation with the OAVS phenotypes in this family. When considering any of the 3 OAVS phenotypes as an outcome, <i>SIX1</i> had the strongest associations in parent-TDTs and sib-TDTs (<i>P</i> = 0.025, <i>P</i> = 0.052) (unadjusted <i>P</i>-values). Burden analysis identified <i>SIX1</i> (RC = 0.87) and <i>PDGFRA</i> (RC = 0.98) strongly associated with OAVS severity. Using phenotype-specific outcomes, sib-TDTs identified <i>SIX1</i> with uni- or bilateral ptosis (<i>P</i> = 0.049) and ear tags (<i>P</i> = 0.01), and <i>PDGFRA</i> and <i>KDR/VEGFR2</i> with ear tags (both <i>P</i> < 0.01).</p><p><strong>Conclusion: </strong><i>SIX1</i>, <i>PDGFRA</i>, and <i>KDR/VEGFR2</i> are strongly associated to OAVS phenotypes. <i>SIX1</i> has been previously associated with OAVS ear malformations and is co-expressed with <i>EYA1</i> during ear development. Efforts to strengthen the genotype-phenotype co-relation underlying the OAVS are key to discover etiology, family counseling, and prevention.</p>","PeriodicalId":49220,"journal":{"name":"Cleft Palate-Craniofacial Journal","volume":" ","pages":"10556656241306202"},"PeriodicalIF":1.1,"publicationDate":"2025-01-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143014741","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Evaluation of PCORnet as an Approach to Accessing Electronic Health Record (EHR) Data for Cleft Outcomes Research: Advantages and Limitations.","authors":"Elaine Lin, Victoria N Yi, Kristina Dunworth, Christopher Runyan, Alexander C Allori","doi":"10.1177/10556656241312747","DOIUrl":"https://doi.org/10.1177/10556656241312747","url":null,"abstract":"<p><p><b>Objective:</b> To evaluate the feasibility of using the National Patient-Centered Clinical Research Network (PCORnet^®) as a source of electronic health record (EHR) data for cleft outcomes research. <b>Design:</b> Exploratory retrospective analysis of multi-year, administrative and clinical, structured data stored in PCORnet. <b>Setting:</b> Academic institution with an ACPA-approved cleft and craniofacial team. <b>Patients/Participants:</b> Encounter-level data pertaining to patients with orofacial clefts treated at this center between 2010 and 2018. <b>Outcome Measures:</b> (1) Ability of PCORnet to report metrics such as the following: number of new and returning patients per year; demographics; phenotype; procedures; readmission or reoperation within 30 days; etc. (2) Accuracy of selected metrics, compared with manual chart review. <b>Results:</b> PCORnet is useful for the calculation of simple process metrics such as patient demographics, phenotype mix, case mix, and number of readmissions. However, as it lacks access to clinical notes, PCORnet alone cannot provide more detailed information. Phenotypic classification (based on ICD codes) and procedural description (based on CPT^®) are subject to inaccuracy. A 1-2 year delay in data upload to PCORnet may be rate-limiting for certain applications. Multi-institutional queries were feasible. <b>Conclusions:</b> PCORnet does not include all necessary data elements from the EHR. While very convenient for the tabulation of simple process metrics, especially from multiple institutions, supplemental data collection will be required for meaningful cleft outcomes research. Cleft teams whose institutions participate in PCORnet might choose to store the supplemental data as \"sidecars\" alongside the standard PCORnet database tables, which would allow for future PCORnet queries to be more informative and impactful.</p>","PeriodicalId":49220,"journal":{"name":"Cleft Palate-Craniofacial Journal","volume":" ","pages":"10556656241312747"},"PeriodicalIF":1.1,"publicationDate":"2025-01-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143014719","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"How Much Pain Medication Do Patients Require After Posterior Iliac Crest Bone Graft to the Alveolar Cleft?","authors":"Andrew J Deek, Snigdha Jindal, Kate Bridges, Alistair Varidel, Bonnie L Padwa, Cory M Resnick","doi":"10.1177/10556656241309157","DOIUrl":"https://doi.org/10.1177/10556656241309157","url":null,"abstract":"<p><strong>Objective: </strong>The purpose of this study was to quantify analgesic use following alveolar cleft bone grafting (ABG) utilizing a posterior iliac crest (PIC) donor site.</p><p><strong>Design: </strong>This is a prospective cohort study of consecutive patients that underwent ABG with PIC in a 10 month period from November 2022 to September 2023.