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Published in: Head & Face Medicine 1/2018

Open Access 01-12-2018 | Research

Orthokeratinized odontogenic cysts: a Spanish tertiary care centre study based on HPV DNA detection

Authors: Beatriz Vera-Sirera, Luis Rubio-Martínez, Leopoldo Forner-Navarro, Francisco Vera-Sempere

Published in: Head & Face Medicine | Issue 1/2018

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Abstract

Background

The role of human papillomavirus (HPV) in orthokeratinized odontogenic cysts (OOCs) has rarely been studied. The objective is to describe the clinicopathological findings in a series of OOCs from a Spanish population that were investigated in relation to the possible presence of HPV.

Methods

A clinicopathological retrospective analysis followed by a molecular analysis of 28 high- and low-risk HPV genotypes was performed in OOC samples of patients seen during the last 15-years in a Spanish tertiary care center.

Results

Of 115 odontogenic cysts with keratinization, 16 cases of OOCs were confirmed and evaluated. OOCs occurred predominantly in the mandible of males (mean age 36.06 ± 13.16 years). Swelling of the jaw followed by pain were the most common clinical symptoms, and 56.5% of the OOC cases were associated with an unerupted tooth. After a mean post-cystectomy follow-up of 3.8 years, only one recurrent case was observed, resulting in a verrucous cystic lesion that was considered premalignant after immunohistological examination. DNA extraction was successful from 14 of the 16 OOC cases. None of the primary OCCs or the single recurrent OOC were positive for HPV in the molecular analysis.

