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Meta-Analysis
. 2022 Apr 5;12(1):5708.
doi: 10.1038/s41598-022-09764-y.

Craniofacial morphology in Apert syndrome: a systematic review and meta-analysis

Mohammad Khursheed Alam  1 , Ahmed Ali Alfawzan  2 , Kumar Chandan Srivastava  3 , Deepti Shrivastava  4 , Kiran Kumar Ganji  4 , Srinivas Munisekhar Manay  4
Affiliations
Meta-Analysis

Craniofacial morphology in Apert syndrome: a systematic review and meta-analysis

Mohammad Khursheed Alam et al. Sci Rep. .

Abstract

This meta-analysis aims to compare Apert syndrome (AS) patients with non-AS populations (not clinically or genetically diagnosed) on craniofacial cephalometric characteristics (CCC) to combine publicly available scientific information while also improving the validity of primary study findings. A comprehensive search was performed in the following databases: PubMed, Google Scholar, Scopus, Medline, and Web of Science, an article published between 1st January 2000 to October 17th, 2021. PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) guidelines were followed to carry out this systematic review. We used the PECO system to classify people with AS based on whether or not they had distinctive CCC compared to the non-AS population. Following are some examples of how PECO has been used: People with AS are labeled P; clinical or genetic diagnosis of AS is labeled E; individuals without AS are labeled C; CCC of AS are labeled O. Using the Newcastle-Ottawa Quality-Assessment-Scale, independent reviewers assessed the articles' methodological quality and extracted data. 13 studies were included in the systematic review. 8 out of 13 studies were score 7-8 in NOS scale, which indicated that most of the studies were medium to high qualities. Six case-control studies were analyzed for meta-analysis. Due to the wide range of variability in CCC, we were only able to include data from at least three previous studies. There was a statistically significant difference in N-S-PP (I2: 76.56%; P = 0.014; CI 1.27 to - 0.28) and Greater wing angle (I2: 79.07%; P = 0.008; CI 3.07-1.17) between AS and control subjects. Cleft palate, anterior open bite, crowding in the upper jaw, and hypodontia occurred more frequently among AS patients. Significant shortening of the mandibular width, height and length is the most reported feature in AS patients. CT scans can help patients with AS decide whether to pursue orthodontic treatment alone or to have their mouth surgically expanded. The role of well-informed orthodontic and maxillofacial practitioners is critical in preventing and rehabilitating oral health issues.

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Conflict of interest statement

The authors declare no competing interests.

Figures

Figure 1
Figure 1
Keywords used in search strategies.
Figure 2
Figure 2
PRISMA flow diagram of the search strategies.
Figure 3
Figure 3
Cephalometric landmark of the different angle.
Figure 4
Figure 4
Forest plot for the outcome of SNA.
Figure 5
Figure 5
Forest plot for the outcome of SNB.
Figure 6
Figure 6
Forest plot for the outcome of PPR-S-PPL.
Figure 7
Figure 7
Forest plot for the outcome of N-S-PP.
Figure 8
Figure 8
Forest plot for the outcome of SNA greater wing angle.
Figure 9
Figure 9
Galbraith plot for the outcome of (a) SNA, (b) SNB, (c) PPR-S-PPL, (d) N-S-PP, and (e) greater wing angle.
Figure 10
Figure 10
Funnel plot for the outcome of (a) SNA, (b) SNB, (c) PPR-S-PPL, (d) N-S-PP, and (e) greater wing angle.
See this image and copyright information in PMC

References

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