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Sociodemographic and clinical characterization of cases of 1,103 non-syndromic and 66 syndromic odontogenic keratocyst: a Brazilian multicenter study

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Abstract

Objectives

This multicenter study aimed to evaluate cases of non-syndrome and syndromic odontogenic keratocyst, as well as cases of recurrence within these two groups.

Methods

This descriptive, analytical, retrospective cross-sectional study evaluated the sex, age and presence of multiple lesions in 1,169 individuals seen at 10 Brazilian oral and maxillofacial pathology centers. Of these, 1,341 odontogenic keratocysts were analyzed regarding clinical diagnosis, size, site, imaging appearance, signs and symptoms, type of biopsy, treatment, and recurrence.

Results

There was a similar distribution by sex. The median age of non-syndromic and syndromic patients was 32 and 17.5 years, respectively. The posterior mandible was the site most affected by small and large lesions in both groups and in recurrent cases. Unilocular lesions were more frequent, also in recurrent cases. Mainly small lesions showed this imaging appearance. Signs and symptoms were absent in most cases. Conservative treatment was the most frequent modality in all age groups, regardless of the patient’s condition and recurrence. Recurrences were uncommon.

Conclusion

This study showed a higher frequency of non-syndromic keratocysts in the population. Clinicopathological features related to the involvement of multiple sites, age, and recurrence may differ between syndromic and non-syndromic cases. Furthermore, we found an association between lesion size and some clinical features and between the time interval to recurrence and the syndromic spectrum.

Clinical relevance

To contribute to a better understanding of the distribution and association between clinical, imaging, and sociodemographic characteristics in each spectrum of the lesion.

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Data Availability

Data from this study can be provided upon request with justification to the corresponding author.

References

  1. Philipsen HP (1956) Om keratocyster (kolesteatom) i kaeberne. Tandlaege Bladet 60:963–981

    Google Scholar 

  2. El-Naggar A et al (2017) WHO Classification of Head and Neck Tumours. World. Health Organization, 4th edition. Lyon: IARC PRESS, 347 p

  3. Schuch LF, Arruda JAA, Mosconi C et al (2020) A Brazilian multicentre study of 2,497 isolated cases of odontogenic keratocysts. Oral Dis 26(3):711–715. https://doi.org/10.1111/odi.13278

    Article  PubMed  Google Scholar 

  4. Boffano P, Cavarra F, Agnone AM et al (2022) The epidemiology and management of odontogenic keratocysts (OKCs): A European multicenter study. J Craniomaxillofac Surg 50(1):1–6. https://doi.org/10.1016/j.jcms.2021.09.022

    Article  PubMed  Google Scholar 

  5. Santana DCP, Garcia JJ, Kusterer LEF et al (2021) Odontogenic keratocyst: eight-year follow-up after conservative treatment. Int J Odontostomat 15(2):520–525. https://doi.org/10.4067/S0718-381X2021000200520

    Article  Google Scholar 

  6. Azevedo RS, Cabral MG, dos Santos TC, de Oliveira AV, de Almeida OP, Pires FR (2012) Histopathological features of keratocystic odontogenic tumor: a descriptive study of 177 cases from a Brazilian population. Int J Surg Pathol 20(2):154–160. https://doi.org/10.1177/1066896911428072

    Article  PubMed  Google Scholar 

  7. WHO Classification of Tumors Editorial Board. Head and neck tumors. Lyon (France): International Agency for Research on Cancer, 2022. (WHO classification of tumors series, 5th ed.; vol.9)

  8. Kimonis VE, Goldstein AM, Pastakia B et al (1997) Clinical Manifestations in 105 persons with nevoid basal cell carcinoma syndrome. Am J Med Genet 69:299–308

    Article  PubMed  Google Scholar 

  9. Kauke M, Safi AF, Kreppe M, Grandoch A, Nickenig HJ, Zöller JE, Dreiseidler T (2018) Size distribution and clinicoradiological signs of aggressiveness in odontogenic myxoma- three-dimensional analysis and systematic review. Dentomaxillofac Radiol 47(2):20170262. https://doi.org/10.1259/dmfr.20170262