Conclusions

OOCs show a very limited potential for recurrence. Our results suggest that neither high- or low-risk HPV subtypes are likely to play a role in the etiology or neoplastic transformation of OOC, at least in the Spanish population.
Literature
1.
go back to reference Dong Q, Pan S, Sun L-S, Li T-J. Orthokeratinized odontogenic cyst. A clinicopathologic study of 61 cases. Arch Pathol Lab Med. 2010;134:271–5.PubMed Dong Q, Pan S, Sun L-S, Li T-J. Orthokeratinized odontogenic cyst. A clinicopathologic study of 61 cases. Arch Pathol Lab Med. 2010;134:271–5.PubMed
2.
go back to reference Schultz L. Cysts of the maxillae and mandibule. J Am Dent Assoc. 1927;14:1395–402. Schultz L. Cysts of the maxillae and mandibule. J Am Dent Assoc. 1927;14:1395–402.
3.
go back to reference Philipsen HP. Keratocystic odontogenic tumour in pathology and genetics of head and neck tumours. In: Barnes EL, Eveson JW, Reichart P, Sidransky D, editors. World Health Organization classification of Tumours. Lyon, France: IARC press; 2005. p. 306–7. Philipsen HP. Keratocystic odontogenic tumour in pathology and genetics of head and neck tumours. In: Barnes EL, Eveson JW, Reichart P, Sidransky D, editors. World Health Organization classification of Tumours. Lyon, France: IARC press; 2005. p. 306–7.
4.
go back to reference Wright JM. The odontogenic keratocyst: orthokeratinized variant. Oral Surg Oral Med Oral Pathol. 1981;51:609–18.CrossRefPubMed Wright JM. The odontogenic keratocyst: orthokeratinized variant. Oral Surg Oral Med Oral Pathol. 1981;51:609–18.CrossRefPubMed
5.
go back to reference Crowley TE, Kaugars GE, Gunsolley JC. Odontogenic keratocysts: a clinical and histologic comparison of the parakeratin and orthokeratin variants. J Oral Maxillofac Surg. 1992;50:22–6.CrossRefPubMed Crowley TE, Kaugars GE, Gunsolley JC. Odontogenic keratocysts: a clinical and histologic comparison of the parakeratin and orthokeratin variants. J Oral Maxillofac Surg. 1992;50:22–6.CrossRefPubMed
6.
go back to reference Da Silva MJ, de Sousa SOM, 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 Radiol Endod. 2002;94:732–7.CrossRef Da Silva MJ, de Sousa SOM, 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 Radiol Endod. 2002;94:732–7.CrossRef
7.
go back to reference Pimpalkar RD, Barpande SR, Bhavthankar ID, Mandale M. Bilateral orthokeratinized odontogenic cyst: a rare case report and review. Oral Maxillofac Pathol. 2014;18:262–6.CrossRef Pimpalkar RD, Barpande SR, Bhavthankar ID, Mandale M. Bilateral orthokeratinized odontogenic cyst: a rare case report and review. Oral Maxillofac Pathol. 2014;18:262–6.CrossRef
8.
go back to reference Cheng Y-SL, Liang H, Wright J, Teenier T. Multiple orthokeratinized odontogenic cysts. A case report. Head Neck Pathol. 2015;9:153–7.CrossRefPubMed Cheng Y-SL, Liang H, Wright J, Teenier T. Multiple orthokeratinized odontogenic cysts. A case report. Head Neck Pathol. 2015;9:153–7.CrossRefPubMed
9.
go back to reference MacDonald-Jankowski DS. Orthokeratinized odontogenic cyst: a systematic review. Dentomaxilloofacial Radiol. 2010;39:455–67.CrossRef MacDonald-Jankowski DS. Orthokeratinized odontogenic cyst: a systematic review. Dentomaxilloofacial Radiol. 2010;39:455–67.CrossRef
10.
go back to reference Yoshida H, Onizawa R, Yusa H. Squamous cell carcinoma arising in association with an orthokeratinized odontogenic keratocyst. Report of a case. J Oral Maxillofac Surg. 1996;54:647–51.CrossRefPubMed Yoshida H, Onizawa R, Yusa H. Squamous cell carcinoma arising in association with an orthokeratinized odontogenic keratocyst. Report of a case. J Oral Maxillofac Surg. 1996;54:647–51.CrossRefPubMed
11.
go back to reference Allon D, Kaplan I, Manor R, Calderon S. Carcinoma cuniculatum of the jaw: a rare variant of oral carcinoma. Oral Surg Oral Med Oral Pathol Radiol Endod. 2002;94:601–8. Allon D, Kaplan I, Manor R, Calderon S. Carcinoma cuniculatum of the jaw: a rare variant of oral carcinoma. Oral Surg Oral Med Oral Pathol Radiol Endod. 2002;94:601–8.
