Main Article Content
Abstract
Introduction: The possibility of transformation of the epithelium lining the cavity of dentigerous cyst to a neoplastic one has been recognized for a number of cases, but it is difficult to establish that such an event has actually occurred. So, the current review aims to implement a systematic review to identify the potential demographic, clinical and radiological characteristics of the malignant transformation that arose in DC. Material and method: The review was carried out per the Preferred Reporting Items for Systematic Reviews and Meta-analyses (PRISMA). 50 articles were reviewed and reported 55 cases with neoplastic changes originated from dentigerous cysts. Result: The demographic and clinical characteristics of the included cases showed that the mean age of the affected patients was 44.04 ±23.36 years, ranged from 1.33 to 84 years. Thirty-three patients (60%) were males. Most of the reported neoplasms were in mandible (72.7%). Primary sites of these neoplasms were the posterior regions (35, 63.6%), with majority of cases recorded in molar areas (26, 47.3%), while the incisor region affected less frequently (5, 9.1%). Radiographically, the majority of neoplasm appeared as unilocular radiolucency (39, 70.9%), while multilocular radiolucency reported in 2 cases only (3.6%). Histopathologically, malignant transformations were noticed in 40 cases (72.7%) and the majority of them were squamous cell carcinoma (35, 63.6%) mostly of well differentiated type (25, 45.5%), provided that 20 cases (36.4%) were not described regarding histological differentiation. On the other hand, adenomatoid odontogenic tumor represented as most benign lesion associated with the dentigerous cyst (10, 18.2% of all neoplasms and 66.7% of the benign lesions). Most of the neoplasm cases were reported in India (19, 34.5%) and were of Asian race (26, 47.3%). Conclusion: Although most studies report that neoplastic transformation rarely occurs in the lining of the dentigerous cyst but unfortunately, the malignancies occur with higher frequency than do the benign tumors.
Keywords
Article Details
Copyright (c) 2025 Ali H. Murad, Muna. S Merza, Maha Waleed Alghazali (Author)

This work is licensed under a Creative Commons Attribution 4.0 International License.
How to Cite
References
- Aboul-Hosn Centenero S, et al. Primary intraosseous carcinoma and odontogenic cyst: three new cases and review of the literature. Med Oral Patol Oral Cir Bucal. 2006;11(1):E61–5.
- Aggarwal P, Saxena S. Aggressive growth and neoplastic potential of dentigerous cysts with particular reference to central mucoepidermoid carcinoma. Br J Oral Maxillofac Surg. 2011;49(6):e36–9.doi: 10.1016/j.bjoms.2010.11.016
- Angelopoulos AP, et al. Malignant transformation of the epithelial lining of the odontogenic cysts: report of a case. Oral Surg Oral Med Oral Pathol. 1966;22(4):415–28.
- Araujo JP, et al. Malignant transformation of an odontogenic cyst over a period of 10 years. Case Rep Dent. 2014;2014:762969.doi: 10.1155/2014/762969
- Berenholz L, et al. Squamous cell carcinoma arising in a dentigerous cyst. Ear Nose Throat J. 1988;67(10):764–8.
- Bhushan NS, et al. Ameloblastoma arising from a dentigerous cyst: a case report. J Clin Diagn Res. 2014;8(5):ZD23–5.doi: 10.7860/JCDR/2014/8593.4392
- Bradfield WJ, Broadway ES. Malignant change in a dentigerous cyst. Br J Surg. 1958;45(194):657–9.
- Bradley N, et al. Squamous cell carcinoma arising in an odontogenic cyst. Int J Oral Maxillofac Surg. 1988;17(4):260–3.
- Cankurtaran CZ, et al. Ameloblastoma and dentigerous cyst associated with impacted mandibular third molar tooth. Radiographics. 2010;30(5):1415–20.doi: 10.1148/rg.305095115
- Charles M, et al. Primary intraosseous malignancy originating in an odontogenic cyst in a young child. J Oral Maxillofac Surg. 2008;66(4):813–9.doi: 10.1016/j.joms.2007.06.663
- Chen YK, et al. Adenomatoid odontogenic tumor arising from a dentigerous cyst: a case report. Int J Pediatr Otorhinolaryngol Extra. 2007;2:257–63.
