Cleft Palate-Craniofacial Journal最新文献

ObjectiveThis study synthesized and described the prevalence and incidence of cleft lip and/or palate (CL/P) reported by different studies in Europe.DesignA scoping review was designed according to the Joanna Briggs Institute methodology and an advanced search was conducted using MedLine, Science Direct, and Semantic Scholar without time or language restrictions. Two reviewers extracted data from the articles included.ParticipantsThe extracted data included specific details on the type of orofacial cleft, the European regions (Northern Europe, Western Europe, Eastern Europe, and Southern Europe).Main Outcome Measure(s)A meta-analysis was conducted with the data reported for the incidence and prevalence of CL/P.ResultsA total of 66 studies were obtained reporting from 29 European countries. According to what was obtained from the meta-analysis, a prevalence of cleft lip and palate (CLP) of 0.7/1000 95% CI: (0.6-0.7) births is reported in Europe, followed by cleft palate (CP) and cleft lip (CL) with 0.5/1000 95% CI: (0.4-0.5) and 0.3/1000 95% CI: (0.3-0.4), respectively. A total prevalence of CL/P in Europe is estimated at 1.1/1000 95% CI: (1.0-1.2) between 1950 and 2014. The incidence of CLP is reported to be 0.7/1000 95% CI: (0.6 to 0.8) followed by CP and CL with 0.5/1000 95% CI: (0.5-0.6) and 0.4/1000 95% CI: (0.3-0.4), respectively. The total incidence of CL/P in Europe is estimated at 1.6/1000 95% CI: (1.5-1.7) between 1948 and 2018.ConclusionThis review provides a description of the status of the prevalence and incidence of CL/P in Europe and its regions, and can be considered as a guide for the development of promotion and prevention programs.

ObjectiveSpiritual distress describes an "impaired ability to experience and integrate meaning and purpose," frequently occurring after receipt of a major diagnosis. We measured spiritual distress in caregivers of children presenting to a multidisciplinary cleft and craniofacial clinic, hypothesizing that a significant percentage of caregivers would demonstrate spiritual distress.DesignCaregivers were surveyed utilizing the Functional Assessment of Chronic Illness Therapy-Spiritual Well-Being scale, non-illness version (FACIT-Sp 12), the eight-item Patient Health Questionnaire depression scale (PHQ-8) and the seven-item General Anxiety Disorder scale (GAD-7), as well as questions evaluating caregiver health, financial strain, and religiosity measured by the Duke Religion Index (DUREL). Logistic regression with cluster adjustment was used to control for demographic and clinical variables.SettingA multidisciplinary cleft and craniofacial clinic.Patients, ParticipantsAll caregivers able to read English were surveyed. 149 of 191 completed the survey (response rate = 78.0%).Main Outcome Measure(s)Caregiver spiritual distress, as measured by the FACIT-Sp 12, non-illness version.ResultsThe median age of caregivers was 33 (interquartile range 25-39). Most were female (76.7%), white (86.7%), non-Latino (91.1%), and Christian (76.7%). Most patients were seen for a cleft diagnosis (90.8%). Thirty percent of caregivers demonstrated spiritual distress. Depression was present in 16.1% and moderate to severe anxiety in 18.1%. On adjusted analysis, Christianity was associated with lower odds of spiritual distress (OR 0.32, p = .039). Additionally, both depression (OR 14.36, p = .001) and anxiety (OR 3.81, p = .006) were associated with higher odds of spiritual distress.ConclusionsNearly one-third of caregivers demonstrated spiritual distress, which was associated with mood disorders. Addressing spiritual stress and providing necessary resources is critical for effective team-based care.

