Non-syndromic and syndromic keratocystic odontogenic tumors: Systematic review and meta-analysis of recurrences

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Abstract

Background

Keratocystic odontogenic tumors (KCOTs) are locally aggressive benign tumors which occur in the bones of both jaws with a high recurrence rate.

The aim of the present study was to define and evaluate the post-treatment recurrence of KCOT lesions in non-syndromic and syndromic patients.

Methods

A systematic review of the literature and meta-analysis was conducted according to the PRISMA statement. Seven electronic databases were searched from their start up to August 2013 for clinical studies on human patients without limitation to year, language or publication status.

Results

A total of five case series studies with 323 treated KCOT lesions were included in the quantitative synthesis. The recurrence rate of KCOTs for three treatment forms ranged from 7% to 28%. Comparisons among the various treatments suggest that resection or marsupialization might be associated with fewer recurrences. However, high risk of bias and effect imprecision preclude the making of clinical recommendation. Existing evidence regarding nevoid basal cell carcinoma patients was likewise scarce.

Conclusions

The absence of studies with low risk of bias precludes the making of safe recommendations about the optimal management of KCOTs.

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.

References (46)

  • T. Kaczmarzyk et al.

    A systematic review of the recurrence rate for keratocystic odontogenic tumour in relation to treatment modalities

    Int J Oral Maxillofac Surg

    (2012)
  • N. Kadlub et al.

    Pediatric keratocystic odontogenic tumor and nevoid basal cell carcinoma syndrome. Predictive factors for recurrence and aggressiveness

    Rev Stomatol Chir Maxillofac

    (2012)
  • A. Kolokythas et al.

    Odontogenic keratocyst: to decompress or not to decompress? A comparative study of decompression and enucleation versus resection/peripheral ostectomy

    J Oral Maxillofac Surg

    (2007)
  • A. Liberati et al.

    The PRISMA statement for reporting systematic reviews and meta-analyses of studies that evaluate health care interventions: explanation and elaboration

    J Clin Epidemiol

    (2009)
  • P. Marker et al.

    Treatment of large odontogenic keratocysts by decompression and later cystectomy: a long-term follow-up and a histologic study of 23 cases

    Oral Surg Oral Med Oral Pathol Oral Radiol Endod

    (1996)
  • T.A. Morgan et al.

    A retrospective review of treatment of the odontogenic keratocyst

    J Oral Maxillofac Surg

    (2005)
  • H. Myoung et al.

    Odontogenic keratocyst: review of 256 cases for recurrence and clinicopathologic parameters

    Oral Surg Oral Med Oral Pathol Oral Radiol Endod

    (2001)
  • N. Nakamura et al.

    Marsupialization for odontogenic keratocysts: long-term follow-up analysis of the effects and changes in growth characteristics

    Oral Surg Oral Med Oral Pathol Oral Radiol Endod

    (2002)
  • P. Pitak-Arnnop et al.

    Management of odontogenic keratocysts of the jaws: a ten-year experience with 120 consecutive lesions

    J Craniomaxillofac Surg

    (2010)
  • R. Sasaki et al.

    Multiple keratocystic odontogenic tumors associated with nevoid basal cell carcinoma syndrome having distinct PTCH1 mutations: a case report

    Oral Surg Oral Med Oral Pathol Oral Radiol Endod

    (2010)
  • P.J. Stoelinga

    Long-term follow-up on keratocysts treated according to a defined protocol

    Int J Oral Maxillofac Surg

    (2001)
  • P.J. Stoelinga

    The treatment of odontogenic keratocysts by excision of the overlying, attached mucosa, enucleation, and treatment of the bony defect with carnoy solution

    J Oral Maxillofac Surg

    (2005)
  • L. Tonietto et al.

    Enucleation and liquid nitrogen cryotherapy in the treatment of keratocystic odontogenic tumors: a case series

    J Oral Maxillofac Surg

    (2011)
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