Head and Neck Pathology

, Volume 3, Issue 2, pp 150–152 | Cite as

Orthokeratinized Odontogenic Cyst of the Mandible with Heterotopic Cartilage

  • Fumio Ide
  • Ichiro Saito
  • Norio Horie
  • Tetsuo Shimoyama
Case Reports


Cartilaginous metaplasia is a rare but well-documented phenomenon occurring in the wall of odontogenic keratocyst. The mural cartilage not associated with odontogenic keratocyst has been reported only once in a maxillary teratoid cyst of congenital origin to our knowledge. A case presented is a 38-year-old man with intraosseous keratinizing epidermoid cyst in the mandible, the wall of which contained a nodule of mature hyaline cartilage. The present lesion likely represents a previously undescribed, histologic hybrid consisting of orthokeratinized odontogenic cyst and cartilaginous heterotopia.


Cartilage Heterotopia Mandible Orthokeratinized odontogenic cyst 


  1. 1.
    Shear M, Speight P. Cysts of the oral and maxillofacial regions. 4th ed. Oxford: Blackwell Munksgaard; 2007. p. 108–18.Google Scholar
  2. 2.
    Ide F, Horie N, Shimoyama T, Saito I, Tanaka A, Kusama K. Infrequent clinicopathologic features of keratocystic odontogenic tumor: a 29-year multi-institutional retrospective review. Oral Surg. 2009 (in press).Google Scholar
  3. 3.
    Arwill T, Kahnberg KE. Odontogenic keratocyst associated with an intramandibular chondroma. J Oral Surg. 1977;35:64–7.PubMedGoogle Scholar
  4. 4.
    Kratochvill FJ, Brannon RB. Cartilage in the walls of odontogenic keratocysts. J Oral Pathol Med. 1993;22:282–5. doi: 10.1111/j.1600-0714.1993.tb01072.x.CrossRefGoogle Scholar
  5. 5.
    Mosqueda-Taylor A, de la Piedra-Garza JM, Troncozo-Vazquez F. Odontogenic keratocyst with chondroid fibrous wall A case report. Int J Oral Maxillofac Surg. 1998;27:58–60. doi: 10.1016/S0901-5027(98)80098-2.PubMedCrossRefGoogle Scholar
  6. 6.
    Fornatora ML, Reich RF, Chotkowski G, Freedman PD. Odontogenic keratocyst with mural cartilaginous metaplasia: a case report and a review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2001;92:430–4. doi: 10.1067/moe.2001.117804.PubMedCrossRefGoogle Scholar
  7. 7.
    Yih WY, Krump JL. Odontogenic keratocyst in the nasopalatine duct associated with mural cartilaginous metaplasia. J Oral Maxillofac Surg. 2005;63:1382–4. doi: 10.1016/j.joms.2005.05.305.PubMedCrossRefGoogle Scholar
  8. 8.
    Pieritz U, Schubert GE. Zystisches Teratom im Oberkiefer. Dtsch Z Mund Kiefer Gesichtschir. 1984;8:260–3.PubMedGoogle Scholar
  9. 9.
    Wright JM. The odontogenic keratocyst: orthokeratinized variant. Oral Surg Oral Med Oral Pathol. 1981;51:609–18. doi: 10.1016/S0030-4220(81)80011-4.PubMedCrossRefGoogle Scholar
  10. 10.
    Siar CH, Ng KH. Orthokeratinised odontogenic keratocysts in Malaysians. Br J Oral Maxillofac Surg. 1988;26:215–20. doi: 10.1016/0266-4356(88)90165-9.PubMedCrossRefGoogle Scholar
  11. 11.
    Vuhahula E, Nikai H, Ijuhin N, et al. Jaw cysts with orthokeratinization: analysis of 12 cases. J Oral Pathol Med. 1993;22:35–40. doi: 10.1111/j.1600-0714.1993.tb00117.x.PubMedCrossRefGoogle Scholar
  12. 12.
    Li TJ, Kitano M, Chen XM, et al. Orthokeratinized odontogenic cyst: a clinicopathological and immunocytochemical study of 15 cases. Histopathology. 1998;32:242–51. doi: 10.1046/j.1365-2559.1998.00380.x.PubMedCrossRefGoogle Scholar
  13. 13.
    Noffke CEE. Implantation-type epidermoid cyst of the mandible. Dentomaxillofac Radiol. 1999;28:383–5. doi: 10.1038/sj.dmfr.4600477.PubMedCrossRefGoogle Scholar
  14. 14.
    Francis TC, Archard HO. Nasopalatine duct cyst with epidermoid features: report of case. J Oral Surg. 1967;25:265–8.Google Scholar
  15. 15.
    Bonder L, Woldenberg Y, Sion-Vardy N. Dermoid cyst of the maxilla. Int J Oral Maxillofac Surg. 2005;34:453–5. doi: 10.1016/j.ijom.2004.10.012.CrossRefGoogle Scholar
  16. 16.
    Chi AC, Neville BW, McDonald TA, Trayham RT, Byram J, Peacock EH. Jaw cysts with sebaceous differentiation: report of 5 cases and a review of the literature. J Oral Maxillofac Surg. 2007;65:2568–74. doi: 10.1016/j.joms.2006.09.014.PubMedCrossRefGoogle Scholar
  17. 17.
    Browne RM. The odontogenic keratocyst. Histological features and their correlation with clinical behavior. Br Dent J. 1971;131:249–59. doi: 10.1038/sj.bdj.4802732.PubMedCrossRefGoogle Scholar
  18. 18.
    Brannon RB. The odontogenic keratocyst. A clinicopathologic study of 312 cases. Part II. Histologic features. Oral Surg Oral Med Oral Pathol. 1977;43:233–55. doi: 10.1016/0030-4220(77)90161-X.PubMedCrossRefGoogle Scholar
  19. 19.
    Jordan RCK. Histology and ultrastructural features of the odontogenic keratocyst. Oral Maxillofac Surg Clin North Am. 2003;15:325–33. doi: 10.1016/S1042-3699(03)00034-7.PubMedCrossRefGoogle Scholar
  20. 20.
    Ide F, Horie N, Shimoyama T. Subpontic cartilaginous hyperplasia of the mandible. Oral Dis. 2003;9:224–5. doi: 10.1034/j.1601-0825.2003.02854.x.PubMedCrossRefGoogle Scholar
  21. 21.
    Everett FG, Bruckner RJ. Cartilage in the periodontal ligament space. J Periodontol. 1970;41:165–9.PubMedGoogle Scholar

Copyright information

© Humana 2009

Authors and Affiliations

  • Fumio Ide
    • 1
    • 2
  • Ichiro Saito
    • 1
  • Norio Horie
    • 2
  • Tetsuo Shimoyama
    • 2
  1. 1.Department of PathologyTsurumi University School of Dental MedicineYokohamaJapan
  2. 2.Department of Oral Surgery, Saitama Medical CenterSaitama Medical UniversityKawagoeJapan

Personalised recommendations