12.
go back to reference Pusiol T, Zorzi MG, Piscioli F. Squamous cell carcinoma arising in epidermal and human papillomavirus associated cysts: report of three cases. Pathologica. 2010;102:88–92.PubMed Pusiol T, Zorzi MG, Piscioli F. Squamous cell carcinoma arising in epidermal and human papillomavirus associated cysts: report of three cases. Pathologica. 2010;102:88–92.PubMed
13.
go back to reference Cox M, Eveson J, Scully C. Human papillomavirus type 16 DNA in an odontogenic keratocyst. J Oral Pathol Med. 1991;20:143–5.CrossRefPubMed Cox M, Eveson J, Scully C. Human papillomavirus type 16 DNA in an odontogenic keratocyst. J Oral Pathol Med. 1991;20:143–5.CrossRefPubMed
14.
go back to reference Knobler RM, Schneider S, Neumann RA, Bodemer W, Radlwimmer B, Aberer E, Söltz-Szöts J, Gebhart W. DNA dot-blot hybridization implicates human papillomavirus type 11-DNA in epithelioma cuniculatum. J Med Virol. 1989;29:33–7.CrossRefPubMed Knobler RM, Schneider S, Neumann RA, Bodemer W, Radlwimmer B, Aberer E, Söltz-Szöts J, Gebhart W. DNA dot-blot hybridization implicates human papillomavirus type 11-DNA in epithelioma cuniculatum. J Med Virol. 1989;29:33–7.CrossRefPubMed
15.
go back to reference Servato JPS, Cardoso SV, Da Silva MCP, Cordeiro MS, Faria PR, Loyola AM. Orthokeratinized odontogenic cysts presenting as a periapical lesion: report of a case and literature review. J Endod. 2014;40:455–8.CrossRef Servato JPS, Cardoso SV, Da Silva MCP, Cordeiro MS, Faria PR, Loyola AM. Orthokeratinized odontogenic cysts presenting as a periapical lesion: report of a case and literature review. J Endod. 2014;40:455–8.CrossRef
16.
go back to reference Aldred MJ, Talacko AA, Allan PG, Shear M. Odontogenic cyst with verrucous proliferation. J Oral Pathol Med. 2002;31:500–3.CrossRefPubMed Aldred MJ, Talacko AA, Allan PG, Shear M. Odontogenic cyst with verrucous proliferation. J Oral Pathol Med. 2002;31:500–3.CrossRefPubMed
17.
go back to reference Bodner L, Manor E, Shear M, van der Waal I. Primary intraosseous squamous cell carcinoma arising in an odontogenic cyst – a clinicopathologic analysis of 116 reported cases. J Oral Pathol Med. 2011;40:733–8.CrossRefPubMed Bodner L, Manor E, Shear M, van der Waal I. Primary intraosseous squamous cell carcinoma arising in an odontogenic cyst – a clinicopathologic analysis of 116 reported cases. J Oral Pathol Med. 2011;40:733–8.CrossRefPubMed
18.
go back to reference Brown RM, Gough NG. Malignant change in the epithelium lining odontogenic cysts. Cancer. 1972;29:1199–207.CrossRef Brown RM, Gough NG. Malignant change in the epithelium lining odontogenic cysts. Cancer. 1972;29:1199–207.CrossRef
19.
go back to reference Chaisuparat R, Coletti D, Kolokythas A, Ord RA, Nikitakis NG. Primary intraosseous odontogenic carcinoma arising in an odontogenic cyst or de novo: a clinicopathological study of six new cases. Oral Surg Oral Med Oral Pathol Radiol Endod. 2006;101:194–200.CrossRef Chaisuparat R, Coletti D, Kolokythas A, Ord RA, Nikitakis NG. Primary intraosseous odontogenic carcinoma arising in an odontogenic cyst or de novo: a clinicopathological study of six new cases. Oral Surg Oral Med Oral Pathol Radiol Endod. 2006;101:194–200.CrossRef
20.
go back to reference Fonseca FP, Pontes HAR, Pontes FSC, Carvalho PL, Sena-Filho M, Jorge J, Santos-Silva AR, De Almeida OP. Oral carcinoma cuniculatum: two cases illustrative of a diagnostic challenge. Oral Surg Oral Med Oral Pathol Oral Radiol. 2013;116:457–63.CrossRefPubMed Fonseca FP, Pontes HAR, Pontes FSC, Carvalho PL, Sena-Filho M, Jorge J, Santos-Silva AR, De Almeida OP. Oral carcinoma cuniculatum: two cases illustrative of a diagnostic challenge. Oral Surg Oral Med Oral Pathol Oral Radiol. 2013;116:457–63.CrossRefPubMed
21.
go back to reference Shiar CH, Ng KH. Squamous cell carcinoma in an orthokeratinised odontogenic keratocyst. Int J Oral Maxillofac Surg. 1987;16:95–8.CrossRef Shiar CH, Ng KH. Squamous cell carcinoma in an orthokeratinised odontogenic keratocyst. Int J Oral Maxillofac Surg. 1987;16:95–8.CrossRef