- Chretien PB, et al. Squamous carcinoma arising in a dentigerous cyst: presentation of a fatal case and review of four previously reported cases. Oral Surg Oral Med Oral Pathol. 1970;30(6):809–16.
- Colbert S, et al. Squamous cell carcinoma arising in dentigerous cysts. J Craniomaxillofac Surg. 2012;40(8):e3557.doi: https://doi.org/10.1016/j.jcms.2012.01.020
- Copete MA, et al. Squamous carcinoma arising from a dentigerous cyst: report of a case. Compend Contin Educ Dent. 1996;17(2):202–4.
- Deshmane S, Khot K. Unicystic ameloblastoma arising from a dentigerous cyst: a case report. Int J Oral Health Med Res. 2016;3:38–40.
- Gay-Escoda C, et al. Primary intraosseous squamous cell carcinoma arising in dentigerous cyst: report of two cases and review of the literature. J Clin Exp Dent. 2015;7(5):e665–70.doi:10.4317/jced.52689
- Geetha NT, Upasi AP, Rai K. Adenomatoid odontogenic tumor arising from dentigerous cyst: report of a rare case. Oral Health Sci. 2014;4:93–6.
- Gulbranson SH, et al. Squamous cell carcinoma arising in a dentigerous cyst in a 16-month-old girl. Otolaryngol Head Neck Surg. 2002;127(5):463–4.doi: 10.1067/mhn.2002.129039
- Jain M, Mittal S, Gupta DK. Primary intraosseous squamous cell carcinoma arising in odontogenic cysts: insight into pathogenesis. J Oral Maxillofac Surg. 2013;71(1):e7–14.doi: 10.1016/j.joms.2012.08.031
- Jayanandan M, et al. Desmoplastic ameloblastoma arising in a dentigerous cyst: a case report. J Clin Diagn Res. 2016;10(8):ZD38–40.doi: 10.7860/JCDR/2016/20013.8362
- John JB, John RR. Adenomatoid odontogenic tumor associated with dentigerous cyst in posterior maxilla: a case report and review of literature. J Oral Maxillofac Pathol. 2010;14(2):59–62.
- Johnson LM, Sapp JP, McIntire DN. Squamous cell carcinoma arising in a dentigerous cyst. J Oral Maxillofac Surg. 1994;52(9):987–90.
- Kalburge JV, et al. Neoplasms associated with dentigerous cyst: insight into pathogenesis and clinicopathologic features. Arch Med Health Sci. 2015;3:309–13.
- Kramer HS, Scribner JH. Squamous cell carcinoma arising in a dentigerous cyst: report of a case. Oral Surg Oral Med Oral Pathol. 1965;19:555–61.
- Lapin R, et al. Squamous cell carcinoma arising in a dentigerous cyst. J Oral Surg. 1973;31(5):354–8.
- Latti BR, Kalburge JV. Dentigerous cyst associated with adenomatoid odontogenic tumor: a rare case report. Med Sci. 2013;1:44–7.
- Lee KW, Loke SJ. Squamous cell carcinoma arising in a dentigerous cyst. Cancer. 1967;20(12):2241–4.
- Manganaro AM, Cross SE, Startzell JM. Carcinoma arising in a dentigerous cyst with neck metastasis. Head Neck. 1997;19(5):436–9.
- Manjunatha BS, et al. Adenomatoid odontogenic tumor arising from a dentigerous cyst. J Maxillofac Oral Surg. 2015;14(2):393–7.
- Maxymiw WG, Wood RE. Carcinoma arising in a dentigerous cyst: a case report and review of the literature. J Oral Maxillofac Surg. 1991;49(6):639–43.
