Cervical vertebrae anomalies and cleft lip and palate: a systematic review and meta-analysis

Abstract

Objectives: This study investigated the association of congenital cervical vertebrae anomalies (CVA) with the prevalence of cleft lip and palate (CLP) specifying the most frequent associations. Methods: A meta-analysis was based on the Preferred Reporting Items of Systematic reviews and Meta-Analyses guideline. A search of the PubMed, Embase, Web of Science, Science Direct, Scopus and Lilacs database was performed until March 2018. Clinical studies that evaluated CVA in individuals with CLP (experimental group) and without CLP (control group) was included. For the statistical analysis, the software Comprehensive Meta-Analysis (Biostat; Englewood, NJ) was used, with a p-value < 0.05 considered significant. Results: A total of 10 articles were included, for a total of 2566 individuals with CLP and 2301 individuals without CLP. The meta-analysis indicated statistically significant differences and the group of individuals with CLP had an increased number of CVA when compared to the group of individuals without CLP (p < 0.05; 95% confidence interval, 2.41–6.11; heterogeneity: Q-value 32.8, I2 72.6%). Conclusions: This study indicated that CVA are associated with the presence of CLP. Among the patients with CVA the most frequent anomalies were the deficiency of the posterior arch, followed by the fusion of cervical vertebrae. Individuals with cleft palate and those with unilateral cleft lip and palateCLP had an increased prevalence of CVA.

Three-Dimensional Assessment of Cervical Vertebrae Anomalies in Patients With Cleft Lip and Palate

“…These cases of high proximity of vertebrae without fusion were classified as narrowing of the intervertebral space and represent 10.68% of the individuals with UCLP and 14.57% with BCLP, for an overall percentage of 11.92%. This prevalence, added to the true fusion cases, would represent 15.89% of the sample, a close value to the mean incidences of fusion reported in the literature (14.82%) (Berrocal et al, 2019). In this sense, one can assume that studies using lateral cephalometric radiographs may lead to false-positive fusion cases since the 2D image cannot depict fine separations between vertebrae.…”

“…When considering the groups separately, 25.24% of the individuals with UCLP presented some type of CVA, which is a higher frequency when compared with the literature (2.99%-24.39%) (Berrocal et al, 2019). Additionally, patients with BCLP also presented higher values (33.33%) than the previously reported (0%-19.1%).…”

“…Several studies evaluated CVA in individuals with and without CLP and it can be stated that patients with CLP are most likely to present some type of CVA (Ross and Lindsay, 1965; Osborne et al, 1971; Sandham, 1986; Horswell, 1991; Hoenig and Schoener, 1992; Uğar and Semb, 2001; Rajion et al, 2006; Sideri, 2013; Datana et al, 2014; Srivastava et al, 2014). Furthermore, a recent systematic review and meta-analysis determined an association of CVA with the presence of CLP (Berrocal et al, 2019).…”

“…Another limitation was related to the lack of patients with CP. In the systematic review conducted by Berrocal et al (2019), it was concluded that individuals with isolated CP had indeed an increased prevalence of CVAs. Nonetheless, since this type of cleft was not included in the present research sample, comparing the frequencies of patients with UCLP and BCLP with CP rates from previous studies would not be appropriate.…”

“…A recent systematic review and meta-analysis concluded that CVAs are associated with the presence of CLP and the most frequent anomalies observed in these patients were deficiency of the posterior arch and fusion of cervical vertebrae (Berrocal et al, 2019). However, as mentioned, the vast majority of studies included in that review consisted of examinations based on 2-dimensional cephalometric radiographs.…”