Multiple Orthokeratinized Odontogenic Cysts: A Report of Two Cases and Review of the Literature

Crane, Hannah; Forno, Philip Da; Kyriakidou, E.; Speight, Paul M.; Hunter, Keith D.

doi:10.1007/s12105-019-01042-0

Cited by 12 publications

(6 citation statements)

References 13 publications

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“…Histologically the key features include; a thin and regular layer of stratified squamous epithelium with orthokeratinization on the surface and prominent granular cell layer. 12,13 This was reported in the histology report for all cases of multiple OOC. OOC have an orthokeratinised epithelial lining with hypergranulosis and lamellated orthokeratin in the cystic cavity.…”

Section: Discussionmentioning

confidence: 76%

“…In the six previous cases of multiple OOCs, 50% of patients reported symptoms. [8][9][10][11][12] Additionally a review of 10 cases of single OOC identified that 90% of patients presented with swelling and pus-like discharge. 13 Although, it has previously been reported that patients are asymptomatic and incidental radiographic findings, 14 it is important to be aware that patients may report symptoms.…”

Section: Discussionmentioning

confidence: 99%

“…5,6,16 The majority of the patients with multiple OOC were treated with cyst enucleation and extraction of the impacted tooth. [8][9][10][11][12] In all cases there was a low recurrence rate which has been reported to be between 0 and 2%. 16 Formerly, Pindborg and Hansen (1963) described a keratocyst as any jaw cyst which formed keratin.…”

Section: Discussionmentioning

confidence: 99%

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A case report and literature review of multiple orthokeratinizing odontogenic cysts: The great mimicker

2021

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Background: Orthokeratinized odontogenic cysts are a rare type of odontogenic cyst, which are identified by an orthokeratinized stratified squamous epithelium. These cysts were originally classified as a subtype of odontogenic keratocysts; however, they have been redefined as a distinct entity. They radiographically mimic dentigerous cysts which can confound the final diagnosis. Most of the literature describes cases of single OOC, with far fewer cases of multiple orthokeratinized odontogenic cysts being reported.Method: This paper details a case of multiple orthokeratinized odontogenic cysts in a patient and compares their presentation to other cases of multiple orthokeratinising odontogenic cysts reported in the literature.Results: Six previous cases of multiple orthokeratinized odontogenic cysts were identified in the literature. The majority of features were similar to the case presented, with most cysts occurring in the mandible, in males in their third decade. In all but one of the cases, these cysts were also associated with unerupted teeth. Discussion: Orthokeratinizied odontogenic cysts display similar features to other odontogenic cysts. However, they possess distinctly different features to odontogenic keratocysts in their histopathology, presentation and recurrence rate. Conclusion:Orthokeratinized odontogenic cysts should be considered as a radiographical differential diagnosis for a dentigerous cyst and a histopathological differential diagnosis for odontogenic keratocyst.

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Section: Discussionmentioning

confidence: 76%

Section: Discussionmentioning

confidence: 99%

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A case report and literature review of multiple orthokeratinizing odontogenic cysts: The great mimicker

2021

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“…Taken together, our targeted NGS did not identify any pathogenic PTCH1 mutations common to multiple OOC cases. Two previous multiple OOC cases failed to detect pathogenic PTCH1 mutations by genetic testing using DNA from one blood sample or one OOC lesion [ 9 , 17 ]. Thus, abnormalities related to pathogenic PTCH1 mutations frequently detected in OKC were not observed in multiple OOC cases.…”

