牙源性角化囊性瘤恶变1例

牙源性角化囊性瘤（KCOT）是一种常见的颌骨牙源性囊性病损,临床上牙源性角化囊性瘤复发常见,而恶变较少见。本文报道1例由牙源性角化囊性瘤恶变为原发性骨内鳞状细胞癌的病例。     

Adenomatoid Odontogenic Tumor (AOT) Arising from a Dentigerous Cyst: Literature Review and Report of a Case

Adenomatoid odontogenic tumor (AOT) is an uncommon benign tumor of odontogenic origin composed of odontogenic epithelium in a variety of histopathological patterns. These lesions are usually solid but are occasionally cystic. AOT has been reported to occur in association with odontogenic cysts. Very few cases have been described that arise in association with a dentigerous cyst. A systematic search of the English-language medical literature in PubMed and Medline search (keywords adenomatoid odontogenic tumor, dentigerous cyst) data bases revealed only ten such cases.The present case is very unique, exceptional and first of this kind that occurred in the posterior mandible associated with an impacted lower first premolar in a 19-year old female patient. The intraosseous follicular AOT, as this case reported here, frequently resemble dentigerous cysts. There is an uncertainty whether the lining of an associated cyst represents a true dentigerous cyst, cystic change within an AOT or may represent a distinct entity. We believe that this case represents an odontogenic cyst with neoplastic change. Hence, in such cases, the final diagnosis should be made on the basis of histological examination of extensive tissue sampling of entire excised tissue specimen. Further studies to determine whether the AOT derived from an odontogenic cyst could represent a distinct variant are to be done.     

Multiple osseous dysplasia arising from impacted teeth: report of a case associated with odontogenic lesions

We report a case of osseous dysplasia (OD) showing extremely rare clinical features. A 37-year-old Japanese woman was referred to our clinic complaining of a left alveolar bony swelling at an impacted canine. Radiographic examinations revealed a mixed radiopaque lesion involving the impacted left canine and also revealed an impacted left wisdom tooth with a cystic change and a clear radiopaque lesion suspected to be odontoma. All the extracted upper teeth and odontoma showed hypercementosis and the canine was fully involved in it. Histopathologically, they showed the same features and were diagnosed as OD. The findings of multiple OD from incompletely erupted teeth and odontoma in our case may well show that OD can arise from any tooth with periodontal ligaments and that focal OD can show expansive growth like the ossifying fibroma.     

External tooth resorption associated with a peripheral odontogenic fibroma: review and case report

The purpose of the study is to document a rare case of a peripheral odontogenic fibroma with associated cervical and coronal tooth resorption in a 38 year old woman. Histopathological features are described, the clinical management outlined and follow‐up observations over 27 years detailed. The exophytic firm lesion, coral pink in appearance, located on the labial aspect of a maxillary right lateral incisor was excised, fixed in formalin and prepared for histological evaluation. The resorption cavity and adjacent soft tissue were treated by the topical application of trichloroacetic acid prior to restoration with a glass‐ionomer cement and subsequent root canal treatment. Histologically, the body of the lesion was characterized by the presence of odontogenic epithelium embedded in a mature fibrous stroma. Areas of dystrophic calcification could also be identified. The features were consistent with a diagnosis of a peripheral odontogenic fibroma. The clinical result of treatment assessed 27 years postoperatively showed no evidence of recurrence of the peripheral odontogenic fibroma. External cervical and coronal tooth resorption can, on rare occasions, prove to be a clinical feature associated with peripheral odontogenic fibroma. Treatment of the tumour mass and the resorptive lesion can provide a successful outcome.     

Podoplanin expression in human tooth germ tissues and cystic odontogenic lesions: an immunohistochemical study

J Oral Pathol Med (2010) 39 : 115–120
Background:  Podoplanin expression was described in mouse tooth germ and apical bud cells. The aim of this study was to analyse the podoplanin expression of human tooth germ tissues, adult teeth and odontogenic lesions immunohistochemically.
Study Design:  Nine human tooth germ biopsies and seven healthy permanent teeth extracted for orthodontic reasons were examined. Anti-podoplanin (D2-40) reactivity was investigated immunohistochemically. Five well-defined cystic odontogenic lesions (10 radicular cysts, 10 follicular cysts, three keratocystic odontogenic tumours, five ameloblastomas, and two adenomatoid odontogenic tumours) were analysed simultaneously.
Results:  Podoplanin expression was detected in the majority of epithelial and ecto-mesenchymal cells of human tooth germ tissues, odontoblasts and superficial dental pulp fibroblasts of permanent teeth. Cystic odontogenic lesions revealed positive reactions predominantly at the invasion front edge within basal epithelial layers.
Conclusion:  Podoplanin appears to be involved in the orthologic and pathologic processes of the formation of elongated cell extensions and odontoblastic fibers, in the epithelial–mesenchymal transition and local invasion during tooth germ development as well as in both reactive and neoplastic odontogenic cystic lesions.     