    Article  PubMed  Google Scholar 

  10. Brannon RB (1976) The odontogenic keratocyst. A clinicopathologic study of 312 cases. Part I: Clinical features. Oral Surg Oral Med Oral Pathol 42(1):54–72. https://doi.org/10.1016/0030-4220(76)90031-1

    Article  PubMed  Google Scholar 

  11. Myoung H, Hong SP, Hong SD et al (2001) Odontogenic keratocyst: review of 256 cases for recurrence and clinicopathologic parameters. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 91(3):328–333. https://doi.org/10.1067/moe.2001.113109

    Article  PubMed  Google Scholar 

  12. El Murtadi A, Grehan D, Toner M, McCartan BE (1996) Proliferating cell nuclear antigen staining in syndrome and nonsyndrome odontogenic keratocysts. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 81(2):217–220. https://doi.org/10.1016/s1079-2104(96)80418-5

    Article  PubMed  Google Scholar 

  13. Hammannavar R, Holikatti K, Basappa S, Shinde N, Reddy M, Chidambaram YS (2014) Multiple, multifocal odontogenic keratocyst in non-syndromic patient: a case-report. J Oral Health Dent Manag 13(2):89–93

    Google Scholar 

  14. Janjua OS, Tariq R, Khalid UM, Qureshi SM, Kamran A (2022) Presentation and management of syndromic and non-syndromic patients with multiple odontogenic keratocysts. Oral Maxil Surg Cases 8(2):100253. https://doi.org/10.1016/j.omsc.2022.100253

    Article  Google Scholar 

  15. Payne TF (1972) An analysis of the clinical and histopathologic parameters of the odontogenic keratocyst. Oral Surg Oral Med Oral Pathol 33(4):538–546. https://doi.org/10.1016/0030-4220(72)90366-0

    Article  PubMed  Google Scholar 

  16. Chirapathomsakul D, Sastravaha P, Jansisyanont P (2006) A review of odontogenic keratocysts and the behavior of recurrences. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 101(1):5–9. https://doi.org/10.1016/j.tripleo.2005.03.023

    Article  PubMed  Google Scholar 

  17. Borghesi A, Nardi C, Gianitto C et al (2018) Odontogenic keratocyst : imaging features of a benign lesion with an aggressive behaviour. Insights Imaging 9(2):883–897. https://doi.org/10.1007/s13244-018-0644-z

    Article  PubMed  PubMed Central  Google Scholar 

  18. Pinheiro JC, Cavalcante IL, Santos HBP, Santos PPA, de Souza LB (2020) Recurrence rate of odontogenic keratocysts: Clinical-radiographic characterization throughout a 48-year period. Rev Port Estomatol Med Dent Cir Maxilofac 61(2):52–56. https://doi.org/10.24873/j.rpemd.2020.09.705

    Article  Google Scholar 

  19. Vallejo-Rosero KA, Camolesi GV, de Sá PLD, Bernaola-Paredes WE (2020) Conservative management of odontogenic keratocyst with long-term 5-year follow-up: case report and literature review. Int J Surg Case Rep 66:8–15. https://doi.org/10.1016/j.ijscr.2019.11.023

    Article  PubMed  Google Scholar 

  20. Donatsky O, Hjörting-Hansen E, Philipsen HP, Fejerskov O (1976) Clinical, radiologic, and histopathologic aspects of 13 cases of nevoid basal cell carcinoma syndrome. Int J Oral Surg 5(1):19–28. https://doi.org/10.1016/S0300-9785(76)80006-3

    Article  PubMed  Google Scholar 

  21. Woolgar JA, Rippin JW, Browne RM (1987) The odontogenic keratocyst and its occurrence in the nevoid basal cell carcinoma syndrome. Oral Surg Oral Med Oral Pathol 64(6):727–730. https://doi.org/10.1016/0030-4220(87)90176-9

    Article  PubMed  Google Scholar 

  22. Browne RM (1975) The pathogenesis of odontogenic cysts: a review. J Oral Pathol 4(1):31–46. https://doi.org/10.1111/j.1600-0714.1975.tb01738.x

