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The Presentation of Odontogenic Keratocysts in the Jaws with an Emphasis on the Tooth-Bearing Area: a Systematic Review and Meta-Analysis

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The Presentation of Odontogenic Keratocysts in the Jaws with an Emphasis on the Tooth-Bearing Area: a Systematic Review and Meta-Analysis Oral and Maxillofacial Surgery (2019) 23:133–147 https://doi.org/10.1007/s10006-019-00754-5 REVIEW ARTICLE The presentation of odontogenic keratocysts in the jaws with an emphasis on the tooth-bearing area: a systematic review and meta-analysis Yuri Slusarenko da Silva1 & Paul J. W. Stoelinga2 & Maria da Graça Naclério-Homem1 Received: 7 October 2018 /Accepted: 21 February 2019 /Published online: 2 March 2019 # Springer-Verlag GmbH Germany, part of Springer Nature 2019 Abstract Purpose This study was conducted in order to gain insight in the actual ratio of odontogenic keratocysts occurring in the tooth- bearing area as compared to the posterior region of the jaws in order to come up with reliable data to base upon a rational treatment policy. Methods We searched MEDLINE, Web of Science, Scopus, and Cochrane databases for studies reporting on the location of mandibular and maxillary odontogenic keratocysts. All records were independently assessed and a meta-analysis was performed. Risk difference with a confidence interval of 95% of having the lesion in the posterior region versus the tooth-bearing area was the effect measure. P value for the summary effect of < 0.05 was considered statistically significant. Results The 2615 records retrieved were reduced to 34 studies to be qualitatively/quantitatively assessed. The pooled values showed that the difference in the clinical risk of having keratocysts in the posterior region of the mandible and in the tooth-bearing area of the maxilla is 21 and 43%, respectively (P <0.02andP <0.00001). Conclusions A substantial amount of keratocysts occur in the tooth-bearing area of the jaws, requiring attention. Keywords Odontogenic keratocyst . Keratocystic odontogenic tumor . Prevalence . Systematic review . Meta-analysis Introduction mandibular area should be treated as if they were OKCs or cystic ameloblastomas. In a recent systematic review and me- It is well known that odontogenic keratocysts (OKCs) are ta-analysis, Al-Moraissi et al. [2]haveproposedtotreatOKCs often occurring in the posterior mandible/ascending ramus, according to a defined protocol as suggested by Stoelinga [3]. and also in the maxilla, they tend to be located in the molar This includes excision of the overlying attached mucosa, enu- tuberosity area. In fact, the vast majority of cysts in the pos- cleation of the cyst, and treatment of the bony defect with terior mandible are OKCs, while sporadically one may en- Carnoy’s solution. This would be entirely possible for cysts counter a cystic ameloblastoma or a regular odontogenic in the posterior mandible (third molar area and extending into (dentigerous) cyst in that area. This has consequences for the the ascending ramus) and even posterior maxilla, but it is surgical approach as outlined by Chapelle et al. [1]. They different with cysts in the tooth-bearing area because they suggested an algorithm for the treatment of cystic lesions of are usually not easily diagnosed as such. They often present the jaws, which entailed that all cysts in the posterior as lateral periodontal or (lateral) dentigerous cysts not arising any suspicion. The diagnosis is usually made after enucleation not followed by any additional treatment. In a prospective * Yuri Slusarenko da Silva study of Stoelinga [3], 24 of 75 OKCs were located in the [email protected] tooth-bearing area of which five recurred between 1 and 10 years, whereas only four recurrences (less than 10%) were 1 Department of Oral & Maxillofacial Surgery, Traumatology and seen of the 51 OKCs located in the posterior mandible or Prosthesis, Faculty of Dentistry of the University of São Paulo, Av maxilla. Prof. Lineu Prestes 2227, Butantã, São Paulo 05508-000, Brazil For reasons mentioned above, it would be advisable to 2 Department of Oral and Maxillofacial Surgery, Radboud University, have some insight in the actual ratio of OKCs occurring in Nijmegen, The Netherlands the tooth-bearing area as compared to the posterior area of the 134 Oral Maxillofac Surg (2019) 23:133–147 jaws. Some solid data on the presentation of OKCs in Search strategy both jaws would be helpful to formulate a treatment policy for those OKCs occurring in the tooth-bearing The first hit was conducted online by two independent re- areas. It is the intention of this systematic review and viewers (YSS and MN), in MEDLINE (via PubMed), Web meta-analysis to come up with reliable data to base of Science, Scopus, Cochrane Library, and Google Scholar upon a rational treatment policy. (other source), from the inception until January 31st of 2018. Publications were searched using the following strate- gy: (((((((odontogenic keratocyst) OR keratocystic Material and methods odontogenic tumor) OR keratocystic odontogenic tumour) OR primordial cyst)) AND (((odontogenic cysts) OR jaw Protocol and registration cysts) OR developmental odontogenic cysts)) AND ((((diag- nosis) OR incidence) OR prevalence) OR