OBJECTIVE: This Bayesian network meta-analysis was performed to analyze the associations between clinicopathological characteristics and BRAF mutations in ameloblastoma (AM) patients and to evaluate the diagnostic accuracy.
METHODS: Four electronic databases were searched from 2010 to 2024. The search terms used were specific to BRAF and AM. Observational studies or randomized controlled trials were considered eligible. The incidence of BRAF mutation and corresponding clinicopathological features in AM patients were subjected to Bayesian network analyses and diagnostic accuracy evaluation.
RESULTS: A total of 937 AM patients from 20 studies were included. The pooled prevalence of BRAF mutations in AM patients was 72%. According to the Bayesian network analysis, BRAF mutations are more likely to occur in younger (odds ratio [OR], 2.3; credible interval [CrI]: 1.2-4.5), mandible site (OR, 3.6; 95% CrI: 2.7-5.2), and unicystic (OR, 1.6; 95% CrI: 1.1-2.4) AM patients. Similarly, higher diagnostic accuracy was found in the younger, mandible, and unicystic AM groups.
CONCLUSIONS: The incidence, risk, and diagnostic accuracy of BRAF mutation in AM were greater in younger patients, those with mandible involvement, and those with unicystic AM than in patients with other clinicopathological features. In addition, there was a strong concordance in the diagnostic accuracy between molecular tests and immunohistochemical analysis.

The meta-analysis of rare events presents unique methodological challenges owing to the small number of events. Bayesian methods are often used to combine rare events data to inform decision-making, as they can incorporate prior information and handle studies with zero events without the need for continuity corrections. However, the comparative performances of different Bayesian models in pooling rare events data are not well understood. We conducted a simulation to compare the statistical properties of four parameterizations based on the binomial-normal hierarchical model, using two different priors for the treatment effect: weakly informative prior (WIP) and non-informative prior (NIP), pooling randomized controlled trials with rare events using the odds ratio metric. We also considered the beta-binomial model proposed by Kuss and the random intercept and slope generalized linear mixed models. The simulation scenarios varied based on the treatment effect, sample size ratio between the treatment and control arms, and level of heterogeneity. Performance was evaluated using median bias, root mean square error, median width of 95% credible or confidence intervals, coverage, Type I error, and empirical power. Two reviews are used to illustrate these methods. The results demonstrate that the WIP outperforms the NIP within the same model structure. Among the compared models, the model that included the treatment effect parameter in the risk model for the control arm did not perform well. Our findings confirm that rare events meta-analysis faces the challenge of being underpowered, highlighting the importance of reporting the power of results in empirical studies.

Scale errors are intriguing phenomena in which a child tries to perform an object-specific action on a tiny object. Several viewpoints explaining the developmental mechanisms underlying scale errors exist; however, there is no unified account of how different factors interact and affect scale errors, and the statistical approaches used in the previous research do not adequately capture the structure of the data. By conducting a secondary analysis of aggregated datasets across nine different studies (n = 528) and using more appropriate statistical methods, this study provides a more accurate description of the development of scale errors. We implemented the zero-inflated Poisson (ZIP) regression that could directly handle the count data with a stack of zero observations and regarded developmental indices as continuous variables. The results suggested that the developmental trend of scale errors was well documented by an inverted U-shaped curve rather than a simple linear function, although nonlinearity captured different aspects of the scale errors between the laboratory and classroom data. We also found that repeated experiences with scale error tasks reduced the number of scale errors, whereas girls made more scale errors than boys. Furthermore, a model comparison approach revealed that predicate vocabulary size (e.g., adjectives or verbs), predicted developmental changes in scale errors better than noun vocabulary size, particularly in terms of the presence or absence of scale errors. The application of the ZIP model enables researchers to discern how different factors affect scale error production, thereby providing new insights into demystifying the mechanisms underlying these phenomena. A video abstract of this article can be viewed at https://youtu.be/1v1U6CjDZ1Q RESEARCH HIGHLIGHTS: We fit a large dataset by aggregating the existing scale error data to the zero-inflated Poisson (ZIP) model. Scale errors peaked along the different developmental indices, but the underlying statistical structure differed between the in-lab and classroom datasets. Repeated experiences with scale error tasks and the children\'s gender affected the number of scale errors produced per session. Predicate vocabulary size (e.g., adjectives or verbs) better predicts developmental changes in scale errors than noun vocabulary size.