    Article  PubMed  Google Scholar 

  23. Johnson NR, Batstone MD, Savage NW (2013) Management and recurrence of keratocystic odontogenic tumor: a systematic review. Oral Surg Oral Med Oral Pathol Oral Radiol 116(4):271–276. https://doi.org/10.1016/j.oooo.2011.12.028

    Article  Google Scholar 

  24. Mendonça JCG, Jardim ECG, da Silva JCL, Valente FRF, dos Santos MES (2022) Surgical treatment of odontogenic keratocyst associated with cryotherapy with refrigerant gas: 10- year follow-up. Res Soc Develop 11(3):e40511326784. https://doi.org/10.33448/rsd-v11i3.26784

    Article  Google Scholar 

  25. Yilmaz O, Sagnak Yilmaz Z, Balaban E, Candirli C (2020) Management of recurrence of ameloblastoma and odontogenic keratocyst: a cross-sectional study. Odovtos-Int J Dent Sci 22(3):174–186. https://doi.org/10.15517/ijds.2020.41564

    Article  Google Scholar 

  26. Forssell K (1980) The primordial cyst. Clinical and radiographic study. Proc Finn Dent Soc 77(5):129–174

    Google Scholar 

  27. Mesquita PMV (2001) Queratocisto Odontogênico. Uma Entidade Clínico- Radiográfica de Diagnóstico Histológico. Dissertation, University of Porto

  28. Habibi A, Saghravanian N, Habibi M, Mellati E, Habibi M (2007) Keratocystic odontogenic tumor: a 10-year retrospective study of 83 cases in an Iranian population. J Oral Sci 49(3):229–235. https://doi.org/10.2334/josnusd.49.229

    Article  PubMed  Google Scholar 

  29. Ianculovici C, Blinder D, Peleg O (2016) Trends in recurrence of keratocystic odontogenic tumor. Int J Oral Dent Health 2:031. https://doi.org/10.23937/2469-5734/1510031

    Article  Google Scholar 

  30. Warburton G, Shihabi A, Ord RA (2015) Keratocystic odontogenic tumor (KCOT/OKC)— clinical guidelines for resection. J Maxillofac Oral Surg 14(3):558–564. https://doi.org/10.1007/s12663-014-0732-7

    Article  PubMed  PubMed Central  Google Scholar 

  31. Baik WK, Baik HS, Choi SH (2020) Comprehensive orthodontic treatment of a young girl with an odontogenic keratocyst and impacted teeth in the mandible. Korean J Orthod 50(1):63–71. https://doi.org/10.4041/kjod.2020.50.1.63

    Article  PubMed  PubMed Central  Google Scholar 

Download references

Acknowledgements

The authors thank the state funding agency Fundação de Amparo à Pesquisa do Estado da Bahia (FAPESB), the Coordenação de Aperfeiçoamento de Pessoal de Nível Superior (CAPES), Conselho Nacional de Desenvolvimento Científico e Tecnológico (CNPq) and the partner institutions that collaborated with the study.

Funding

This study was funded by Fundação de Amparo à Pesquisa do Estado da Bahia (FAPESB), the Coordenação de Aperfeiçoamento de Pessoal de Nível Superior (CAPES), Conselho Nacional de Desenvolvimento Científico e Tecnológico (CNPq) which granted fellowships to the students participating in this study.

Author information

Authors and Affiliations

  1. School of Dentistry, Postgraduation Program in Dentistry and Health, Federal University of Bahia, Salvador, Brazil

    Daiana Cristina Pereira Santana & Larissa Abbehusen Couto

  2. Department of Social and Pediatric Dentistry, School of Dentistry da Federal, Postgraduation Program in Dentistry and Health, University of Bahia, Salvador, Brazil

    Johelle de Santana Passos-Soares

  3. Surgical Pathology Service, School of Dentistry, Postgraduation Program in Dentistry and Health, Federal University of Bahia, Salvador, Brazil

    Flávia Caló de Aquino Xavier, Jean Nunes dos Santos & Águida Cristina Gomes Henriques

  4. Department of Dental Clinical Practice, Pathology and Surgery, School of Dentistry, Federal University of Minas Gerais, Belo Horizonte, Brazil

