Orphanet Journal of Rare Diseases | Treatment and Recurrence Analysis of Peripheral Jaw Cysts

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This study systematically reviews initial and recurrent cases of Peripheral Odontogenic Keratocysts (POKC) occurring in the buccal mucosa and provides long-term follow-up data. By analyzing two representative cases and literature review, it emphasizes the importance of microsurgical excision in POKC treatment.

 

Literature Review
The article "Clinical manifestations and treatment of peripheral odontogenic keratocysts: two cases and a literature review" published in Orphanet Journal of Rare Diseases summarizes two POKC cases occurring in the right buccal mucosa treated at Beijing Stomatological Hospital, along with clinical features, treatment methods and prognosis from previous literature. The article highlights that POKC's soft tissue location makes complete resection challenging, leading to high recurrence rates. Complete microsurgical excision significantly reduces recurrence risks.

Background Knowledge
Odontogenic keratocysts (OKC) are developmental odontogenic cysts with high recurrence rates (12%-50%), primarily occurring in jawbones, especially mandibles. Peripheral OKC (POKC) represents the soft tissue manifestation of OKC with extremely low incidence, commonly found in gingiva and buccal mucosa predominately affecting males. POKC shares similar pathological features with OKC but lacks standardized treatment protocols. Literature shows high recurrence rates after simple curettage/excision, while complete microsurgical resection offers better efficacy. POKC is associated with Gorlin-Goltz syndrome, with some cases showing PTCH1-GLI gene mutations. Due to its unique location and adhesion tendencies, treatment requires multidisciplinary collaboration. Current research lacks systematic approaches, necessitating international registry systems for accumulating clinical evidence.

 

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Research Methods and Experiments
The study included two POKC cases in right buccal mucosa treated at Beijing Stomatological Hospital from 2018-2020, both showing recurrence after initial surgeries. Secondary surgeries employed microsurgical complete excision removing cysts and adhesed soft tissues. Long-term follow-ups (37 and 16 months) showed no recurrence. Systematic literature review used Elsevier Journals, Web of Science, Wiley Online Library and PubMed databases with keywords "Peripheral odontogenic keratocyst", "Keratocystic odontogenic tumor" and "Review", screening 37 English articles documenting 51 POKC cases to analyze clinical features, treatment approaches and recurrence patterns.

Key Conclusions and Perspectives

• Both POKC patients experienced recurrence after initial surgeries, but no recurrence after secondary microsurgical complete excision.
• Among 51 cases, 29 (56.9%) occurred in gingiva, 14 (27.5%) in buccal mucosa, with remaining cases involving temporal muscles, temporomandibular joints, masseter muscles and pterygomandibular spaces.
• Treatment approaches include curettage, excision, cystectomy and microsurgery-assisted resection, with high recurrence rates (5/14) for simple curettage.
• POKC patients show average age of 54 years, primarily presenting with soft tissue swelling, some with pain, trismus or numbness.
• Microsurgical assistance improves resection accuracy, reduces tissue damage and significantly lowers recurrence rates.
• Postoperative follow-up should exceed 3 years for accurate treatment evaluation.

Research Significance and Future Directions
This study provides the longest follow-up data for POKC, detailing recurrence patterns and retreatment outcomes in buccal mucosa cases. It recommends microsurgical excision as first-line treatment for improved cure rates. Future efforts should establish international POKC registry systems to collect clinical data and explore optimal treatment strategies.

 

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Conclusion
This article summarizes two POKC cases in buccal mucosa with long-term follow-up results. Both cases showed recurrence after initial surgeries, but no recurrence after secondary microsurgical excision. Literature review reveals POKC has 20.6% overall recurrence rate (31.25% in gingiva, 9.1% in buccal mucosa). However, short follow-up periods in literature likely underestimate actual recurrence rates. The study emphasizes that simple curettage cannot completely eradicate POKC, while complete excision with microsurgical techniques effectively reduces recurrence. Long-term postoperative follow-up (≥3 years) is crucial for accurate efficacy evaluation. Future research should establish international POKC registry systems for systematic case collection and treatment strategy optimization.

 

Yu Huang, Lirui Zhang, Xiaohong Yuan, and Zhien Feng. Clinical manifestations and treatment of peripheral odontogenic keratocysts: two cases and a literature review. Orphanet Journal of Rare Diseases.