Skip to main content

Advertisement

SpringerLink
Log in

Multiple Orthokeratinized Odontogenic Cysts: A Case Report

  • Case Report
  • Published:
Head and Neck Pathology Aims and scope Submit manuscript

Abstract

The purpose of this report is to document the clinical, radiographic, pathological and molecular findings of the first case of multiple orthokeratinized odontogenic cysts (OOCs). Multiple odontogenic keratocysts are one of the major features of nevoid basal cell carcinoma syndrome (NBCCS), and loss of heterozygosity in the PTCH gene, the culprit gene for NBCCS, has recently been found in sporadic OOC cases. Therefore, in this presenting case, we also investigated the possibility that this patient might also have NBCCS, by comparing the available clinical information and the molecular findings of this case to the diagnostic criteria for NBCCS (as proposed by the First International Colloquium on NBCCS in 2011). However, this patient with multiple OOCs showed no evidence of having NBCCS. This conclusion supports the findings from previous case series based on sporadic cases that OOC does not appear to be associated with NBCCS.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1
Fig. 2

References

  1. Wright JM. The odontogenic keratocyst: orthokeratinized variant. Oral Surg Oral Med Oral Pathol. 1981;51(6):609–18.

    Article  CAS  PubMed  Google Scholar 

  2. Li TJ, Kitano M, Chen XM, et al. Orthokeratinized odontogenic cyst: a clinicopathological and immunocytochemical study of 15 cases. Histopathology. 1998;32(3):242–51.

    Article  CAS  PubMed  Google Scholar 

  3. Dong Q, Pan S, Sun LS, Li TJ. Orthokeratinized odontogenic cyst: a clinicopathologic study of 61 cases. Arch Pathol Lab Med. 2010;134(2):271–5.

    PubMed  Google Scholar 

  4. Crowley TE, Kaugars GE, Gunsolley JC. Odontogenic keratocysts: a clinical and histologic comparison of the parakeratin and orthokeratin variants. J Oral Maxillofac Surg. 1992;50(1):22–6.

    Article  CAS  PubMed  Google Scholar 

  5. Neville BW, Damm DD, Allen CM, Bouquot JE. Orthokeratinized odontogenic cyst. In: Neville BW, Damm DD, Allen CM, Bouquot JE, editors. Oral and maxillofacial pathology. St. Louis: Saunders Elsevier; 2009. p. 687–8.

    Google Scholar 

  6. Bree AF, Shah MR. Consensus statement from the first international colloquium on basal cell nevus syndrome (BCNS). Am J Med Genet A. 2011;155A(9):2091–7.

    Article  PubMed  Google Scholar 

  7. Diniz MG, Galvao CF, Macedo PS, Gomes CC, Gomez RS. Evidence of loss of heterozygosity of the PTCH gene in orthokeratinized odontogenic cyst. J Oral Pathol Med. 2011;40(3):277–80.

    Article  CAS  PubMed  Google Scholar 

  8. Macdonald-Jankowski DS. Orthokeratinized odontogenic cyst: a systematic review. Dentomaxillofac Radiol. 2010;39(8):455–67.

    Article  PubMed Central  CAS  PubMed  Google Scholar 

  9. Kasat VO, Saluja H, Kalburge JV, et al. Multiple bilateral supernumerary mandibular premolars in a non-syndromic patient with associated orthokeratised odontogenic cyst—a case report and review of literature. Contemp Clin Dent. 2012;3(Suppl 2):S248–52.

    Article  PubMed Central  PubMed  Google Scholar 

  10. Aragaki T, Michi Y, Katsube K, et al. Comprehensive keratin profiling reveals different histopathogenesis of keratocystic odontogenic tumor and orthokeratinized odontogenic cyst. Hum Pathol. 2010;41(12):1718–25.

    Article  CAS  PubMed  Google Scholar 

  11. Zhang JY, Dong A, Li TJ. Differences in collagen fibres in the capsule walls of parakeratinized and orthokeratinized odontogenic cysts. Int J Oral Maxillofac Surg. 2011;40(11):1296–300.

    Article  PubMed  Google Scholar 

  12. Vered M, Shohat I, Buchner A, Dayan D. Myofibroblasts in stroma of odontogenic cysts and tumors can contribute to variations in the biological behavior of lesions. Oral Oncol. 2005;41(10):1028–33.

    Article  PubMed  Google Scholar 

  13. da Silva MJ, de Sousa SO, Correa L, Carvalhosa AA, De Araujo VC. Immunohistochemical study of the orthokeratinized odontogenic cyst: a comparison with the odontogenic keratocyst. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2002;94(6):732–7.

