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:: Volume 4, Issue 4 (Journal of Research in Dental & Maxillofacial Sciences Autumn 2019) ::
J Res Dentomaxillofac Sci 2019, 4(4): 43-48 Back to browse issues page
Use of Topical 5-Fluorouracil for Keratocystic Odontogenic Tumors of Gorlin-Goltz Syndrome as a New Targeted Therapy: A Case Report and Review of Literature
Hossein Shiran * 1, SH Shafaei Fard2, S Hakimi3, A Azari Pour Esfahani4
1- Postgraduate Student, Oral and Maxillofacial Surgery Dept, faculty of Dentistry, Tehran Medical Sciences, Islamic Azad University, Tehran, Iran , hossein.shiran@gmail.com
2- Associated Professor, Oral and Maxillofacial Surgery Dept , faculty of Dentistry, Tehran Medical Sciences,, Islamic Azad University, Tehran, Iran
3- Associated Professor , Oral and Maxillofacial Surgery Dept, faculty of Dentistry,Medical Sciences University of Zahedan,Iran
4- Postgraduate Student, Oral and Maxillofacial Surgery Dept,faculty of Dentistry, Tehran Medical Sciences,, Islamic Azad University, Tehran, Iran
Abstract:   (887 Views)
Background: Keratocystic odontogenic tumors (KOT) have a high rate of recurrence, which is higher in patients diagnosed with Gorlin-Goltz syndrome (GGS). Adjunctive therapies, such as fixative chemical solutions, decrease the rate of recurrence after enucleation and peripheral ostectomy but have high morbidity rates. Topical 5-Fluorouracil (5-FU) has been suggested as a new therapy that provides a directed molecular approach to treatment.
Case Presentation: This is a case report of GGS treated using topical 5-FU as an adjunctive material after enucleation and peripheral ostectomy. New bone formation sites were identified in the radiographic follow-up. The patient was followed up for 10 months regularly without any evidence of recurrence.
Conclusion: 5-FlU is an effective and novel targeted treatment for KOTs. Topical application of 5-FU, following
enucleation and peripheral ostectomy, effectively treats syndromic KOTs, resulting in normal bony healing with no adverse local or systemic effects.
Keywords: Odontogenic Tumors, Basal Cell Nevus Syndrome, Recurrence, 5-fluorouracil, salicylic acid drug combination [Supplementary Concept]
Full-Text [PDF 391 kb]   (498 Downloads) |   |   Full-Text (HTML)  (162 Views)  
Type of Study: Case report | Subject: Oral & maxillofacial surgery
* Corresponding Author Address: Islamic Azad University, Tehran, Iran
References
1. Evans DG, Ladusans EJ, Rimmer S, Burnell LD, Thakker N, Farndon PA. Complications of the naevoid basal cell carcinoma syndrome: results of a population based study. J Med Genet. 1993 Jun;30(6):460-4. [DOI:10.1136/jmg.30.6.460] [PMID] [PMCID]
2. Gu XM, Zhao HS, Sun LS, Li TJ. PTCH mutations in sporadic and Gorlin-syndrome-related odontogenic keratocysts. J Dent Res. 2006 Sep;85(9):859-63. [DOI:10.1177/154405910608500916] [PMID]
3. Acocella A, Sacco R, Bertolai R, Sacco N. Genetic and clinicopathologic aspects of Gorlin-Goltz syndrome (NBCCS): presentation of two case reports and literature review. Minerva Stomatol. 2009 Jan-Feb;58(1-2):43-53.