    Ricardo Alves Mesquita, Fernanda Aragão Felix & Tarcília Aparecida da Silva

  5. Department of Dentistry, State University of Paraíba, Campina Grande, Brazil

    Cassiano Francisco Weege Nonaka, Pollianna Muniz Alves & Christany Rodrigues Ferreira

  6. Department of Pathology and Forensic Medicine (DPML), Federal University of Amazonas, Manaus, Brazil

    Tatiana Nayara Libório-Kimura, Lucileide Castro de Oliveira Moreira, Jeconias Câmara & Caroline Alfaia Silva

  7. Laboratory of Oral Pathology, School of Dentistry, Federal University of Goiás, Goiânia, Brazil

    Elismauro Francisco de Mendonça & Nádia do Lago Costa

  8. Oral Pathology Service, João de Barros, Barreto University Hospital, Federal University of Pará, Belém, Brazil

    Hélder Antônio Rebelo Pontes & Lucas Lacerda de Souza

  9. Department of Clinical and Preventive Dentistry, Federal University of Pernambuco, Recife, Brazil

    Jurema Freire de Castro Lisboa, Gustavo Pina Godoy & Laís Azevedo Lins de Holanda

  10. Department of Oral Pathology, School of Dentistry, Federal University of Rio Grande Do Sul, Porto Alegre, Brazil

    Manoela Domingues Martins, Pantelis Varvaki Rados & Tuany Rafaeli Schmidt

  11. Postgraduate Program in Dental Sciences, Federal University of Rio Grande Do Norte, Natal, Brazil

    Roseana de Almeida Freitas, Lélia Batista de Souza & Everton Freitas de Morais

  12. Department of Oral Diagnosis, Piracicaba Dental School, State University of Campinas, Piracicaba, Brazil

    Pablo Agustin Vargas, Márcio Ajudarte Lopes & Gabriella Alves Quixabeira Oliveira

Authors
  1. Daiana Cristina Pereira Santana
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  2. Larissa Abbehusen Couto
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  3. Johelle de Santana Passos-Soares
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  4. Flávia Caló de Aquino Xavier
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  8. Cassiano Francisco Weege Nonaka
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  12. Lucileide Castro de Oliveira Moreira
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  14. Caroline Alfaia Silva
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  15. Elismauro Francisco de Mendonça
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  16. Nádia do Lago Costa
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  17. Hélder Antônio Rebelo Pontes
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  18. Lucas Lacerda de Souza
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  19. Jurema Freire de Castro Lisboa
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  20. Gustavo Pina Godoy
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  21. Laís Azevedo Lins de Holanda
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  22. Manoela Domingues Martins
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  23. Pantelis Varvaki Rados
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  24. Tuany Rafaeli Schmidt
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  25. Roseana de Almeida Freitas
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  26. Lélia Batista de Souza
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  27. Everton Freitas de Morais
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  28. Pablo Agustin Vargas
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  29. Márcio Ajudarte Lopes
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  30. Gabriella Alves Quixabeira Oliveira
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  31. Jean Nunes dos Santos
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  32. Águida Cristina Gomes Henriques
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Contributions

All authors whose names appear on the submission.

1) made substancial contributions to the concept or design of the work; or the acquisition, analysis, or interpretation of data;

2) drafeted the work or revised it critically for important intellectual content;

3) approved the version to be published; and.

4) agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.

Corresponding author

Correspondence to Águida Cristina Gomes Henriques.

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All procedures involving human participants were conducted in accordance with the ethical standards of the institutional and national committees (number 52704421.8.1001.5024) and with the 1964 Helsinki Declaration and its later amendments or comparable ethical standards.

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 The authors declare no competing interests.

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Pereira Santana, D.C., Abbehusen Couto, L., de Santana Passos-Soares, J. et al. Sociodemographic and clinical characterization of cases of 1,103 non-syndromic and 66 syndromic odontogenic keratocyst: a Brazilian multicenter study. Clin Oral Invest 27, 6951–6959 (2023). https://doi.org/10.1007/s00784-023-05313-7

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