demographics)) The present review followed the Preferred Reporting Items for NOT case report. Duplicate records were subsequently Systematic Reviews and Meta-Analyses—PRISMA protocol removed. [4] and was registered in the International Prospective Register of Systematic Reviews— PROSPERO Study selection (CRD42018092088). Records that remained from the first hit were independently Focused question selected by reading their title and abstract (first round). Disagreements in this selection were resolved by mutual dis- We intended to answer the following focused question: cussion. Afterwards, all records screened from the first round BWhat’s the overall ratio of OKCs that occur in the posterior had their full text independently assessed for eligibility by the region versus the dentate area of the mandible and maxilla?^ same reviewers. Eligibility criteria Data collection process Original randomized/nonrandomized clinical trials and The reviewers YSS and MN separately submitted all eli- observational studies written in English were considered gible studies to a qualitative synthesis using an extraction in the present systematic review (https://prsinfo. data form including mainly country and total period of the clinicaltrials.gov/definitions.html [Accessibility verified study, clinical characteristics of the patients, and number/ June 11, 2018]). Records that fulfilled the following location of the primary OKCs. The mandible and maxilla items were included: (a) human patients with primary were considered separately. Whenever the article men- OKC histologically diagnosed [5] and (b) location of tioned only Bposterior or anterior region,^ without a pre- the OKCs in the posterior region and/or in the dentate cise description of the cysts location, we considered these area of the mandible and/or maxilla. According to our areas as being the same as of our protocol, as stated in the protocol, the posterior region was considered to be the BEligibility criteria^ section. mandibular ramus/angle and maxillary tuberosity, in- Subsequently, all extraction data forms with the results of cluding those OKCs in the third molar area. On the each included study were verified together in order to calibrate other hand, the dentate area was considered to be the validity and reliability of this process. tooth-bearing area/alveolar process, including those OKCs intimately related with teeth (i.e., incisors, ca- Risk of bias in individual studies nines, premolars and first/second molars). Records presenting data from (a) recurrent OKCs; (b) To assess the quality of the studies, we adapted a few OKCs associated with the nevoid basal cell carcinoma syn- checklists from the Joanna Briggs Institute (http:// drome; (c) cystic lesions that are lined only by orthokeratotic joannabriggs.org/research/critical-appraisal-tools.html epithelium; (d) histological and molecular studies; and (e) [Accessibility verified June 11, 2018]) and applied them in vitro and animal experiments, literature reviews, systematic to be used in Review Manager Software 5.3 (Review reviews, case reports, and studies with less than ten partici- Manager (RevMan) [Computer program]. Version 5.3. pants were not considered. Copenhagen: The Nordic Cochrane Centre, The Studies that met the inclusion criteria or those with doubtful Cochrane Collaboration, 2014). information either in the title or abstract were selected for full- The queries of the included studies are briefly ex- text assessment in a second round of this review. Reasons for plained as follows: (a) cases from the same country; (b) rejection of studies were recorded for each report. validity of data collection (i.e., valid clinical and Oral Maxillofac Surg (2019) 23:133–147 135 radiographic assessment or valid retrospective analysis); the searched records and the number of studies finally includ- (c) evaluation of confounding factors such as the inclu- ed [3, 6–38] are shown in the flow diagram (Fig. 1). sion of OKCs based on histological criteria other than that recommended by the World Health Organization [5]and the presence of recurrent OKCs, syndromic OKCs, and Excluded studies orthokeratocysts in the study sample; and (d) strategies to deal with confounding factors. The studies that were not included in the present systematic We also assessed the risk of bias of the OKCs occupying were excluded for several reasons: the posterior maxilla, and large cysts may have occurred con- comitantly in the posterior region and molar area (dentate & No specification if the OKC was located in the mandible area). or maxilla [39–80] & No specification if the OKC was located in the dentate Summary measures and synthesis of results area or in the posterior region of the jaws [39–44, 51, 60, 63, 78, 81–135] Meta-analysis was performed in the Review Manager soft- & Impossibility to distinguish nonsyndromic from ware 5.3, and the risk difference (RD) between the proportions syndromic OKCs [46, 60, 63, 69, 76, 80, 81, 83, 89, 92, of individuals having primary OKCs (event) in the posterior 99, 100, 102–106, 111, 119, 126, 129, 136–154] region versus in the tooth-bearing area with a confidence in- & Impossibility to distinguish OKCs from orthokeratocysts terval (CI) of 95% was the effect measure.
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