22.
go back to reference Yoshida H, Onizawa K, Yusa H. Squamous cell carcinoma arising in association with an orthokeratinized odontogenic keratocyst. J Oral Maxillofac Surg. 1996;54:647–51.CrossRefPubMed Yoshida H, Onizawa K, Yusa H. Squamous cell carcinoma arising in association with an orthokeratinized odontogenic keratocyst. J Oral Maxillofac Surg. 1996;54:647–51.CrossRefPubMed
23.
go back to reference Cox DP. P53 expression and mutation analysis of odontogenic cysts with and without dysplasia. Oral Surg Oral Med Oral Pathol Oral Radiol. 2012;113:90–8.CrossRefPubMed Cox DP. P53 expression and mutation analysis of odontogenic cysts with and without dysplasia. Oral Surg Oral Med Oral Pathol Oral Radiol. 2012;113:90–8.CrossRefPubMed
24.
go back to reference Argyris PP, Nelson AC, Koutlas IG. Keratinizing odontogenic cyst with verrucous pattern featuring negative human papillomavirus status by polymerase chain reaction. Oral Surg Oral Med Oral Pathol Oral Radiol. 2015;119:e233–40.CrossRefPubMed Argyris PP, Nelson AC, Koutlas IG. Keratinizing odontogenic cyst with verrucous pattern featuring negative human papillomavirus status by polymerase chain reaction. Oral Surg Oral Med Oral Pathol Oral Radiol. 2015;119:e233–40.CrossRefPubMed
25.
go back to reference Samman M, Wood H, Conway C, Berri S, Pentenero M, Gandolfo S, Cassenti A, Cassoni P, Al Ajlan A, Barrett AW, Chengot P, MacLennan K, High AS, Rabbitts P. Next-generation sequencing analysis for detecting human papillomavirus in oral verrucous carcinoma. Oral Surg Oral Med Oral Pathol Oral Radiol. 2014;118:117–25.CrossRefPubMed Samman M, Wood H, Conway C, Berri S, Pentenero M, Gandolfo S, Cassenti A, Cassoni P, Al Ajlan A, Barrett AW, Chengot P, MacLennan K, High AS, Rabbitts P. Next-generation sequencing analysis for detecting human papillomavirus in oral verrucous carcinoma. Oral Surg Oral Med Oral Pathol Oral Radiol. 2014;118:117–25.CrossRefPubMed
26.
go back to reference Del Pino M, Bleeker MC, Quint WG, Snijders PJ, Meijer CJ, Steenbergen RD. Comprehensive analysis of human papillomavirus prevalence and the potential role of low-risk types in verrucous carcinoma. Mod Pathol. 2012;10:1354–63.CrossRef Del Pino M, Bleeker MC, Quint WG, Snijders PJ, Meijer CJ, Steenbergen RD. Comprehensive analysis of human papillomavirus prevalence and the potential role of low-risk types in verrucous carcinoma. Mod Pathol. 2012;10:1354–63.CrossRef
27.
go back to reference Lalla K, Mahomed F, Meer S. Keratinizing odontogenic cysts with a spectrum of verrucoid morphology: investigation of a potential role of human papillomavirus. Oral Surg Oral Med Oral Pathol Oral Radiol. 2016;122:625–30.CrossRefPubMed Lalla K, Mahomed F, Meer S. Keratinizing odontogenic cysts with a spectrum of verrucoid morphology: investigation of a potential role of human papillomavirus. Oral Surg Oral Med Oral Pathol Oral Radiol. 2016;122:625–30.CrossRefPubMed
28.
go back to reference Hauck F, Oliveira-Silva M, Dreyer JH, Perrusi VJ, Arcuri RA, Hassan R, Bonvicino CR, Barros MH, Niedobitek G. Prevalence of HPV infection in head and neck carcinomas shows geographical variability: a comparative study from Brazil and Germany. Virchows Arch. 2015;466:685–93.CrossRefPubMed Hauck F, Oliveira-Silva M, Dreyer JH, Perrusi VJ, Arcuri RA, Hassan R, Bonvicino CR, Barros MH, Niedobitek G. Prevalence of HPV infection in head and neck carcinomas shows geographical variability: a comparative study from Brazil and Germany. Virchows Arch. 2015;466:685–93.CrossRefPubMed
Metadata
Title
Orthokeratinized odontogenic cysts: a Spanish tertiary care centre study based on HPV DNA detection
Authors
Beatriz Vera-Sirera
Luis Rubio-Martínez
Leopoldo Forner-Navarro
Francisco Vera-Sempere
Publication date
01-12-2018
Publisher
BioMed Central
Published in
Head & Face Medicine / Issue 1/2018
Electronic ISSN: 1746-160X
DOI
https://doi.org/10.1186/s13005-018-0167-3

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