</p><p><strong>Setting: </strong>Tertiary care free-standing pediatric hospital.</p><p><strong>Participants: </strong>Patients with cleft lip and alveolus with or without cleft of the secondary palate undergoing alveolar bone graft with cancellous marrow from the PIC.</p><p><strong>Interventions: </strong>All patients underwent ABG and recorded analgesic use afterwards.</p><p><strong>Main outcome measure: </strong>The main outcome variable was total pain medication utilization during the first 10 postoperative days (PODs).</p><p><strong>Results: </strong>22 subjects (72.7% male, 54.5% Caucasian 72.7% unilateral and 27.3% bilateral clefts) recorded pain medication use. Total duration of analgesic use (any type) was 3.86 ± 1.7 postoperative days. 10 subjects (45.5%) did not use opioid medication postoperatively. The remaining 12 subjects (54.5%) used a median of 12 (0, 19) morphine milli-equivalents (MME), equal to 1.6 (0, 2.53) doses of 5 mg oxycodone. An average of 9.9 ± 5.6 doses of acetaminophen and 9.1 ± 4.7 doses of a non-steroidal anti-inflammatory drug (NSAID) were administered. No subject used opioids after POD3. Patients with unilateral clefts used more acetaminophen (<i>p</i> = 0.02); otherwise, there were no significant differences in outcomes based on any predictor variable (<i>p</i> > 0.05).</p><p><strong>Conclusions: </strong>Analgesic requirements after ABG with PIC were minimal, with nearly half of patients taking no postoperative opioids. As such, judicious opioid prescribing is prudent.  .</p>","PeriodicalId":49220,"journal":{"name":"Cleft Palate-Craniofacial Journal","volume":" ","pages":"10556656241309157"},"PeriodicalIF":1.1,"publicationDate":"2025-01-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142972977","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Benefit Finding in Craniofacial Conditions: A Qualitative Analysis of Patient and Parent Perspectives.","authors":"Alyssa K Choi, Megan Korhummel, Caitlyn C Belza, Josseline Herrera Eguizabal, Sydney Olfus, Chelsea S Rapoport, Jessica Blum, Viridiana J Tapia, Julia H Drizin, Amanda Gosman, Vanessa L Malcarne","doi":"10.1177/10556656241305889","DOIUrl":"https://doi.org/10.1177/10556656241305889","url":null,"abstract":"<p><strong>Objective: </strong>Craniofacial conditions (CFCs) can be associated with adverse effects on quality of life (QoL). However, few studies have examined perceived benefits related to CFCs. This study described perceived benefits in an international sample of children and adolescents with CFCs and their parents.</p><p><strong>Design: </strong>Semistructured qualitative interviews were completed in English or Spanish as part of a larger study. Deductive content analysis described and quantified perceived benefits associated with CFCs.</p><p><strong>Setting: </strong>Interviews were during standard visits at Rady Children's Hospital-San Diego in the USA and the Hospital Infantil de las Californias in Tijuana, Baja California, Mexico.</p><p><strong>Participants: </strong>Patients were ages 7 to 20 years (<i>n</i> = 32) with CFCs (cleft lip and/or palate, craniosynostosis, microtia, hemifacial microsomia, dermatologic conditions/neurovascular malformations, and trauma-acquired CFC), and parents (<i>n</i> = 71) had children ages 5 months to 23 years with CFCs. Of the total sample, there were 14 patient-parent dyads.</p><p><strong>Results: </strong>A total of 230 benefits were identified. Of the patients (47%) and parents (73%) who identified at least 1 benefit, themes included personal growth (40%), understanding or helping others facing challenges (25%), social relationships (23%), spiritual or religious beliefs (4%), philanthropy (4%), material or external gains (3%), and personal health (1%).</p><p><strong>Conclusions: </strong>Patients with CFCs and their parents report multiple positive effects of CFCs on their QoL. These findings indicate that benefit finding is a common experience in this population and may be leveraged by clinicians to help promote positive adjustment to living with a CFC.