Two-stage treatment protocol of keratocystic odontogenic tumour in young patients with Gorlin–Goltz syndrome: Marsupialization and later enucleation with peripheral ostectomy. A 5-year-follow-up experience

Borgonovo, Andrea Enrico; Lascia, Stefano Di; Grossi, Giovanni Battista; Maiorana, Carlo

doi:10.1016/j.ijporl.2011.09.009

Cited by 21 publications

(22 citation statements)

References 36 publications

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“…It is linked with mutation in the PTCH gene. Partial expression of the gene may result in occurrence of only multiple-recurring OKC Our patient presented with nine cysts with multiple recurrences over a period of 11 years without any other manifestation of the syndrome Borgonovo et al [56] Case report…”

Section: Description Of Nbccs Resultsmentioning

confidence: 85%

Possible recurrence of keratocyst in nevoid basal cell carcinoma syndrome: A review of literature

et al. 2013

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This review will highlight some current areas of difficulty or controversy in diagnosis and treatment of nevoid basal cell carcinoma syndrome (NBCCS). The odontogenic keratocyst (OKC) has significant growth capacity and recurrence potential and is occasionally indicative of the NBCCS. The objective of this study is to clarify the causes of the recurrence of OKC in NBCCS. A literature search was conducted using Medline, accessed via the National Library of Medicine PubMed interface, searching for articles relating to the cause of recurrence of keratocyst in NBCCS written in English. This study has described the previous and the current outcomes of the treatment of OKC (recurrent cause). A protocol was then agreed to search for the possible causes of keratocyst recurrence in NBCCS. The general treatment of other manifestation of NBCCS has excluded from this study. Studies describing cohort, case series and miscellaneous clinical reports were retrieved and evaluated from 2010 to 2012.

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Section: Description Of Nbccs Resultsmentioning

confidence: 85%

Possible recurrence of keratocyst in nevoid basal cell carcinoma syndrome: A review of literature

et al. 2013

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“…Marsupialization is a surgical technique by which a window is produced in the wall of the cyst to relieve the intra-cystic pressure and so enable the cavity to decrease slowly in size, then complete enucleation will be performed as a second-stage procedure to reduce recurrence rate (22). To date, a large amount of literature has proved that marsupialization is an effective method for treating large odontogenic cysts (23)(24)(25). However, this period ranges from 6 to 14 months on average, which seems too long because of the limited osteogenic capacity of jaw (26).…”

Section: Discussionmentioning

confidence: 99%

Activation of Mesenchymal Stem Cells Promotes New Bone Formation Within Dentigerous Cyst

Zhou

et al. 2020

Preprint

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Background: Dentigerous cyst (DC) is a bone destructive disease and remains a challenge for clinicians. Marsupialization enables bone to regenerate with capsules maintaining, making it a preferred therapeutic means for DC adjacent to vital anatomical structures. Given that capsules of DC derive from odontogenic epithelium remnants at embryonic stage, we investigated whether there were mesenchymal stem cells (MSCs) located in DC capsules and the role that they played in the bone regeneration after marsupialization.Methods: Samples obtained before and after marsupialization were used for histological detection and cell culture. The stemness of cells isolated from fresh tissues were analyzed by morphology, surface marker and multi-differentiation assays. Comparison of proliferation ability between Am-DCSCs and Bm-DCSCs were evaluated by Cell Counting Kit-8 (CCK-8), fibroblast colony-forming units (CFU-F) and 5’‐ethynyl‐2’‐deoxyuridine (EdU) assay. Their osteogenic capacity in vitro was detected by Alkaline phosphatase (ALP) and Alizarin Red staining (ARS), combined with Real-time polymerase chain reaction (RT-PCR) and immunofluorescence (IF) staining. Subcutaneous ectopic osteogenesis as well as cranial bone defect model in nude mice were performed to detect their bone regeneration and bone defect repair ability.Results: Bone tissue and strong ALP activity were detected in the capsule of DC after marsupialization. Two types of MSCs were isolated from fibrous capsules of DC both before (Bm-DCSCs) and after (Am-DCSCs) marsupialization. These fibroblast-like, colony forming cells expressed MSC markers (CD44+, CD90+, CD31-, CD34-, CD45-), and they could differentiate into osteoblast-, adipocyte- and chondrocyte-like cells under induction. Notably, Am-DCSCs performed better in cell proliferation and self-renewal. Moreover, Am-DCSCs showed greater osteogenic capacity both in vitro and in vivo compared with Bm-DCSCs. Conclusions: There are MSCs residing in capsules of DC, and the cell viability as well as osteogenic capacity of them are largely enhanced after marsupialization. Our findings suggested that MSCs might play a crucial role in the healing process of DC after marsupialization, thus providing new insight into the treatment for DC by promoting the osteogenic differentiation of MSCs inside capsules.

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“…In fact the recurrence rate of KOT in different studies varies in a wide range from 0 to 100 % that is related to the number of evaluated cases, various follow-up periods and different treatment techniques [14][15][16]. KOT associated with the nevoid basal cell carcinoma syndrome compared to non-syndromic lesions are usually more extensive in size and numbers and occur in younger ages [7]. Also recurrence rate in KOTs associated with the nevoid basal cell carcinoma syndrome is much higher than non-related syndrome lesions, in which the reported recurrence rate is about 82 % [17].…”

Section: Discussionmentioning

confidence: 99%

“…KOT is one of the most common complications of Gorlin's syndrome that is found at least in 75 % of patients [6]. Nevoid basal cell carcinoma syndrome associated KOTs in comparison with nonsyndromic KOTs are usually more extensive and more numerous and recognized in younger ages [7]. Considering the extension of syndromic lesions, emotional status of children, growth of jaws and involvement of permanent tooth buds, choosing the most appropriate treatment is so challenging.…”

Section: Introductionmentioning

confidence: 99%

Conservative Treatment Protocol of Keratocystic Odontogenic Tumor: Report of a Case with Nevoid Basal Cell Carcinoma Syndrome and Literature Review

Hasheminia

Naemi

Naghdi

2014

J. Maxillofac. Oral Surg.

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Keratocystic odontogenic tumor (KOT) is one of the major components of nevoid basal cell carcinoma syndrome (NBCCS), which usually occurs in young ages and includes significant structures of jaws. According to high recurrence rate of KOT, there are many controversies in treatment of the lesion, especially in syndromic cases with younger ages. In current report, authors suggest a conservative protocol in the treatment of KOT in a patient with NBCCS.

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Two-stage treatment protocol of keratocystic odontogenic tumour in young patients with Gorlin–Goltz syndrome: Marsupialization and later enucleation with peripheral ostectomy. A 5-year-follow-up experience

Cited by 21 publications

References 36 publications

Possible recurrence of keratocyst in nevoid basal cell carcinoma syndrome: A review of literature

Possible recurrence of keratocyst in nevoid basal cell carcinoma syndrome: A review of literature

Activation of Mesenchymal Stem Cells Promotes New Bone Formation Within Dentigerous Cyst

Conservative Treatment Protocol of Keratocystic Odontogenic Tumor: Report of a Case with Nevoid Basal Cell Carcinoma Syndrome and Literature Review

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