- Moosvi Z, Tayaar SA, Kumar GS. Neoplastic potential of odontogenic cysts. Contemp Clin Dent. 2011;2(2):106–9.doi: 10.4103/0976-237x.83073
- Nagarajappa AK, Pandya D, Jain N. Ameloblastoma arising from dentigerous cyst: an unusual case. J Orofac Res. 2015;5:146–50.
- Oshimata A, et al. Squamous cell carcinoma arising in a dentigerous cyst. Jpn J Oral Maxillofac Surg. 2002;48:530–3.
- Peng CY, et al. Intraosseous verrucous carcinoma arising from an infected dentigerous cyst. J Formos Med Assoc. 2015;114(8):764–8.doi: 10.1016/j.oooo.2012.08.430
- Roofe SB, et al. Squamous cell carcinoma arising in the epithelial lining of a dentigerous cyst. South Med J. 1999;92(6):611–4.
- Sandhu SV, et al. Adenomatoid odontogenic tumor associated with dentigerous cyst of the maxillary antrum. J Oral Maxillofac Pathol. 2010;14(1):24–8.
- Scheer M, et al. Primary intraosseous carcinoma of the jaws arising from an odontogenic cyst. J Craniomaxillofac Surg. 2004;32(3):166–9.doi: 10.1016/j.jcms.2004.03.003
- Shukla S, et al. Squamous cell carcinoma arising in a dentigerous cyst: a case report. World J Pathol. 2014;6.
- Singh V, et al. Adenomatoid odontogenic tumor with dentigerous cyst. Contemp Clin Dent. 2012;3(Suppl 2):S244–7.doi: 10.4103/0976-237X.105107
- Spoorthi BR, et al. Predominantly cystic central mucoepidermoid carcinoma developing from a dentigerous cyst. Clin Pract. 2013;3(2):e19.
- Uchida K, et al. Primary intraosseous odontogenic carcinoma arising from a dentigerous cyst. J Hard Tissue Biol. 2013;22:375–82.doi: 10.32388/igs2oc
- Van der Waal I, et al. Squamous cell carcinoma arising in the lining of odontogenic cysts. Int J Oral Surg. 1985;14:146–52.
- Williams IE, Newman CW. Squamous cell carcinoma associated with a dentigerous cyst of the maxilla. Oral Surg Oral Med Oral Pathol. 1963;16:1012–6.
- Yasuoka T, et al. Squamous cell carcinoma arising in a dentigerous cyst. J Oral Maxillofac Surg. 2000;58(8):900–5.doi: 10.1002/1097-0142(196712)20:12<2241::aid-cncr2820201226>3.0.co;2-f
- Zapala-Pospiech A, et al. Malignant transformation of a dentigerous cyst. Pol J Pathol. 2013;64(1):64–8.
- Bodner L, et al. Primary intraosseous squamous cell carcinoma arising in odontogenic cysts: analysis of 116 cases. J Oral Pathol Med. 2011;40(10):733–8.doi: 10.1111/j.1600-0714.2011.01058.x
- Dubiel-Bigaj M. Odontogenic cysts: histogenesis, histology and neoplastic potential. Patol Pol. 1993;44(2):45–54.doi: 10.4103/0976-237x.83073
- Gardner AF. The odontogenic cyst as a potential carcinoma. J Am Dent Assoc. 1969;78(4):746–55.
- Schwimmer AM, Aydin F, Morrison SN. Squamous cell carcinoma arising in residual odontogenic cyst. Oral Surg Oral Med Oral Pathol. 1991;72(2):218–21.
- Foley WL, Terry BC, Jacoway JR. Malignant transformation of an odontogenic keratocyst. J Oral Maxillofac Surg. 1991;49(7):768–71.
- Borras-Ferreres J, Sanchez-Torres A, Gay-Escoda C. Malignant changes developing from odontogenic cysts: a systematic review. J Clin Exp Dent. 2016;8(5):e622–8.doi: /10.4317/jced.53256
- Pavelic B, et al. PTCH gene alterations in dentigerous cysts. J Oral Pathol Med. 2001;30(9):569–76.
