Department of Oral Medicine and Pathology, Faculty of Dentistry, National and Kapodistrian University of Athens, Athens, Greece.
Center of Systems Biology, Biomedical Research Foundation, Academy of Athens, Athens, Greece.
Clin Oral Investig. 2021 Jun;25(6):3351-3367. doi: 10.1007/s00784-021-03877-w. Epub 2021 Mar 17.
To provide a systematic review of the literature on studies comparing the immunoprofile of nevoid basal cell carcinoma syndrome (BCNS)-associated and sporadic odontogenic keratocysts (OKCs), in order to identify markers that could accurately distinguish the two OKC subtypes.
We searched MEDLINE/Pubmed, Web of Science, EMBASE via OVID, and grey literature for publications until December 28th, 2019, that compared the immunohistochemical expression of the two OKC subtypes. The studies were qualitatively assessed using the Critical Appraisal Tool for Case Series (Joana Briggs Institute). Sensitivity and specificity, positive and negative likelihood ratio, diagnostic odds ratio and area under the curve, and pooled estimates were calculated, using a random-effects model.
Seventy-one studies were qualitatively analyzed; 61 markers were evaluated in one study and 32 in ≥ 2 studies. Twenty-five studies reported differential expression of 29 markers in the form of higher number of positive cells or greater staining intensity usually in BCNS-associated OKCs. Meta-analysis for bcl-2, Cyclin D1, CD56, CK18, p53, and PCNA showed that none of those markers is distinguishable between BCNS-associated and sporadic OKCs, in a 95% confidence interval. The risk of bias was high in 34 studies, moderate in 22, and low in 15.
The present systematic review and meta-analysis uncovered that, although several immunohistochemical markers might characterize the OKC phenotype, they cannot discriminate between the BCNS-associated and sporadic OKCs.
This study highlighted the requirement for additional screening for markers by immunohistochemistry, preferentially coupled to alternative diagnostic applications such as genomics technologies.
对比较神经嵴发育不良性基底细胞癌综合征（BCNS）相关和散发性牙源性角化囊肿（OKC）免疫表型的研究进行系统综述，以确定可准确区分两种 OKC 亚型的标志物。
我们检索了 MEDLINE/Pubmed、Web of Science、EMBASE via OVID 和灰色文献，以获取截至 2019 年 12 月 28 日比较两种 OKC 亚型免疫组织化学表达的出版物。使用病例系列的批判性评估工具（Joana Briggs 研究所）对研究进行定性评估。使用随机效应模型计算敏感性和特异性、阳性和阴性似然比、诊断优势比和曲线下面积以及汇总估计值。
对 71 项研究进行了定性分析；1 项研究评估了 61 个标志物，≥2 项研究评估了 32 个标志物。25 项研究报告了 29 个标志物的差异表达，通常表现为 BCNS 相关 OKC 中阳性细胞数量较多或染色强度较大。bcl-2、Cyclin D1、CD56、CK18、p53 和 PCNA 的 meta 分析表明，在 95%置信区间内，这些标志物均不能区分 BCNS 相关和散发性 OKC。34 项研究的偏倚风险较高，22 项研究为中度，15 项研究为低度。
本系统综述和 meta 分析发现，尽管几种免疫组织化学标志物可能具有特征性 OKC 表型，但它们不能区分 BCNS 相关和散发性 OKC。
本研究强调需要通过免疫组织化学进行额外的标志物筛选，最好与基因组学技术等替代诊断应用相结合。
