Cleft Palate-Craniofacial Journal最新文献

In 2012, the James Lind Alliance (JLA) worked with individuals with cleft lip and/or palate (CL/P), their families and clinicians to identify priority areas for future research. This article reviews progress conducted in the United Kingdom in the 3 JLA priorities most closely related to Clinical Psychology. It then builds upon the original priorities to identify 4 future directions, based on contemporary literature and in-depth discussions between clinical and research experts. Finally, recommendations for next steps toward meeting these future directions are outlined, including consistent outcome measurement, engaging diverse groups of people with CL/P and embedding the principles of codesign.

Background: Paucity exists in the literature on uvular repair while documenting palatoplasty techniques for children with cleft palate.

Solution: We propose a modified approach without losing any soft tissue structures, gaining better cosmetic outcome, and possibly having functional gains postoperatively.

What we did that is new: The proposed technique provides better cosmesis for the structures postoperatively and enhances the palatoglossal arch, which may prevent the spillage of saliva to the oropharyngeal region.

Objective: The Melbourne technique for total cranial vault remodeling aims to address all aspects of scaphocephaly in sagittal craniosynostosis. These features include anterior-posterior excessive length, anteriorly displaced vertex position, frontal bossing, vertex narrowing, and occipital bulleting. This study aimed to determine the progressive cranial changes that occur following the Melbourne technique for sagittal craniosynostosis.

Design: Retrospective review of 3-dimensional images collected preoperatively and postoperatively at 3 weeks, 3 months, 1 year, and 2 years.

Setting: Tertiary care pediatric institution.

Participants: Twenty-five patients with sagittal craniosynostosis.

Interventions: The Melbourne technique for total cranial vault remodeling.

Main outcome measure(s): Head circumference, cephalic index, frontal bossing index, occipital bulleting index, vertex narrowing index, and vertex-nasion-opisthocranion (VNO) angle were evaluated.

Results: The cephalic index significantly increased postoperatively (P = .04) with a subsequent relapse at 3 months followed by progressively increased growth. The frontal bossing index significantly decreased postoperatively (P = .02) with a progressive decrease. The occipital bullet index had a relative decline postoperatively with relapse at 3 months, followed by a progressive decrease. The vertex narrowing index significantly decreased postoperatively (P < .001), with a plateau and slight relapse. The VNO angle showed a relative decline over time with a significant decrease by 1 year of age (P = .002).

Conclusions: The Melbourne technique improved the cephalic index, frontal bossing, vertex narrowing, occipital bulleting, and vertex positioning at 2 years of age. Cephalic index and occipital bulleting showed slight relapse at 3 months, followed by progressive improvement over time.

Objective: To investigate whether differences in early cleft care increase risk of velopharyngeal insufficiency (VPI) after maxillary advancement.

Design: Retrospective cohort study.

Setting: Large pediatric tertiary care hospital.

Patients/participants: Adolescents and young adults (AYAs) with cleft palate (∓cleft lip) who underwent maxillary advancement between 2008 and 2019.

Interventions/comparisons: Initial palate repair at a different institution (early care elsewhere, ECE) versus care at a single institution (consistent care, CC).

Main outcome measures: Post-maxillary advancement VPI.

Results: One-hundred seventy-eight AYAs underwent maxillary advancement, 74 in the ECE group and 104 in the CC group. The ECE group was more likely to be internationally adopted (34% versus 4%), to have a history of VPI surgery (54% versus 32%) and to be older at time of palate repair (mean 25 versus 16 months). Of anatomical measures, only velar length differed, with the ECE group tending to have a shorter velum (mean 26 mm versus 28 mm). Proportional odds regression revealed increased odds of post-operative VPI in the ECE group (OR 1.46, 95% CI 0.75-2.85) relative to the CC group. This relationship was stronger among those with bilateral cleft lip and palate (OR 3.29, 95% CI 0.86-13.52). For patients with history of prior VPI surgery, the odds of post-operative VPI in the ECE group was more than 3 times that in the CC group (OR 3.06, 95% CI 1.08-9.16).

Conclusions: VPI after maxillary advancement is more likely among individuals who received early cleft care elsewhere compared to those who underwent all cleft operations at a single center.

