Cases reported "Odontogenic Cysts"

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1/63. Odontogenic keratocysts in a 5-year-old: initial manifestations of nevoid basal cell carcinoma syndrome.

    The purpose of this paper is to report the occurrence of odontogenic keratocysts in a young child. Odontogenic keratocysts are one of the principal features of nevoid basal cell carcinoma syndrome. Their occurrence in this syndrome is usually during the second or third decades of life. This report describes the occurrence of odontogenic keratocysts in a 5-year-old, which proved to be the initial presentation of nevoid basal cell carcinoma syndrome and highlights the need to consider this syndrome as a possible diagnosis in all cases of odontogenic keratocysts.
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keywords = carcinoma
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2/63. Glandular odontogenic cyst: a case report.

    Glandular odontogenic cyst (GOC) is an extremely rare lesion occurring in the jawbones. Sialo-odontogenic cyst was first described as a multicystic lesion resembling a botryoid odontogenic cyst (BOC) or a central mucoepidermoid carcinoma by Padayachee in 1987. In 1988, Gardner used the term "glandular odontogenic cyst" and considered it as a histologic variant of BOC. Most authors agreed that GOC was odontogenic because of the concurrent ball-like epithelial structure, ameloblastoma, squamous odontogenic tumor-like proliferation in its wall, or hyaline bodies in the epithelium lining. Recently, immunohistochemical studies of the cytokeratin profiles have also supported this concept. Its aggressive behavior and the recurrent tendency make it important. A new case of GOC in a 59-year-old male presented as a multilocular radiolucency in the anterior region of the mandible, invading the marrow space by epithelial islands is described with other clinicopathologic features and the literature is briefly discussed.
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keywords = carcinoma
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3/63. PTCH gene mutations in odontogenic keratocysts.

    An odontogenic keratocyst (OKC) is a benign cystic lesion of the jaws that occurs sporadically or in association with nevoid basal cell carcinoma syndrome (NBCCS). Recently, the gene for NBCCS was cloned and shown to be the human homologue of the drosophila segment polarity gene Patched (PTCH), a tumor suppressor gene. The PTCH gene encodes a transmembrane protein that acts in opposition to the Hedgehog signaling protein, controlling cell fates, patterning, and growth in numerous tissues, including tooth. We investigated three cases of sporadic odontogenic keratocysts and three other cases associated with NBCCS, looking for mutations of the PTCH gene. Non-radioactive single-strand conformational polymorphism and direct sequencing of PCR products revealed a deletion of 5 base pairs (bp) in exon 3 (518delAAGCG) in one sporadic cyst as well as mutations in two cysts associated with NBCCS, a nonsense (C2760A) and a missense (G3499A) alteration. This report is the first to describe a somatic mutation of PTCH in sporadic odontogenic keratocysts as well as two novel mutations in cysts associated with NBCCS, indicating a similar pathogenesis in a subset of sporadic keratocysts.
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ranking = 0.16666666666667
keywords = carcinoma
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4/63. Eosinophilic pustular folliculitis in association with nevoid basal cell carcinoma syndrome.

    This study reports on the clinical and light microscopic features of a nevoid basal cell carcinoma syndrome with the complication of eosinophilic pustular folliculitis. To the authors' knowledge, this is the first report of such an association, which is possibly due to immune dysregulation. Moreover, the patient experienced remission of eosinophilic pustular folliculitis after removal of the jaw cyst. One possible explanation for the remission is that a long-lasting TH, type inflammatory response as a result of the bone defect produces effective cytokines such as interferon-gamma.
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ranking = 0.83333333333333
keywords = carcinoma
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5/63. Primary intraosseous verrucous carcinoma developing from a maxillary odontogenic cyst: case report.

    Primary intraosseous carcinoma (PIOC) is an extremely rare lesion, almost always occurring in cranial bones. The origin of this tumor, specific to the maxillae, is associated with the cells of the epithelial rests of Malassez. Among the histotypes which can be included in these neoplasms, verrucous carcinoma is of particular interest due to its rarity: only a single case has been reported to date. After a short survey of the literature, the authors describe a directly observed case of verrucous carcinoma arising from a maxillary odontogenic cyst.
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ranking = 1.1666666666667
keywords = carcinoma
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6/63. Primary intraosseous carcinoma arising from an odontogenic cyst: a case report and review of the Japanese cases.

