Maxillofacial Plastic and Reconstructive Surgery

Korean Association of Maxillofacial Plastic and Reconstructive Surgeons (대한악안면성형재건외과학회)
권호 표지

LONG TERM FOLLOW-UP OF MULTIPLE ODONTOGENIC KERATOCYSTS ASSOCIATED WITH BASAL CELL NEVUS SYNDROME: A CASE REPORT

기저세포모반 증후군과 관련된 다발성 낭종의 장기 치료결과: 증례보고

  • Lee, Eun-Young (Department of Oral & Maxillofacial Surgery, College of Medicine and Medical Research Institute, Chungbuk National University) ;
  • Kim, Kyoung-Won (Department of Oral & Maxillofacial Surgery, College of Medicine and Medical Research Institute, Chungbuk National University)
  • 이은영 (충북대학교 의과대학 구강악안면외과학교실, 의학연구소) ;
  • 김경원 (충북대학교 의과대학 구강악안면외과학교실, 의학연구소)
  • Received : 2009.07.21
  • Accepted : 2009.11.02
  • Published : 2010.01.29
Download PDF
⟨ Previous Next ⟩

Abstract

Multiple jaw cysts are one of the most constant features of the basal cell nevus syndrome. Basal cell nevus syndrome is inherited as an autosomal dominant trait with variable expressiveness. This syndrome comprises a number of abnormalities such as multiple nevoid basal cell carcinomas of the skin, skeletal abnormalities as bifid rib and fusion of vertebrae, central nervous system abnormalities as mental retardation, eye abnormalities with multiple jaw cysts. The odontogenic keratocysts in patients with this syndrome are often associated with the crowns of unerupted teeth and huge size; on radiographs they may mimic dentigerous cysts. The most important feature of the cyst is its extraordinary recurrence rate. Since recurrence may be long delayed in this lesion, follow-up of any case of odontogenic keratocyst with roentgenograms and clinical examination of basal cell carcinoma are essential for at least five years after surgery. We report the result of 7-year follow up after cyst enucleation associated with basal cell nevus syndrome with the literature of review.

Keywords

References

  1. Binkley GW, Johnson HH : Epithelioma Adenoids cystium: Basal cell nevi, Agenesis of the Corpus callosum and dental cysts. Ama Arch of Derm syph 63 : 73, 1951. https://doi.org/10.1001/archderm.1951.01570010076006
  2. Gorlin KJ, Goltz RW : Multiple nevoid basal cell epithelioma, jaw cysts and bifid rib: A syndrome. N Engl J Med 262 : 908, 1960. https://doi.org/10.1056/NEJM196005052621803
  3. Donatsky O, Hjorting-Hansen E : Recurrence of the odontogenic keratocyst in 13 patients with the nevoid basal cell carcinoma syndrome. lnt J Oral Surg 9 : 173, 1980. https://doi.org/10.1016/S0300-9785(80)80016-0
  4. Cha SK. Kim IK. Oh SS : Clinical study of cyst in the jaw. J Kor Oral Macillofac Surg 27 : 167, 2001
  5. Koutnik AW. Kolodny SC, Hoker SP et al : MUltiple nevoid basal cell epithelium, cyst of jaw, and bifid rib syndrome : report of case. J Oral Surg 33 : 686, 1975.
  6. Browne RM : The odontogenic keratocyst: Clinical aspects. Br Dent J 130 : 225. 1970.
  7. AhIfors E, Larsson a, Sjorgen S : The odontogenic keratocyst : A benign cystic tumor. J Oral Maxillofac Surg 42 : 10, 1984. https://doi.org/10.1016/0278-2391(84)90390-2
  8. Woolgar JA, Rippin JW, Browne RM : A comparative histological study of odontogenic keratocysts in basal cell nevus syndrome and control patients. J Oral Pathol 16 : 75, 1987. https://doi.org/10.1111/j.1600-0714.1987.tb00691.x
  9. Lim YS, Kim DR, Kim SG et al : Basal cell nevus syndrome: Review of 33 affected korean individuals. J Kor Aca Maxillofac Plast Reconst Surg 25 : 337, 2003.
  10. Clendrnning WE. Block JB. Radde IC et al : Basal cell nevus syndrome. Arch Derma 90 : 3814, 1964.
  11. Evans DG, Ladusans EJ, Rimmer S et al : Complication of the nevoid basal cell carcinoma syndrome : results of a population based study. J med Genet 30 : 460, 1993. https://doi.org/10.1136/jmg.30.6.460
  12. Stenman G, Magnusson B, Lennartsson B, et al : In vitro growth characteristics of human odontogenic keratocysts and dentigerous cysts. J Oral Pathol 15 : 143, 1986. https://doi.org/10.1111/j.1600-0714.1986.tb00595.x
  13. Stoelinga PJW, Peters JH : A note on the origin of keratocysts of the jaws. Int J Oral Surg 2 : 37, 1973. https://doi.org/10.1016/S0300-9785(73)80001-8
  14. Soskolne WA, Shear M : Observations on the pathogenesis of primordial cysts. Br Dent J 123 : 321, 1967.
  15. Vedtofte P, Praetorius F : Recurrence of the odontogenic ketatocyst in relation to clinical and histological features: A 20 year follow-up study of 72 patients. Int J Oral Surg 8 : 112, 1979.
  16. Williams TP, Connor FA : Surgical management of the odontogenic keratocyst: Aggressive approach. J Oral Maxillofac Surg 52 : 964, 1994. https://doi.org/10.1016/S0278-2391(10)80081-3
  17. Meilelman F : Surgical management of the odontogenic keratocyst: Conservative approach. J Oral Maxillofac Surg 52 : 960, 1994. https://doi.org/10.1016/S0278-2391(10)80080-1