Non-syndromic and syndromic keratocystic odontogenic tumors: Systematic review and meta-analysis of recurrences
Introduction
The keratocystic odontogenic tumor (KCOT) obtained its name in 2005 when the World Health Organization (Philipsen, 2005), decided to choose KCOT over the traditionally used term of odontogenic keratocyst. KCOTs are locally aggressive benign tumors which occur in the bone of both jaws with a notably high recurrence rate. A number of treatments have been suggested for KCOTs and various groups have attempted to determine which is the most efficacious (Blanas et al., 2000, Sharif et al., 2010, Kaczmarzyk et al., 2012, Johnson et al., 2013). One group (Johnson et al., 2013) suggests that enucleation followed by the application of Carnoy's solution and resection resulted in the lowest recurrence rates, but this left surgeons to weigh the implications of a more aggressive approach such as resection. Another group (Kaczmarzyk et al., 2012) strongly critiques the available evidence and states that it is impossible to make a strong conclusion regarding a universal treatment of choice. The gold standard for the treatment of KCOT's is still debatable, mainly because there is no reliable summary of recurrence rates associated with the treatments available to date.
The aim of the present study was to extract the data available for solitary KCOT lesions occurring in non-syndromic patients from a large number of published studies regarding KCOTs. The authors aimed to investigate the association between different treatment forms and recurrence rates of solitary non-syndromic KCOT lesions. In addition to the previous aim, data were collected from trials or trial arms which included nevoid basal cell carcinoma syndrome (NBCCS) patients to determine the recurrence rates following various forms of treatment.
Section snippets
Protocol and eligibility criteria
The present systematic review is conducted and reported according to the Cochrane Handbook (Higgins and Green, 2011) and the Preferred Reporting Items for Systematic Reviews and Meta-analysis (PRISMA) statement (Liberati et al., 2009) and its extension for abstracts (Beller et al., 2013), respectively. Since no randomized controlled trials involving KCOTs were identified in the preliminary literature search, a systematic review of both randomized and non-randomized studies was planned. The
Results
The electronic search yielded 2742 results and hand searching 27 (Fig. 1). The search of trial registers retrieved no matching results. After removing duplicates, selecting potentially relevant studies and screening for eligibility criteria the authors included 43 studies regarding treatment in non-syndromic patients and 11 studies regarding treatment of KCOTs in syndromic patients. The agreement level reached 93% the disagreements were resolved by discussion and there was no need for a third
Summary of evidence
The low quality of evidence prevented the drawing of concrete conclusions regarding the treatment form with the lowest recurrence over three years in non-syndromic or syndromic patients. We attempted to reach clinically relevant conclusions and in our analyses a minimum follow-up period of three years was used as most recurrences appear during the first five years (Zhao et al., 2002, Gosau et al., 2010, Apajalahti et al., 2011), although the recurrence rate might be greater, if the follow up is
Conclusion
Based on existing evidence no clinical recommendations can be made for the treatment of KCOTs in either non-syndromic or syndromic patients. Additional prospective controlled clinical studies, ideally randomized and blinded, with adequate sample size and a follow-up of at least three years are needed for both NBCCS and non-NBCCS cohorts.
Funding
This study was funded by an EVO grant from the Oulu University Hospital Pohjois-Pohjanmaan Sairaanhoitopiiri.
Financial disclosure
None.
Conflict of interest
None.
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