4. Pruvost-Balland C, Gorry P, Boutet N, Magnaldo T, Mamelle G, Margulis A, et al. [Clinical and genetic study in 22 patients with basal cell nevus syndrome]. [Article in French]. Ann Dermatol Venereol. 2006 Feb;133(2):117-23. [DOI:10.1016/S0151-9638(06)70861-4]
5. Kimonis VE, Goldstein AM, Pastakia B, Yang ML, Kase R, DiGiovanna JJ, et al. Clinical manifestations in 105 persons with nevoid basal cell carcinoma syndrome. Am J Med Genet. 1997 Mar 31;69(3):299-308. https://doi.org/10.1002/(SICI)1096-8628(19970331)69:3<299::AID-AJMG16>3.0.CO;2-M [DOI:10.1002/(SICI)1096-8628(19970331)69:33.0.CO;2-M]
6. Myoung H, Hong SP, Hong SD, Lee JI, Lim CY, Choung PH, et al. Odontogenic keratocyst: Review of 256 cases for recurrence and clinicopathologic parameters. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2001 Mar;91(3):328-33. [DOI:10.1067/moe.2001.113109] [PMID]
7. Dominguez FV, Keszler A. Comparative study of keratocysts, associated and non-associated with nevoid basal cell carcinoma syndrome. J Oral Pathol. 1988 Jan;17(1):39-42. [DOI:10.1111/j.1600-0714.1988.tb01503.x] [PMID]
8. Woolgar JA, Rippin JW, Browne RM. The odontogenic keratocyst and its occurrence in the nevoid basal cell carcinoma syndrome. Oral Surg Oral Med Oral Pathol. 1987 Dec;64(6):727-30. [DOI:10.1016/0030-4220(87)90176-9]
9. Stoelinga PJ. The treatment of odontogenic keratocysts by excision of the overlying, attached mucosa, enucleation, and treatment of the bony defect with carnoy solution. J Oral Maxillofac Surg. 2005 Nov;63(11):1662-6. [DOI:10.1016/j.joms.2005.08.007] [PMID]
10. Schmidt BL, Pogrel MA. The use of enucleation and liquid nitrogen cryotherapy in the management of odontogenic keratocysts. J Oral Maxillofac Surg. 2001 Jul;59(7):720-5. [DOI:10.1053/joms.2001.24278] [PMID]
11. Tolstunov L, Treasure T. Surgical treatment algorithm for odontogenic keratocyst: combined treatment of odontogenic keratocyst and mandibular defect with marsupialization, enucleation, iliac crest bone graft, and dental implants. J Oral Maxillofac Surg. 2008 May;66(5):1025-36. [DOI:10.1016/j.joms.2007.08.014] [PMID]
12. Stoelinga PJ. Excision of the overlying, attached mucosa, in conjunction with cyst enucleation and treatment of the bony defect with carnoy solution. Oral Maxillofac Surg Clin North Am. 2003 Aug;15(3):407-14. [DOI:10.1016/S1042-3699(03)00033-5]
13. Gosau M, Draenert FG, Müller S, Frerich B, Bürgers R, Reichert TE, et al. Two modifications in the treatment of keratocystic odontogenic tumors (KCOT) and the use of Carnoy's solution (CS)--a retrospective study lasting between 2 and 10 years. Clin Oral Investig. 2010 Feb;14(1):27-34. [DOI:10.1007/s00784-009-0264-6] [PMID]
14. Zhang L, Sun ZJ, Zhao YF, Bian Z, Fan MW, Chen Z. Inhibition of SHH signaling pathway: molecular treatment strategy of odontogenic keratocyst. Med Hypotheses. 2006;67(5):1242-4. [DOI:10.1016/j.mehy.2006.04.062] [PMID]
15. Booms P, Harth M, Sader R, Ghanaati S. Vismodegib hedgehog-signaling inhibition and treatment of basal cell carcinomas as well as keratocystic odontogenic tumors in Gorlin syndrome. Ann Maxillofac Surg. 2015 Jan-Jun;5(1):14-9. [DOI:10.4103/2231-0746.161049] [PMID] [PMCID]
16. Gross K, Kircik L, Kricorian G. 5% 5-Fluorouracil cream for the treatment of small superficial Basal cell carcinoma: efficacy, tolerability, cosmetic outcome, and patient satisfaction. Dermatol Surg. 2007 Apr;33(4):433-9. [DOI:10.1111/j.1524-4725.2007.33090.x] [PMID]
17. Papanastasopoulos P, Stebbing J. Molecular basis of 5-fluorouracil-related toxicity: lessons from clinical practice. Anticancer Res. 2014 Apr;34(4):1531-5.
18. Ledderhof NJ, Caminiti MF, Bradley G, Lam DK. Topical 5-Fluorouracil is a Novel Targeted Therapy for the Keratocystic Odontogenic Tumor. J Oral Maxillofac Surg. 2017 Mar;75(3):514-24. [DOI:10.1016/j.joms.2016.09.039] [PMID]
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shiran H, Shafaei Fard S, Hakimi S, Azari Pour Esfahani A. Use of Topical 5-Fluorouracil for Keratocystic Odontogenic Tumors of Gorlin-Goltz Syndrome as a New Targeted Therapy: A Case Report and Review of Literature. J Res Dentomaxillofac Sci. 2019; 4 (4) :43-48
URL: http://jrdms.dentaliau.ac.ir/article-1-255-en.html


Volume 4, Issue 4 (Journal of Research in Dental & Maxillofacial Sciences Autumn 2019) Back to browse issues page
Journal of Research in Dental and Maxillofacial Sciences
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