- da Silva Baumgart C, et al. Epidermal growth factor receptor distribution in pericoronal follicles. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2007;103(2):240–5.doi: 10.1016/j.tripleo.2005.11.009
- Gardner AF. A survey of odontogenic cysts and their relationship to squamous cell carcinoma. Dent J. 1975;41(3):161–7.
- Zhang LL, et al. Dentigerous cyst: a retrospective clinicopathological analysis of 2082 cases. Int J Oral Maxillofac Surg. 2010;39(9):878–82.doi: 10.1016/j.ijom.2010.04.048
- Li N, et al. Prevalence of developmental odontogenic cysts in children and adolescents. Acta Odontol Scand. 2014;72(8):795–800.doi: /10.3109/00016357.2014.913192
- Neville BW, Damm DD, Allen CM. Odontogenic cysts and tumors. In: Gnepp DR, editor. Diagnostic Surgical Pathology of the Head and Neck. China: Elsevier; 2009. p. 785–838.
References
Aboul-Hosn Centenero S, et al. Primary intraosseous carcinoma and odontogenic cyst: three new cases and review of the literature. Med Oral Patol Oral Cir Bucal. 2006;11(1):E61–5.
Aggarwal P, Saxena S. Aggressive growth and neoplastic potential of dentigerous cysts with particular reference to central mucoepidermoid carcinoma. Br J Oral Maxillofac Surg. 2011;49(6):e36–9.doi: 10.1016/j.bjoms.2010.11.016
Angelopoulos AP, et al. Malignant transformation of the epithelial lining of the odontogenic cysts: report of a case. Oral Surg Oral Med Oral Pathol. 1966;22(4):415–28.
Araujo JP, et al. Malignant transformation of an odontogenic cyst over a period of 10 years. Case Rep Dent. 2014;2014:762969.doi: 10.1155/2014/762969
Berenholz L, et al. Squamous cell carcinoma arising in a dentigerous cyst. Ear Nose Throat J. 1988;67(10):764–8.
Bhushan NS, et al. Ameloblastoma arising from a dentigerous cyst: a case report. J Clin Diagn Res. 2014;8(5):ZD23–5.doi: 10.7860/JCDR/2014/8593.4392
Bradfield WJ, Broadway ES. Malignant change in a dentigerous cyst. Br J Surg. 1958;45(194):657–9.
Bradley N, et al. Squamous cell carcinoma arising in an odontogenic cyst. Int J Oral Maxillofac Surg. 1988;17(4):260–3.
Cankurtaran CZ, et al. Ameloblastoma and dentigerous cyst associated with impacted mandibular third molar tooth. Radiographics. 2010;30(5):1415–20.doi: 10.1148/rg.305095115
Charles M, et al. Primary intraosseous malignancy originating in an odontogenic cyst in a young child. J Oral Maxillofac Surg. 2008;66(4):813–9.doi: 10.1016/j.joms.2007.06.663
Chen YK, et al. Adenomatoid odontogenic tumor arising from a dentigerous cyst: a case report. Int J Pediatr Otorhinolaryngol Extra. 2007;2:257–63.
Chretien PB, et al. Squamous carcinoma arising in a dentigerous cyst: presentation of a fatal case and review of four previously reported cases. Oral Surg Oral Med Oral Pathol. 1970;30(6):809–16.
Colbert S, et al. Squamous cell carcinoma arising in dentigerous cysts. J Craniomaxillofac Surg. 2012;40(8):e3557.doi: https://doi.org/10.1016/j.jcms.2012.01.020
Copete MA, et al. Squamous carcinoma arising from a dentigerous cyst: report of a case. Compend Contin Educ Dent. 1996;17(2):202–4.
Deshmane S, Khot K. Unicystic ameloblastoma arising from a dentigerous cyst: a case report. Int J Oral Health Med Res. 2016;3:38–40.