Objectives: This study compares perioperative outcomes between spring-assisted cranioplasty (SAC), distraction osteogenesis (DO) and conventional expansion in craniosynostosis surgery.

Design: Systematic review and meta-analysis.

Setting: Retrospective and prospective cohort.

Patients and participants: A comprehensive electronic search was conducted using PubMed/Medline, Scopus, Science Direct, EBSCO, Web of Science, and Cochrane Library, alongside Gray literature sources (SSRN, Scopus preprint, and MedRxiv). Publication bias was assessed and study quality was evaluated using the Newcastle Ottawa Scales (NOS).

Interventions: SAC or DO.

Main outcome measures: Blood loss and blood transfusion.

Results: Thirteen studies were included, with 7 studies comparing DO versus conventional and 6 comparing SAC vs conventional. All studies met eligibility criteria for meta-analysis, with study quality ranged from good to very good. Compared to conventional, the SAC or DO significantly reduced blood loss (MD = -190.42 mL), and blood transfusion (MD = -227.22). Additionally, SAC and DO shorten operative time (MD = -94.38 min), anesthesia duration (MD = -114.81 min), hospital stay (MD = -0.68 days), and ICU stay (MD = -1.00 days). Long-term follow-up showed a lower reoperation rate (OR = 0.20), but no significant change in cranial index at 10 years (MD = 0.06, P = .74).

Conclusions: SAC or DO result in lower perioperative complications, overall shorter durations, and reduced reoperation rates compared to conventional expansion. Standardized postoperative outcome reports are useful to classify the severity of complications and guide the future long-term treatment strategies for craniosynostosis surgery.

Objective: This study aims to estimate the geospatial distribution of cleft lip/palate (CL/P) cases in northwestern Nigeria and to estimate the prevalence and patterns of CL/P across wards.

Design: This retrospective study utilized information from health records for inpatients with CL/P. These data were analyzed via descriptive statistics. Spatial mapping involves geocoding street addresses into map coordinate system before aggregation for subsequent spatial analyses.

Setting: The center used was a nongovernmental organization clinic that manages CL/P.

Participants: All patients managed during the study period under 15 years were selected for the study, while the spatial analysis included all patients with valid addresses.

Results: A total of 1556 cases were selected, while spatial analyses mapped 928 cases. The analysis indicated over half (54.4%, n = 505 patients) lived in Kano Metropolis, whereas 45.6% (n = 423 patients) lived outside the Metropolis. CL/P prevalence correlated with the population density pattern in Kano. The prevalence of clefts was highest in Metropolis in 2008, and the Metropolis had the highest prevalence of clefts for children under 5 years of age. The significant patient and maternal factors were age (P < .001), weight (P < .001), socioeconomic status (P < .003), positive family history of cleft (P < .001), and maternal factors.

Conclusion: The GIS analysis revealed that most patients with CL/P who received treatment at the NGO lived nearby, with the greatest prevalence of clefts occurring in the Metropolis. The cost of travel may explain why those further afield do not come in for treatment or lack public awareness about the services provided at the cleft clinic.

CAAs are congenital malformations of the auricle ranging from ear underdevelopment to anotia, lacks standardized classification, impacting our outcome of different reconstruction approaches. This scoping review aimed to explore which CAA classifications are most used in current ear reconstruction practices.

We conducted a scoping review following the PRISMA guidelines, searching MEDLINE and Embase databases on November 1st, 2023. Studies on CAA reconstruction that included clear descriptions of the used classification published in the past 5 years were included. Studies were appraised using the Joanna Briggs Institute checklist.

Out of 293 screened studies, 45 met inclusion criteria, encompassing 19 case series and 5 cohort studies. Findings revealed a predominant use of the Nagata classification across rib cartilage and alloplastic material reconstructions, despite noted application inconsistencies. Other systems like Marx's, Weerda's, and Meurman's remain underutilized.

Most studies used the Nagata's classification. Its widespread use underscores the necessity for an easy to use, but standardized classification to improve surgical outcome reporting and assessment accuracy. Further investigation and standardization efforts regarding the Nagata system are recommended.

