Authors :
Sarra Azzez; Hela Zouaghi; Chaima Haj Hassine; Chiraz Baccouche
Volume/Issue :
Volume 11 - 2026, Issue 10 - October
DOI :
https://doi.org/10.38124/ijisrt/26Oct560
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Abstract :
The odontogenic keratocyst (OKC) is a benign but locally aggressive intraosseous lesion with a high recurrence rate and a well-established association with Gorlin–Goltz syndrome. In young patients with Gorlin–Goltz syndrome and extensive mandibular involvement, radical resection can cause severe functional and aesthetic morbidity, prompting interest in conservative, bone-sparing alternatives. Previous reports have described decompression and staged enucleation as effective strategies for reducing lesion volume and enabling less morbid definitive surgery, yet evidence in syndromic cases with bilateral or multilocular mandibular disease remains limited. This case report describes the outcome of a two-stage conservative approach in an 18-year-old man with Gorlin–Goltz syndrome and extensive multilocular mandibular OKCs at high risk of pathological fracture. The scope includes clinical, radiographic, and histopathological assessment, decompression with a custom stent, and secondary enucleation with peripheral ostectomy. The novelty lies in achieving complete resolution of one compartment and marked volumetric reduction of the other, enabling nerve-sparing surgery without neurovascular deficit or fracture, thereby supporting decompression as a viable first-line option in selected syndromic patients.
Keywords :
Odontogenic Keratocyst; Gorlin-Goltz Syndrome; Nevoid Basal Cell Carcinoma Syndrome; Decompression; Mandible; Bone Regeneration.
References :
- Philipsen HP. On keratocysts in the jaws. Tandlaegebladet. 1956;60:963-971.
- Pindborg JJ, Hansen J. Studies on odontogenic cyst epithelium. 2. Clinical and roentgenologic aspects of odontogenic keratocysts. Acta Pathol Microbiol Scand. 1963;58:283-294.
- Wright JM, Vered M. Update from the 4th edition of the World Health Organization classification of head and neck tumours: odontogenic and maxillofacial bone tumors. Head Neck Pathol. 2017;11(1):68-77.
- Soluk-Tekkesin M, Wright JM. (2002). The World Health Organization Classification of Odontogenic Lesions: A Summary of the Changes of the 2022 (5th) Edition. Turk Patoloji Derg. ;38(2):168-184. doi: 10.5146/tjpath.2022.01573.
- Al-Moraissi EA, Kaur A, Gomez RS, Ellis E 3rd. Effectiveness of different treatments for odontogenic keratocyst: a network meta-analysis. Int J Oral Maxillofac Surg. 2023;52(1):32-43.
- Nayak MT, Singh A, Singhvi A, Sharma R. Odontogenic keratocyst: What is in the name? J Nat Sci Biol Med. 2013 Jul;4(2):282-5. doi: 10.4103/0976-9668.116968. PMID: 24082717; PMCID: PMC3783765.
- Freddo AL, Oliveira HT, Furlanetto AP, Heidrich J, Corsetti A. Decompression as conservative treatment of multiple odontogenic keratocysts in a patient with Gorlin-Goltz syndrome: 6-year follow-up. Braz J Oral Sci. 2026;25:e262079.
- Stanbouly D, Lee KC, Fazzolari JR, Philipone E. Decompression of odontogenic keratocysts before enucleation reduces the rate of recurrence. J Craniofac Surg. 2022;33(6):1806-1808.
- He J, Wang H, Zeng J, Zhou L. Large mandibular odontogenic keratocyst treated by decompression and secondary enucleation: a case report. J Clin Pediatr Dent. 2024;48(6):213-220.
- Kim SH, Oh MS, Seo YS, Kim JY, Nam SH, Lim SM. Conservative Treatment of Multiple Keratocystic Odontogenic Tumors in a Young Patient with Nevoid Basal Cell Carcinoma Syndrome by Decompression: A 7-year Follow-up Study. J Clin Pediatr Dent. 2017;41(4):300-306.
- Jungwirth MT, Ziccardi VB. Decompression of Mandibular Odontogenic Keratocyst Using A Custom Drain: A Technical Note. Dent Oral Biol Craniofac Res. 2019;3(1):1-4.
- Kumar NN, Padmashree S, Jyotsna TR, Shastry SP. Gorlin-Goltz Syndrome: A Rare Case Report. Contemp Clin Dent. 2018;9(3):478-483.
- Albuquerque LV, Medeiros YL, Santos LM, Pereira KAM, Andrade BAB, Marlière DAA. Conservative surgical approach for mandibular odontogenic keratocyst associated with guided tissue regeneration: a case report. J Oral Diagn. 2026;11:e0434.
- Kwon YJ, Ko KS, So BK, Kim DH, Jang HS, Kim SH, et al. Effect of Decompression on Jaw Cystic Lesions Based on Three-Dimensional Volumetric Analysis. Medicina (Kaunas). 2020;56(11):602.
The odontogenic keratocyst (OKC) is a benign but locally aggressive intraosseous lesion with a high recurrence rate and a well-established association with Gorlin–Goltz syndrome. In young patients with Gorlin–Goltz syndrome and extensive mandibular involvement, radical resection can cause severe functional and aesthetic morbidity, prompting interest in conservative, bone-sparing alternatives. Previous reports have described decompression and staged enucleation as effective strategies for reducing lesion volume and enabling less morbid definitive surgery, yet evidence in syndromic cases with bilateral or multilocular mandibular disease remains limited. This case report describes the outcome of a two-stage conservative approach in an 18-year-old man with Gorlin–Goltz syndrome and extensive multilocular mandibular OKCs at high risk of pathological fracture. The scope includes clinical, radiographic, and histopathological assessment, decompression with a custom stent, and secondary enucleation with peripheral ostectomy. The novelty lies in achieving complete resolution of one compartment and marked volumetric reduction of the other, enabling nerve-sparing surgery without neurovascular deficit or fracture, thereby supporting decompression as a viable first-line option in selected syndromic patients.
Keywords :
Odontogenic Keratocyst; Gorlin-Goltz Syndrome; Nevoid Basal Cell Carcinoma Syndrome; Decompression; Mandible; Bone Regeneration.