</p>","PeriodicalId":49220,"journal":{"name":"Cleft Palate-Craniofacial Journal","volume":" ","pages":"10556656241305889"},"PeriodicalIF":1.1,"publicationDate":"2025-01-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142957580","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Design, Development, and Evaluation of a 3D-Printed Buccal Myomucosal Flap Simulator.","authors":"Mitchell D Cin, Zahra Nourmohammadi, Usama Hamdan, David A Zopf","doi":"10.1177/10556656241311044","DOIUrl":"https://doi.org/10.1177/10556656241311044","url":null,"abstract":"<p><strong>Objective: </strong>Buccal myomucosal flap procedures have become a critical tool in the armamentarium of the cleft surgeon. Mastering this technique is complex and providing sufficient training opportunities presents significant challenges. Our study details the design, development, and evaluation of a low-cost, high-fidelity buccal myomucosal flap surgical simulator. Our goal is to establish a reliable teaching tool for early learners, validated through craniofacial surgeon assessment.</p><p><strong>Design: </strong>The simulator comprises of an anatomical model and a stand created using computer-aided design software. Hard tissues were 3D-printed, while soft tissues were cast in silicone. The model underwent review by craniofacial surgeons utilizing a 1 to 5 Likert scale across six evaluation domains.</p><p><strong>Setting: </strong>In-person simulated dissection session.</p><p><strong>Patients/participants: </strong>Sixteen craniofacial surgery providers from various subspecialties.</p><p><strong>Interventions: </strong>None.</p><p><strong>Main outcome measure: </strong>Anonymized survey responses.</p><p><strong>Results: </strong>The simulator received fair to high scores across all evaluation domains, notably 4.31 as a training tool, 3.77 as a competency evaluation tool, 3.92 as a rehearsal tool, and 3.93 in relevance to practice.</p><p><strong>Conclusions: </strong>The validated buccal myomucosal flap simulator theoretically enables the acquisition of surgical skills in a zero-risk simulated environment. Plans involve integration into a structured curriculum with diverse participants. Continued iteration and adoption hold the promise of significantly enhancing access to training for competency of cleft and craniofacial procedures.</p>","PeriodicalId":49220,"journal":{"name":"Cleft Palate-Craniofacial Journal","volume":" ","pages":"10556656241311044"},"PeriodicalIF":1.1,"publicationDate":"2025-01-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142957582","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Mandibular Distraction Osteogenesis vs. Tracheostomy in the Management of Pierre Robin Sequence: A Systematic Review and Meta-Analysis.","authors":"Indri Lakhsmi Putri, Kusuma Islami, Imaniar Fitri Aisyah, Rachmaniar Pramanasari, Citrawati Dyah Kencono Wungu","doi":"10.1177/10556656241311549","DOIUrl":"https://doi.org/10.1177/10556656241311549","url":null,"abstract":"<p><strong>Objective: </strong>This study compares mandibular distraction osteogenesis (MDO) and tracheostomy in managing severe airway obstruction in patients with the Pierre Robin sequence (PRS).</p><p><strong>Design: </strong>A systematic review and meta-analysis following PRISMA guidelines was performed. Literature searches were conducted across PubMed, ScienceDirect, Cochrane Library, Scopus, E.B.S.C.O., and Web of Science, including grey literature, covering studies until May 30, 2024. Study quality was assessed using the Newcastle-Ottawa Scale.</p><p><strong>Patients: </strong>patients with Pierre Robin Sequence.</p><p><strong>Interventions: </strong>Mandibular distraction osteogenesis (MDO) and tracheostomy.</p><p><strong>Main outcome measures: </strong>Primary outcomes included airway management (tracheostomy avoidance for MDO, decannulation for tracheostomy) and feeding outcomes (G-tube placement). Secondary outcomes were hospital length of stay and associated costs.</p><p><strong>Results: </strong>Thirteen studies were included. MDO and the MDO-first approach demonstrated significantly better airway outcomes (OR = 10.72, 95% CI = 1.97-58.44, p = 0.006; OR = 4.51, 95% CI = 2.61-7.79, p < 0.00001). MDO also reduced the need for G-tube placement (OR = 0.09, 95% CI = 0.04-0.18, p < 0.00001) and lowered hospital costs (MD = -47.90 thousand USD, 95% CI = -59.93 to -35.87, p < 0.0001). A shorter hospital stay was observed but was not statistically significant.</p><p><strong>Conclusions: </strong>MDO offers better airway outcomes, lower G-tube placement rates, and reduced costs, making it a preferred option. Larger studies within the same syndromic status are needed to minimize confounding factors and validate these findings.</p>","PeriodicalId":49220,"journal":{"name":"Cleft Palate-Craniofacial Journal","volume":" ","pages":"10556656241311549"},"PeriodicalIF":1.1,"publicationDate":"2025-01-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142957604","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}