The purpose of this study was to quantitatively assess the alveolar bone support of teeth adjacent to the cleft site in individuals with nonsyndromic cleft lip and palate (CLP) who have undergone either orthodontic space closure or space opening for missing lateral incisors.

A cross-sectional retrospective study.

University orthodontic clinic serving individuals with CLP.

Twenty-eight individuals with nonsyndromic CLP who were missing lateral incisors divided into 2 groups: space closure (21 subjects) and space opening (7 subjects).

Orthodontic space closure or space opening for replacement of missing lateral incisors in individuals with nonsyndromic CLP.

Buccal and palatal alveolar bone thickness were measured at 5 mm and 10 mm above the cementoenamel junction (CEJ) for cleft-adjacent central incisors and canines. Additionally, buccal, lingual, and proximal alveolar bony coverage ratio on cleft-side central incisors and canines were recorded.

No significant differences were observed in alveolar bone thickness and bony coverage between the space closure group and the space opening group, except for the buccal thickness 5 mm above the CEJ, which was thinner in the space closure group.

The overall alveolar bone support in the grafted alveolus in both the space closure and space opening groups were comparable.

Objective: Post-surgical lip symmetry assessment is a key indicator of cleft repair success. Traditional methods rely on distances between anatomical landmarks, which are impractical for video analysis and overlook texture and appearance. We propose an artificial intelligence (AI) approach to automate this process, analyzing lateral lip morphology for a quantitative symmetry evaluation.

Design: We utilize contrastive learning to quantify lip symmetry by measuring the similarity between the representations of the sides, which is subsequently used to classify the severity of asymmetry. Our model does not require patient images for training. Instead, we introduce dissimilarities in face images from open datasets using two methods: temporal misalignment for video frames and face transformations to simulate lip asymmetry observed in the target population. The model differentiates the left and right image representations to assess asymmetry. We evaluated our model on 146 images of patients with repaired cleft lip.

Results: The deep learning model trained with face transformations categorized patient images into five asymmetry levels, achieving a weighted accuracy of 75% and a Pearson correlation of 0.31 with medical expert human evaluations. The model utilizing temporal misalignment achieved a weighted accuracy of 69% and a Pearson correlation of 0.27 for the same classification task.

Conclusions: We propose an automated approach for assessing lip asymmetry in patients with repaired cleft lip by transforming facial images of control subjects to train a deep learning model, eliminating manual anatomical landmarks. Our promising results provide a more efficient and objective tool for evaluating surgical outcomes.

To describe the frequency and types of hearing loss in children with syndromic and non-syndromic craniosynostosis.

Retrospective cohort study.

Large tertiary pediatric hospital.

Children with craniosynostosis that underwent at least one audiological evaluation between the years of 2010 and 2021 at a single institution. Hearing loss was defined as conductive or permanent (sensorineural or mixed).

Of 130 total children examined, 107 (82.3%) had non-syndromic craniosynostosis and 23 (17.7%) had syndromic craniosynostosis. Within the non-syndromic cohort, 77 (72%) had normal hearing and 30 (28%) had hearing loss, of which 21/30 (70%) had conductive hearing loss and 9/30 (30%) had permanent hearing loss. Within the syndromic cohort, two (9.5%) had normal hearing and 21 (90.5%) had hearing loss, of which 16/21 (76.2%) had conductive hearing loss and 5/21 (23.8%) had permanent hearing loss. Multivariable analysis involving syndromic status, anatomical type of craniosynostosis, and medical complexity revealed that patients with syndromic craniosynostosis had higher odds for conductive and permanent hearing loss (49.80 OR, P = .002 and 34.91 OR, P < .05, respectively). Patients with unicoronal craniosynostosis and those with significant medical comorbidities had higher odds for permanent hearing loss (19.50 OR, P = .03 and 23.36 OR, P < .05, respectively).

Children with craniosynostosis had high rates of hearing loss, especially those with syndromic or unicoronal craniosynostosis and those with significant medical comorbidities. Twenty-eight percent of patients with non-syndromic craniosynostosis also had hearing loss. Larger prospective studies are necessary to more precisely estimate hearing loss associated with non-syndromic craniosynostosis.