    A rare case of primary intraosseous carcinoma (PIOC) arising from an odontogenic cyst in a 58-year-old man is reported. Clinical and radiological examinations revealed an odontogenic cyst of the maxilla. Histopathologically, the lesion was composed of a cyst with a parakeratotic epithelial lining and well-differentiated squamous cell carcinoma, showing continuity between them without a connection to the oral mucosa. Twenty-eight well-documented Japanese cases of Type-1 PIOC, including the present case, were reviewed. The mean age of the 28 patients was 56.1 years, and the male to female ratio was 1.8:1.0. Compared with currently reported Japanese reviews of Type-3, foreign Type-1 and Type-3, there were no significant differences in mean patient age and sexual predominance, and no racial difference. The pathogenesis of Type-1 PIOC is also discussed.
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keywords = carcinoma
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7/63. Malignant transformation in odontogenic keratocysts. Case report.

    Squamous cell carcinoma arising in the epithelial lining of an odontogenic keratocyst is a rare finding. Up to now, only 12 cases have been reported in the literature. The present work reports a new case diagnosed in a 70 year old man. The clinical, radiographic, and histopathological findings and the treatment are described.
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ranking = 0.16666666666667
keywords = carcinoma
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8/63. A large glandular odontogenic cyst of the mandible: report of case.

    Glandular odontogenic cyst (GOC) is generally considered uncommon, but several investigators claim there is a more frequent occurrence than previously thought. However these case reports lacked confirming data to validate their claim. On the other hand, it is possible that cases of central mucepidermoid carcinoma or later periodontal cyst might be viewed as glandular odontogenic cyst. This is a report of a case of a 70-year old male who presented with a firm swelling in the right side of his edentulous mandible. Radiographic examination revealed a multilocular radiolucent lesion in the mandible extending from the right first premolar to the left second premolar and reaching the inferior mandible. Clinical findings, the health history, and microscopic examination of excised tissue confirmed the diagnosis of GOC. The lesion was excised and post-operative healing was uneventful.
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ranking = 0.16666666666667
keywords = carcinoma
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9/63. Multiple recurrent and de novo odontogenic keratocysts associated with oral-facial-digital syndrome.

    In 1954, Papillon-Leage and Psaume were the first to describe the clinical characteristics of oral-facial-digital syndrome (OFDS). On the basis of their clinical features and the inheritance pattern, 2 variants were initially distinguished, namely OFDS type I (Papillon-Leage and Psaume) and OFDS type II, or Mohr syndrome. At present, 11 types of OFDS have been discovered. OFDS represents a heterogeneous group of disorders characterized by oral manifestations including oral frenula, cleft or lobulated tongue, high arched palate, cleft lip and/or palate, facial anomalies, and digital abnormalities such as syndactyly, polydactyly, brachydactyly, and clinodactyly. Depending on the type of OFDS, abnormalities may be present in other organs, such as the brain and heart. We report a patient with OFDS in whom multiple recurrent and de novo keratocysts were found. Although multiple keratocysts are commonly found in Gorlin-Goltz nevoid basal cell carcinoma syndrome, a relationship between OFDS and multiple keratocysts has not been described.
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ranking = 0.16666666666667
keywords = carcinoma
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10/63. Implant prosthodontic rehabilitation of a patient with nevoid basal cell carcinoma syndrome: a clinical report.

    This clinical report describes the prosthodontic rehabilitation of an edentulous patient with compromised maxillary and mandibular anatomy, caused by the resection of several odontogenic keratocysts associated with nevoid basal cell carcinoma syndrome. Preprosthetic surgical preparation included iliac crest bone grafting to both arches and placement of implants. Prosthodontic treatment included the fabrication of maxillary and mandibular dental implant-supported connecting bars and construction of attachment-retained overdentures.
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ranking = 0.83333333333333
keywords = carcinoma
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