Recurrent peripheral odontogenic keratocyst: Review of the literature and presentation of a novel case initially masquerading as an atypical infected lateral periodontal cyst

John K Brooks; Ahmed S Sultan; Martha P Rabkin; John M Sands; Dwayne E Everett; Jeffery B Price; John R Basile

doi:10.1016/j.jormas.2023.101540

Recurrent peripheral odontogenic keratocyst: Review of the literature and presentation of a novel case initially masquerading as an atypical infected lateral periodontal cyst

J Stomatol Oral Maxillofac Surg. 2023 Jun 15:101540. doi: 10.1016/j.jormas.2023.101540. Online ahead of print.

Authors

John K Brooks¹, Ahmed S Sultan², Martha P Rabkin³, John M Sands⁴, Dwayne E Everett⁵, Jeffery B Price⁶, John R Basile⁷

Affiliations

¹ Clinical Professor, Department of Oncology and Diagnostic Sciences, University of Maryland School of Dentistry, Baltimore, MD 21201, USA. Electronic address: jbrooks@umaryland.edu.
² Assistant Professor, Department of Oncology and Diagnostic Sciences, University of Maryland School of Dentistry, and Member, University of Maryland Marlene and Stewart Greenebaum Comprehensive Cancer Center, Baltimore, Maryland 21201, USA.
³ Formerly, Predoctoral student, University of Maryland School of Dentistry, Baltimore, Maryland 21201, At Present, Dentist, United States Navy, Norfolk, Virginia 23511, USA.
⁴ Senior Resident, Department of Oral and Maxillofacial Surgery, University of Maryland School of Dentistry, Baltimore, Maryland 21201, USA.
⁵ Clinical Instructor, Department of Oral and Maxillofacial Surgery, University of Maryland School of Dentistry, Baltimore, Maryland 21201, USA.
⁶ Clinical Professor, Director of Oral and Maxillofacial Radiology, Department of Oncology and Diagnostic Sciences, University of Maryland School of Dentistry, Baltimore, Maryland 21201, USA.
⁷ Associate Professor, Department of Oncology and Diagnostic Sciences, University of Maryland School of Dentistry, Baltimore, Maryland, Member, Molecular and Structural Biology Branch, The Marlene and Stewart Greenebaum Center, Baltimore, Maryland 21201, USA.

PMID: 37327876
DOI: 10.1016/j.jormas.2023.101540

Abstract

Aim: To review published cases and case series of the peripheral odontogenic keratocyst (POKC) of the gingiva, report an unusual presentation, and discuss lesional recurrence.

Materials and methods: A search of the English language literature for gingival OKCs was conducted. The inclusion of new case yielded a database containing 29 affected patients. Clinical, surgical, radiographic, and histopathologic findings have been summarized.

Results: With available patient demographics, 62.5% were female and 37.5% were male, with an overall mean age at diagnosis of 53.8 years. There was near-equal lesional affinity for the jaws, of which 44.0% occurred in the posterior region, 32.0% anteriorly, and 24.0% overlapped these areas. Twenty-five percent of lesions had a normal color, 30.0% appeared yellow, 20.0% were white, and 10.0% were blue. The majority of lesions were < 1 cm and nearly 42% manifested exudation or fluctuance. Lesional pain was infrequent. Pressure resorption was recorded in 45.8% of cases. Most lesions were managed with conservative surgical modalities. Follow-up information was available in 16 primary cases, of which 5 recurred, signifying a 31.3% recurrence rate, including the featured case, which recurred twice.

Conclusion: To reduce recurrence of a gingival OKC, supraperiosteal dissection is advocated. Further, it is advised to follow POKCs for 5-7 years postoperatively, remaining vigilant for subtle clinical manifestations of recurrence. Timely discovery and excision of a POKC of the gingiva may decrease the incidence of a mucogingival defect.

Keywords: Atypical; Lateral periodontal cyst; Maxillary; Peripheral odontogenic keratocyst; Recurrent; Review.