Gay-Escoda C, et al. Primary intraosseous squamous cell carcinoma arising in dentigerous cyst: report of two cases and review of the literature. J Clin Exp Dent. 2015;7(5):e665–70.doi:10.4317/jced.52689
Geetha NT, Upasi AP, Rai K. Adenomatoid odontogenic tumor arising from dentigerous cyst: report of a rare case. Oral Health Sci. 2014;4:93–6.
Gulbranson SH, et al. Squamous cell carcinoma arising in a dentigerous cyst in a 16-month-old girl. Otolaryngol Head Neck Surg. 2002;127(5):463–4.doi: 10.1067/mhn.2002.129039
Jain M, Mittal S, Gupta DK. Primary intraosseous squamous cell carcinoma arising in odontogenic cysts: insight into pathogenesis. J Oral Maxillofac Surg. 2013;71(1):e7–14.doi: 10.1016/j.joms.2012.08.031
Jayanandan M, et al. Desmoplastic ameloblastoma arising in a dentigerous cyst: a case report. J Clin Diagn Res. 2016;10(8):ZD38–40.doi: 10.7860/JCDR/2016/20013.8362
John JB, John RR. Adenomatoid odontogenic tumor associated with dentigerous cyst in posterior maxilla: a case report and review of literature. J Oral Maxillofac Pathol. 2010;14(2):59–62.
Johnson LM, Sapp JP, McIntire DN. Squamous cell carcinoma arising in a dentigerous cyst. J Oral Maxillofac Surg. 1994;52(9):987–90.
Kalburge JV, et al. Neoplasms associated with dentigerous cyst: insight into pathogenesis and clinicopathologic features. Arch Med Health Sci. 2015;3:309–13.
Kramer HS, Scribner JH. Squamous cell carcinoma arising in a dentigerous cyst: report of a case. Oral Surg Oral Med Oral Pathol. 1965;19:555–61.
Lapin R, et al. Squamous cell carcinoma arising in a dentigerous cyst. J Oral Surg. 1973;31(5):354–8.
Latti BR, Kalburge JV. Dentigerous cyst associated with adenomatoid odontogenic tumor: a rare case report. Med Sci. 2013;1:44–7.
Lee KW, Loke SJ. Squamous cell carcinoma arising in a dentigerous cyst. Cancer. 1967;20(12):2241–4.
Manganaro AM, Cross SE, Startzell JM. Carcinoma arising in a dentigerous cyst with neck metastasis. Head Neck. 1997;19(5):436–9.
Manjunatha BS, et al. Adenomatoid odontogenic tumor arising from a dentigerous cyst. J Maxillofac Oral Surg. 2015;14(2):393–7.
Maxymiw WG, Wood RE. Carcinoma arising in a dentigerous cyst: a case report and review of the literature. J Oral Maxillofac Surg. 1991;49(6):639–43.
Moosvi Z, Tayaar SA, Kumar GS. Neoplastic potential of odontogenic cysts. Contemp Clin Dent. 2011;2(2):106–9.doi: 10.4103/0976-237x.83073
Nagarajappa AK, Pandya D, Jain N. Ameloblastoma arising from dentigerous cyst: an unusual case. J Orofac Res. 2015;5:146–50.
Oshimata A, et al. Squamous cell carcinoma arising in a dentigerous cyst. Jpn J Oral Maxillofac Surg. 2002;48:530–3.
Peng CY, et al. Intraosseous verrucous carcinoma arising from an infected dentigerous cyst. J Formos Med Assoc. 2015;114(8):764–8.doi: 10.1016/j.oooo.2012.08.430
Roofe SB, et al. Squamous cell carcinoma arising in the epithelial lining of a dentigerous cyst. South Med J. 1999;92(6):611–4.
Sandhu SV, et al. Adenomatoid odontogenic tumor associated with dentigerous cyst of the maxillary antrum. J Oral Maxillofac Pathol. 2010;14(1):24–8.