ObjectiveTo compare the use of porous polyethylene implants versus autologous reconstruction for ear reconstruction in patients with microtia or anotia.DesignRetrospective cohort study.SettingHospitals included in the American College of Surgeons National Surgical Quality Improvement Project-Pediatrics, between 2016 and 2022.PatientsChildren with diagnoses of microtia or anotia.InterventionsEar reconstruction utilizing either a porous-polyethylene implant or autologous reconstruction.Main Outcome MeasuresRates of wound complication, hospital readmission, reoperation, and hospital lengths-of-stay within 30 days of index surgery.ResultsThere were 986 patients meeting inclusion criteria, including 893 receiving autograft and 93 receiving synthetic implant. Complication rates, including wound complication, dehiscence, and surgical site infection are similar between the 2 cohorts, though deep surgical site infections are more frequent in patients receiving implant reconstruction.Patients receiving implant reconstruction have a 4-fold higher odds of hospital readmission and a 9-fold higher odds of reoperation within 30 days after controlling for patient characteristics and surgeon specialty. Patients receiving implant reconstruction also have shorter hospital lengths-of-stay.ConclusionsEar reconstruction using autologous cartilage provides better 30-day outcomes compared to implant-based reconstruction with respect to rates of reoperation and readmission. However, these differences may be reflective more of limited surgeon experience with using synthetic implants than of the best possible outcomes achievable with each technique.

ObjectiveTo assess the efficacy of Khon Kaen University presurgical nasoalveolar molding (KKU PNAM) by evaluating nasolabial morphological changes in patients with complete unilateral cleft lip and palate (UCLP) using three-dimensional (3D) anthropometric measurements and comparing nasal morphology to age-matched noncleft controls.DesignA retrospective case series of infants with complete UCLP who underwent KKU PNAM therapy. Patients with complete 3D stereophotogrammetric records at initial (T₀) and pre-cheiloplasty (T₁) visits were included.SettingKhon Kaen University Cleft Lip and Palate CenterPatients/ParticipantsSixteen infants with complete UCLPInterventionsKKU PNAMMain Outcome MeasuresAnthropometric measurements of nasolabial tissue of infants with complete UCLP were compared between pre- (T₀) and post-PNAM treatment (T₁) and against age-matched noncleft controls at both time points.ResultsFrom T₀ to T₁, cleft-sided nostril height and columellar length, height, and angle increased significantly (0.85 mm, 0.83 mm, 1.54 mm, and 17.38 degrees, respectively). Conversely, cleft-sided nostril width, deviated alar base and columella, and vermilion gap significantly decreased (1.57, 1.04, 0.68, and 4.19 mm, respectively). The difference of nostril width and height between cleft and noncleft side was decreased. At T₀, alar base, subnasal, and nostril width, alongside the nostril and columellar height, differed from those of control infants. At T₁, the columellar height in the treatment cohort was comparable to that of the control. Other parameters remained significantly different; however, these differences decreased post treatment.ConclusionsKKU PNAM therapy improved nasolabial morphology and symmetry in patients with UCLP, suggesting its potential to facilitate successful future cheiloplasty.

ObjectiveTo evaluate the readability of online patient education materials (PEMs) for cleft lip and/or palate and assess their alignment with recommended readability levels.DesignThis study is a systematic review and meta-analysis.SettingLiterature search conducted in PubMed, Scopus, and Embase databases following PRISMA guidelines.MaterialsStudies evaluating online PEMs for cleft care with reported readability metrics, including Flesch-Kincaid Grade Level, Flesch Reading Ease, SMOG Index, or Gunning Fog Index.InterventionsAssessment of readability metrics of online PEMs and evaluation of artificial intelligence tools (eg, ChatGPT) for text simplification.Main Outcome Measure(s)Pooled readability estimates (eg, Flesch-Kincaid Grade Level, Flesch Reading Ease, SMOG Index, Gunning Fog Index), heterogeneity (I²), and confidence intervals (CIs).ResultsNine studies were included, consistently showing that PEMs exceed readability recommendations. Pooled estimates revealed a Flesch-Kincaid Grade Level of 9.48 (95% CI: 8.51-10.45), Flesch Reading Ease score of 52.98 (95% CI: 42.62-63.34), SMOG Index of 9.27 (95% CI: 5.97-12.57), and Gunning Fog Index of 9.94 (95% CI: 8.90-10.98). Heterogeneity was minimal (I² = 0%). Artificial intelligence tools like ChatGPT demonstrated potential in simplifying text to the recommended sixth-grade reading level but lacked usability and comprehension testing.ConclusionsOnline PEMs for cleft care are consistently written at reading levels too complex for the average caregiver, underscoring the need for improved readability and accessibility. Future research should focus on developing multimodal resources, conducting usability assessments, and including non-English materials to address global disparities in cleft care education.