Section: Discussionmentioning

confidence: 99%

Multiple orthokeratinized odontogenic cysts: clinical, pathological, and genetic characteristics

et al. 2022

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Background: Orthokeratinized odontogenic cyst (OOC) is a rare developmental odontogenic cyst of the jaw. It was originally believed to be a variant of odontogenic keratocyst (OKC) but is now considered to be a distinct entity. OOC usually presents as a single lesion and recurs infrequently. On the other hand, OKC often presents with multiple lesions and displays locally aggressive behavior and a high recurrence rate associated with the protein patched homolog 1 (PTCH1) gene mutation. Multiple OOC cases are extremely rare and seem to be aggressive, but their pathogenesis is not fully understood. This study aimed to determine the clinical, pathological, and genetic characteristics of multiple OCC. Methods: Three cases of multiple OOC were evaluated for clinical and histological findings, and immunohistochemical expression of Ki-67 and Bcl-2. Furthermore, PTCH1 mutations were analyzed by next-generation sequencing using a custom panel to cover the entire exon of PTCH1. Results: The three cases of multiple OOC included two men and one woman with a mean age of 25.3 years old (range, 18–38 years old). Each case had two or three OOCs (total of seven OOCs), all of which were simultaneously detected. Of the seven OOCs that manifested as multiple jaw cysts, seven (100%) occurred in the posterior regions, four (57.1%) occurred in the mandible, and four (57.1%) were associated with an impacted tooth. Histological examination revealed cysts lined by orthokeratinized stratified squamous epithelium. Immunohistochemistry showed a low Ki-67 labeling index and no Bcl-2 expression in the seven OOCs. No pathogenic PTCH1 mutations were detected in any of the seven OOCs. None of the patients had any other symptoms or signs of recurrence at the last follow-up (6–60 months). Conclusion: Multiple OOCs appeared to occur more often in younger patients than solitary OOC. Both multiple and solitary OOCs may be related diseases within the entity of odontogenic cysts. Multiple OOCs are clinicopathologically and genetically distinct from OKC.

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“…4 It shows peculiar features under the microscope like a thick orthokeratinized keratin layer due to the presence of keratohyaline granules, lack of surface corrugations, prominent granular cell layer and flat to cuboidal basal cell layer. 5 2011. 3 Because of different treatment approach in OOC compared to OKC it has to be differentiated from the later.…”

Section: Introductionmentioning

confidence: 99%

A series of six uncanny orthokeratinized odontogenic cysts: Revisiting the literature

Khatoon

Srinath²,

Jayaram³

et al. 2022

JOOO

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Orthokeratinized odontogenic cyst is a rare odontogenic cyst which was considered to be a variant of odontogenic keratocyst, but has been recognized as a separate entity due to its varied behavior and histology. Commonly associated with an unerupted tooth in the posterior mandible, it mimics various lesions like dentigerous cyst, odontogenic keratocyst, unicystic ameloblastoma etc. Orthokeratinized odontogenic cyst was thought to be less destructive compared to odontogenic keratocyst. This case series highlights the clinically aggressive nature of Orthokeratinized odontogenic cyst.In this case series six cases of diagnosed Orthokeratinized Odontogenic cyst have been described along with one case of multiple Orthokeratinized odontogenic cysts in a female patient. The clinical, radiographic and histopathologic features have been described in great detail in order to correlate with the previous literature and highlight their clinically aggressive behavior.Orthokeratinized odontogenic cyst can be deceptive and should be diagnosed appropriately to avoid aggressive treatment protocol. Most of these cysts are associated with inflammation which makes it harder to be diagnosed as the epithelium and connective tissue would have undergone structural changes. Differentiating the cystic lesions of the oral cavity is utmost important to devise appropriate treatment suitable for the patient and for assessing the prognosis, recurrence and rehabilitation to improve the quality of life.

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Multiple Orthokeratinized Odontogenic Cysts: A Report of Two Cases and Review of the Literature

Cited by 12 publications

References 13 publications

A case report and literature review of multiple orthokeratinizing odontogenic cysts: The great mimicker

A case report and literature review of multiple orthokeratinizing odontogenic cysts: The great mimicker

Multiple orthokeratinized odontogenic cysts: clinical, pathological, and genetic characteristics

A series of six uncanny orthokeratinized odontogenic cysts: Revisiting the literature

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