    Article  PubMed  Google Scholar 

  14. Roy S, Garg V. Evaluation of stromal myofibroblasts expression in keratocystic odontogenic tumor and orthokeratinized odontogenic cysts: a comparative study. J Oral Maxillofac Pathol. 2013;17(2):207–11.

    Article  PubMed Central  PubMed  Google Scholar 

  15. Caetano Ados S, Tjioe KC, Faustino SE, et al. Immunolocalization of podoplanin in benign odontogenic tumours with and without ectomesenchyme. Arch Oral Biol. 2013;58(4):408–15.

    Article  PubMed  Google Scholar 

  16. Deyhimi P, Hashemzade Z. Comparative study of TGF-alpha and P53 markers’ expression in odontogenic keratocyst and orthokeratinaized odontogenic cyst. Dent Res J (Isfahan). 2012;9(Suppl 1):S39–44.

    Google Scholar 

  17. Rangiani A, Motahhary P. Evaluation of bax and bcl-2 expression in odontogenic keratocysts and orthokeratinized odontogenic cysts: a comparison of two cysts. Oral Oncol. 2009;45(7):e41–4.

    Article  CAS  PubMed  Google Scholar 

  18. Iezzi G, Piattelli A, Artese L, et al. KAI-1 protein expression in odontogenic cysts. J Endod. 2007;33(3):235–8.

    Article  PubMed  Google Scholar 

  19. Thosaporn W, Iamaroon A, Pongsiriwet S, Ng KH. A comparative study of epithelial cell proliferation between the odontogenic keratocyst, orthokeratinized odontogenic cyst, dentigerous cyst, and ameloblastoma. Oral Dis. 2004;10(1):22–6.

    Article  CAS  PubMed  Google Scholar 

  20. Piattelli A, Fioroni M, Iezzi G, Rubini C. Calretinin expression in odontogenic cysts. J Endod. 2003;29(6):394–6.

    Article  PubMed  Google Scholar 

  21. Vuhahula E, Nikai H, Ijuhin N, et al. Jaw cysts with orthokeratinization: analysis of 12 cases. J Oral Pathol Med. 1993;22(1):35–40.

    Article  CAS  PubMed  Google Scholar 

  22. Johnson RL, Rothman AL, Xie J, et al. Human homolog of patched, a candidate gene for the basal cell nevus syndrome. Science. 1996;272(5268):1668–71.

    Article  CAS  PubMed  Google Scholar 

  23. Hahn H, Wicking C, Zaphiropoulous PG, et al. Mutations of the human homolog of Drosophila patched in the nevoid basal cell carcinoma syndrome. Cell. 1996;85(6):841–51.

    Article  CAS  PubMed  Google Scholar 

  24. Fan Z, Li J, Du J, et al. A missense mutation in PTCH2 underlies dominantly inherited NBCCS in a Chinese family. J Med Genet. 2008;45(5):303–8.

    Article  CAS  PubMed  Google Scholar 

  25. Pastorino L, Ghiorzo P, Nasti S, et al. Identification of a SUFU germline mutation in a family with Gorlin syndrome. Am J Med Genet A. 2009;149A(7):1539–43.

    Article  CAS  PubMed  Google Scholar 

  26. Jones EA, Sajid MI, Shenton A, Evans DG. Basal cell carcinomas in gorlin syndrome: a review of 202 patients. J Skin Cancer. 2011. doi:10.1155/2011/217378.

  27. Neville BW, Damm DD, Allen CM, Bouquot JE. Nevoid basal cell carcinoma syndrome (Gorlin syndrome). In: Neville BW, Damm DD, Allen CM, Bouquot JE, editors. Oral and maxillofacial pathology. St. Louis: Saunders Elsevier; 2009. p. 688–91.

    Google Scholar 

Download references

Conflict of interest

The authors declare that they have no conflict of interest.

Author information

Authors and Affiliations

  1. Diagnostic Sciences, Texas A&M University-Baylor College of Dentistry, 3302 Gaston Ave, Dallas, TX, 75246, USA

    Yi-Shing Lisa Cheng, Hui Liang & John Wright

  2. Oral and Maxillofacial Surgery Private Practice, 5402 Holly Road, Suite 302, Building 3, Corpus Christi, TX, 78411, USA

    Tom Teenier

Authors
  1. Yi-Shing Lisa Cheng
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Hui Liang
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. John Wright
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Tom Teenier
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Yi-Shing Lisa Cheng.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Cheng, YS.L., Liang, H., Wright, J. et al. Multiple Orthokeratinized Odontogenic Cysts: A Case Report. Head and Neck Pathol 9, 153–157 (2015). https://doi.org/10.1007/s12105-014-0545-5

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s12105-014-0545-5

Keywords

Search

Navigation