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Multiple orthokeratinized odontogenic cysts: a case report.
Yi-Shing Lisa Cheng1, Hui Liang, John Wright
1Diagnostic Sciences, Texas A&M University-Baylor College of Dentistry, 3302 Gaston Ave, Dallas, TX, 75246, USA, ycheng@bcd.tamhsc.edu.
Head and Neck Pathology
|April 17, 2014
Summary
This report details the first case of multiple orthokeratinized odontogenic cysts (OOCs). The study found no association between OOCs and nevoid basal cell carcinoma syndrome (NBCCS).
Area of Science:
- Oral Pathology
- Oncology
- Genetics
Background:
- Multiple odontogenic keratocysts are a key feature of nevoid basal cell carcinoma syndrome (NBCCS).
- Loss of heterozygosity in the PTCH gene has been observed in sporadic orthokeratinized odontogenic cyst (OOC) cases.
- Investigating potential links between OOCs and NBCCS is crucial for understanding these conditions.
Observation:
- This report documents the clinical, radiographic, pathological, and molecular findings of the first documented case of multiple OOCs.
- The patient with multiple OOCs was evaluated against the diagnostic criteria for NBCCS.
- No clinical or molecular evidence of NBCCS was found in the patient presenting with multiple OOCs.
Findings:
- The presented case of multiple OOCs did not meet the diagnostic criteria for NBCCS.
- Molecular analysis did not reveal an association with NBCCS in this OOC case.
- These findings align with previous research suggesting OOCs are not typically associated with NBCCS.
Implications:
- Orthokeratinized odontogenic cysts (OOCs) appear to be distinct from the manifestations of nevoid basal cell carcinoma syndrome (NBCCS).
- Further research may clarify the specific genetic pathways and clinical significance of OOCs.
- This case contributes to the understanding of rare cystic lesions in oral pathology.