Scheer M, et al. Primary intraosseous carcinoma of the jaws arising from an odontogenic cyst. J Craniomaxillofac Surg. 2004;32(3):166–9.doi: 10.1016/j.jcms.2004.03.003
Shukla S, et al. Squamous cell carcinoma arising in a dentigerous cyst: a case report. World J Pathol. 2014;6.
Singh V, et al. Adenomatoid odontogenic tumor with dentigerous cyst. Contemp Clin Dent. 2012;3(Suppl 2):S244–7.doi: 10.4103/0976-237X.105107
Spoorthi BR, et al. Predominantly cystic central mucoepidermoid carcinoma developing from a dentigerous cyst. Clin Pract. 2013;3(2):e19.
Uchida K, et al. Primary intraosseous odontogenic carcinoma arising from a dentigerous cyst. J Hard Tissue Biol. 2013;22:375–82.doi: 10.32388/igs2oc
Van der Waal I, et al. Squamous cell carcinoma arising in the lining of odontogenic cysts. Int J Oral Surg. 1985;14:146–52.
Williams IE, Newman CW. Squamous cell carcinoma associated with a dentigerous cyst of the maxilla. Oral Surg Oral Med Oral Pathol. 1963;16:1012–6.
Yasuoka T, et al. Squamous cell carcinoma arising in a dentigerous cyst. J Oral Maxillofac Surg. 2000;58(8):900–5.doi: 10.1002/1097-0142(196712)20:12<2241::aid-cncr2820201226>3.0.co;2-f
Zapala-Pospiech A, et al. Malignant transformation of a dentigerous cyst. Pol J Pathol. 2013;64(1):64–8.
Bodner L, et al. Primary intraosseous squamous cell carcinoma arising in odontogenic cysts: analysis of 116 cases. J Oral Pathol Med. 2011;40(10):733–8.doi: 10.1111/j.1600-0714.2011.01058.x
Dubiel-Bigaj M. Odontogenic cysts: histogenesis, histology and neoplastic potential. Patol Pol. 1993;44(2):45–54.doi: 10.4103/0976-237x.83073
Gardner AF. The odontogenic cyst as a potential carcinoma. J Am Dent Assoc. 1969;78(4):746–55.
Schwimmer AM, Aydin F, Morrison SN. Squamous cell carcinoma arising in residual odontogenic cyst. Oral Surg Oral Med Oral Pathol. 1991;72(2):218–21.
Foley WL, Terry BC, Jacoway JR. Malignant transformation of an odontogenic keratocyst. J Oral Maxillofac Surg. 1991;49(7):768–71.
Borras-Ferreres J, Sanchez-Torres A, Gay-Escoda C. Malignant changes developing from odontogenic cysts: a systematic review. J Clin Exp Dent. 2016;8(5):e622–8.doi: /10.4317/jced.53256
Pavelic B, et al. PTCH gene alterations in dentigerous cysts. J Oral Pathol Med. 2001;30(9):569–76.
da Silva Baumgart C, et al. Epidermal growth factor receptor distribution in pericoronal follicles. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2007;103(2):240–5.doi: 10.1016/j.tripleo.2005.11.009
Gardner AF. A survey of odontogenic cysts and their relationship to squamous cell carcinoma. Dent J. 1975;41(3):161–7.
Zhang LL, et al. Dentigerous cyst: a retrospective clinicopathological analysis of 2082 cases. Int J Oral Maxillofac Surg. 2010;39(9):878–82.doi: 10.1016/j.ijom.2010.04.048
Li N, et al. Prevalence of developmental odontogenic cysts in children and adolescents. Acta Odontol Scand. 2014;72(8):795–800.doi: /10.3109/00016357.2014.913192
Neville BW, Damm DD, Allen CM. Odontogenic cysts and tumors. In: Gnepp DR, editor. Diagnostic Surgical Pathology of the Head and Neck. China: Elsevier; 2009. p. 785–838.
