Introduction and importance: The association between Dentigerous cysts (DCs) and Gorlin-Goltz syndrome (GGS) was claimed theoretically in a very few reports, with very few clinical foundations. The aim of this report was to present a unique case of multiple DCs in the mandible in a patient showing features of GGS.
Case presentation: A 63-year-old male patient presented with multiple cyst-like lesions in the mandible associated with some clinical and radiological features of GGS, and that raised the suspension of odontogenic keratocyst (OKC). The patient underwent marsupialization and enucleation of these cysts, and the histopathological examination confirmed the diagnosis of DCs.
Clinical discussion: In this report, the patient presented with symptoms related to multiple unilocular radiolucent lesions found in the mandible and the clinical and radiological features were highly suggestive of OKCs associated with GGS. However, the perioperative findings raised the suspicion of DCs, which was confirmed by histopathology. Interestingly, GGS is an inherited autosomal dominant disorder arising from mutations in the patched tumor suppressor gene (PTCH). Previous studies showed this gene alteration in DCs; this can possibly be implicated in the pathogenesis of the association found in this report.
Conclusion: This report presented a case of bilateral DC in the mandible in a patient showing features of GGS. Therefore, this report verified the very rare association between DC and GGS. This may help dentists and physicians in reaching an accurate and early diagnosis of GGS.
Keywords: Case report; Dentigerous cyst; Gorlin-Goltz syndrome; Third molar.
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