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AuthorOmer Sefvan, Janjua
AuthorTariq, Ramish
AuthorKhalid, Muhammad Usman
AuthorQureshi, Sana Mehmood
AuthorAli, Kamran
Available date2022-04-04T07:54:18Z
Publication Date2022-06-30
Publication NameOral and Maxillofacial Surgery Cases
Identifierhttp://dx.doi.org/10.1016/j.omsc.2022.100253
CitationJanjua, O. S., Tariq, R., Khalid, M. U., Qureshi, S. M., & Ali, K. (2022). Presentation and management of syndromic and non-syndromic patients with multiple odontogenic keratocysts. Oral and Maxillofacial Surgery Cases, 8(2), 100253.
ISSN22145419
URIhttps://www.sciencedirect.com/science/article/pii/S2214541922000128
URIhttp://hdl.handle.net/10576/29275
AbstractObjectiveThis case series reports the clinical presentation and management of multiple odontogenic keratocysts (OKCs) in patients with Gorlin Goltz syndrome and in non-syndromic patients. Material and methodsEight patients presented with features of multiple odontogenic keratocyst at department of Oral and Maxillofacial Surgery. The diagnosis was confirmed following clinical, radiographic and histopathological examination. Initially the diagnosis of OKC was confirmed on an incisional biopsy. Major and minor criteria were followed for the diagnosis of Gorlin Goltz syndrome. Smaller cysts in all patients were enucleated and for larger cysts marsupialization was planned either alone or followed by enucleation. Patients were followed at 1 month, 3 months, 6 months and yearly interval to check for bone healing and recurrences. ResultsAssociation of Gorlin Goltz syndrome was identified in four patients all of whom were males with age range of 12–37 years. Among four patients that had non-syndromic OKCs, three were female and one was male with age range of 09–50 years; two patients had familial non-syndromic multiple OKCs. In syndromic patients, multiple OKCs, recognized manifestations of Gorlin Goltz syndrome were identified with variable frequency: calcification of falx cerebri and chest deformity (100%); macrocephaly with frontal bossing (100%); hypertelorism (75%); basal cell carcinoma (25%); pectus deformity with flame shaped hands and feet (25%) and syndactyly (50%). Palmar or plantar pits, cleft lip or palate, ovarian fibroma and medulloblastoma was not identified in any patient. In patients with non-syndromic multiple OKCs mandible was more commonly involved than maxilla. ConclusionPatients with multiple OKCs, should be evaluated thoroughly and basal cell carcinomatous lesions should be ruled out. Meticulous follow up is vital as Gorlin Goltz syndrome is associated with malignancies and OKCs may be the first manifestation of this syndrome. Given the fact that OKCs associated with this syndrome have higher rate of recurrence than the isolated OKCs, long term follow up is warranted.
Languageen
PublisherElsevier
SubjectGorlin Goltz syndrome
Odontogenic
Keratocyst
Nevoid basal cell carcinoma syndrome
TitlePresentation and management of syndromic and non-syndromic patients with multiple odontogenic keratocysts
TypeArticle
Issue Number2
Volume Number8
Open Access user License http://creativecommons.org/licenses/by-nc-nd/4.0/
dc.accessType Open Access


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