IntroductionChildren with clefts have been shown to have various nutrient deficiencies including protein energy malnutrition but the levels of vitamin D as compared to normal population have seldom been researched.Materials and MethodsSerum levels of Vitamin D in children with clefts (12 years and less), belonging to the Sub-Himalayan and Gangetic Plain regions were compared with age matched normal population using non-parametric tests. Bivariate Logistic regression for estimation of odds ratio for cleft versus control were performed for risk estimation of vitamin D insufficiency (less than 30 ng/mL).ResultsSerum Vitamin D levels of cleft group was significantly lower than control group significantly (P = 0.02) and the age adjusted odds ratio was 2.819 compared to controls.ConclusionThe study reveals Vitamin Deficiency in the general population of the sub-Himalayan region, albeit, the levels in the population of clefts are significantly less as compared to the control population. Hence it is imperative to consider prospective trials and if proven then to recommend alterations in the existing vitamin D supplementation for children in this age group belonging to this belt in general. The normalization of vitamin D levels in children with cleft can help in wound healing after surgeries in addition to their normal growth and development.

ObjectiveThis pilot study sought to determine whether adolescent and adult patients benefit from weekly cleft-specific speech language therapy (SLT) services, and to understand how concurrent psychology clinic services influence patient-reported outcomes.MethodsTwelve patients (12 y+) with cleft-related speech concerns were seen at the University of Canterbury Speech and Psychology clinics in 2023. Patients elected to receive SLT and psychology services in-person, online, or a combination of both. Patient-reported outcome measures of speech function, intelligibility and acceptability, speech distress, and psychological and social function were completed before therapy, midway, immediately post, and 3 months post their 4- to 8-week block of treatment. Quantitative data were analyzed using descriptive statistics. Patients and clinicians completed exit interviews to understand their clinic experience. Interviews were analyzed using conventional content analysis.ResultsParticipants reported improved speech function, intelligibility, and acceptability following the clinic. Participants also conveyed improvements in speech distress and in psychological and social functions, which peaked after receiving psychology services. Patient exit interviews suggested positive clinic experiences and that the intensity saw improvements realized in a short timeframe. Facilitators and barriers to the clinic's success were identified. Clinicians gained confidence working with patients with CL/P and valued the cross-discipline working opportunity.ConclusionsCleft-specific SLT services and routine psychological care should be available across the lifespan, and with regular frequency. Accessibility options enhanced engagement with the clinic. Regular multidisciplinary working between SLTs and psychologists facilitates clinical success, and university clinics can provide a valuable adjunct to hospital CL/P services. Ongoing clinical training opportunities such as collaborative clinics to gain experience working with CL/P are warranted.

ObjectiveTo synthesize research exploring the impact of surgery on quality of life (QoL) in families of children with a cleft lip and/or palate (CL/P).DesignMedline, PsycINFO, CINAHL, and ProQuest databases were systematically searched (from database inception to July 2024) to identify studies exploring QoL in families following surgery for a child with CL/P. Studies were quality appraised using Quality Assessment for Diverse Studies criteria, and a narrative synthesis was undertaken.Main Outcome Measure: Quality of life was explored but specific measures varied across studies.Interventions: Type of surgery being investigated varied across the reviewed studies. This systematic review focussed on both functional and aesthetic surgeries throughout childhood.ResultsOf 157 identified studies, 9 were eligible for inclusion (1 mixed-methods, 1 observational, 1 qualitative, 2 cross-sectional, 4 quasi-experimental). Analysis indicated that QoL was positively impacted by surgery through reduced financial burden, increased sense of mastery/coping, and reduced social impact. However, for some families, new demands (eg, medical/dental appliances, food restrictions), intolerance of uncertainty, and adjustment to changes to their child's appearance were identified following surgery.ConclusionsThere is limited qualitative and quantitative research exploring the impact of a child's cleft surgery on family QoL. The quality of studies included in the review was variable. Supporting families accessing cleft services, especially where QoL and mental health are negatively impacted, is important. Due to the variability within and between the studies, and lack of detail relating to rationale and processes taken, the conclusions from this review are tentative. Further research is needed that addresses methodological and conceptual limitations.

ObjectiveThe reported prevalence of cleft lip and palate within the state of Arkansas (10.63 to 12.22 per 10,000 live births) is much higher than the national average. With greater understanding of at-risk populations and risk factors, we can provide more targeted education to improve patient outcomes and potentially reduce the incidence of orofacial clefts.DesignThis is a database review of cleft lip and/or palate patients using data obtained from the Arkansas Reproductive Health Monitoring System (ARHMS) database, a statewide birth defects registry that collects data from 83 regional hospitals.SettingStatewide database study.PatientsPatients diagnosed with cleft lip and/or palate between the years 1993 and 2015 registered in the ARHMS database.Main Outcome MeasurePrevalence rates of orofacial clefts within each Arkansas county.ResultsA total of 1345 unique patients with a diagnosis of cleft lip and/or palate were identified in the database with an average prevalence of 14.9 per 10,000 live births [95% CI: 14.1-15.7]. Of the 75 counties in Arkansas, 37 counties had prevalence rates higher than the state rate. Three counties had particularly higher prevalence rates of more than 33 per 10,000 live births. On the other hand, 3 counties had very low rates of 5 or less per 10,000 live births.ConclusionsThe rates of orofacial cleft anomalies are not distributed as expected among Arkansas counties. Further exploration is necessary to determine what, if any, environmental factors are at play.

ObjectivesAssessment of simultaneous 3-dimensional facial and maxillary archform changes after primary lip repair in cUCLP.DesignRetrospective longitudinal case series.SettingsKing's College London, United Kingdom.PatientsConsecutive facial plaster cast sets of 18 infants with cUCLP, pre lip (3 months), and pre palate repair (6 months).InterventionsSingle Consultant Cleft Plastic Surgeon in single cleft area operated on all cases and created all casts once anesthetized. One operator scanned all casts, exporting .stl files for analysis.Main outcome measuresTwenty-six facial and maxillary arch landmarks were defined. From these, 11 variables describing linear and volumetric changes were calculated. Landmark identification precision was assessed through a repeatability study. Color maps for visualization of changes after lip repair were created by superimposition. Parametric 1- and 2- sample t tests were used to compare changes between lip and palate repairs. The study had sufficient power (0.80). Clinically significant changes were defined ≥2 mm for nose and ≥4 mm for cleft widths, and ≥5 mm3 for nose and ≥2 mm³ for maxillary archform volumes.ResultsGood precision of landmark identification was confirmed. The main study showed a statistically and clinically significant decrease in overall nasal width (3.24 mm, 95%CI [1.76, 4.71], P = .04) and decrease in average cleft width volume (4.62 mm³, 95% CI [2.58, 6.66], P < .01) after lip repair. Color maps showed concordance with numerical findings.ConclusionsAfter lip repair, there was simultaneous nose width reduction, better nasal symmetry, changes in overall palatal alveolar ridges volume, and decrease in volume of alveolar cleft and anterior ridge of palate.