36例与阻生牙相关的牙源性角化囊肿的临床影像学特点分析

目的:分析与阻生牙有关的牙源性角化囊肿的临床和影像学特点。方法:对235例牙源性角化囊肿中36例与阻生牙有关的临床、影像学资料进行回顾分析。结果:在与阻生牙有关的牙源性角化囊肿病例中,男女比例为1.77∶1,下颌磨牙-下颌支部26例,占72.22%;19例（52.8%）在20~30岁就诊。单房型27例（75%）,多房型9例（25%）;牙冠位于囊内13例（36.11%）,整个牙位于病变内15例（41.67%),而阻生牙位于病变一侧8例（22.22%）。结论:与阻生牙有关的牙源性角化囊肿的一个主要影像学特点是,整个牙被病变包绕或位于囊肿一侧,而表现为冠周密度减低影像者较少。     

下颌阻生智齿拔除后第二磨牙远中牙槽骨变化的临床研究

目的追踪调查下颌阻生智齿拔除后,邻近的第二磨牙远中牙槽骨的远期预后。方法挑选因下颌智齿水平或近中阻生而在2年前要求拔牙的患者,在接受拔牙时,邻近第二磨牙远中牙槽骨已经发生水平吸收的病例,共计104例。复查内容包括：下颌第二磨牙牙周深度探诊,摄术后数字牙片,测量近远中牙槽骨垂直高度,然后与拔牙前的相应数据做比较。采取多项措施减少误差以保证数据可比性。结果72.1%的下颌第二磨牙远中牙周探诊深度≥6mm;术后远中牙槽骨可以再生,高度大于术前（P〈0.01）,但仍小于近中牙槽骨高度（P〈0.01）;年龄对牙槽骨增量无影响。结论下颌阻生智齿拔除后,邻近的第二磨牙远中牙槽骨可以再生,但无法恢复至正常水平;应提倡一期植入人工骨改善其远期预后。     

Management of the malposition tooth that was caused by dentigerous cyst associated with impacted supernumerary tooth by using an obturator

This report presents a case of eruption guidance for an upper right central incisor with a dentigerous cyst associated with an impacted supernumerary tooth in a 5-year-9-month-old boy. A swelling was seen in the region of an upper right central incisor. Radiographic examinations showed that a well-defined cystic lesion, containing an impacted supernumerary tooth crown in it, was located between the root of the primary upper right central incisor and a permanent upper central incisor germ. Under general anesthesia, the dentigerous cyst was removed at extraction of the upper impacted supernumerary tooth. And then, a socket was kept open wound. A histopathological examination confirmed the diagnosis of a dentigerous cyst associated with a supernumerary tooth. By using an obturator, we guided eruption of an upper right central incisor with malposition that was caused by the dentigerous cyst, and observed the way of the tooth eruption for two years and seven months. The obturator was inserted in the socket directly for three months until it was closed. After that, the obturator was used as only a removable space maintainer. Eight months after the operation, the obturator was removed because the upper left central incisor began to erupt. Two years and seven months after the operation, both upper central and lateral incisors had erupted completely. In this case, the obturator was used for expectation of natural eruption of permanent incisors, space maintenance and aesthetic recovery. The permanent incisors had been able to be guided to almost correct position without traction.     

Calcifying epithelial odontogenic tumour presenting at a surgical site: case report

We describe the management of a calcifying epithelial odontogenic tumour with an atypical clinical presentation at the site of a previously surgically exposed impacted maxillary canine in a 51-year-old woman.     

Adenomatoid odontogenic tumor: a report of a rare case with recurrence

Adenomatoid odontogenic tumor (AOT) is an uncommon tumor of odontogenic origin, composed of odontogenic epithelium in a variety of histoarchitectural patterns, and characterized by slow but progressive growth. Generally, recurrences seldom, if ever, occur after surgical curettage, while here we report an extremely rare case of AOT with recurrence. The patient was a 36-year-old Chinese man who presented with a palpable bony-hard swelling in the anterior maxillary region initially at the age of 16 and the lesion recurred twice over a 20-year period. The tumor was diagnosed as AOT with well-defined unilocular radiolucency shown in X-ray graphs and solid nodule of cuboidal or columnar cells of odontogenic epithelium forming typical nests or rosette-like structures as well as characteristic duct-like spaces in histologic findings.     

Calcifying epithelial odontogenic tumor of the maxilla with ulcerative stomatitis: a case report

Calcifying epithelial odontogenic tumor (CEOT) is a rare benign odontogenic tumor, known as Pindborg tumor. Although ulcer formation was reported in one previously involving the peripheral maxilla, such change of the overlying mucosa has been reported in intraosseous CEOT. We report maxillary CEOT in a patient who complained of spontaneous pain due to extensive ulcer formation of the oral mucosa.     

Ameloblastoma, calcifying epithelial odontogenic tumor, and glandular odontogenic cyst show a distinctive immunophenotype with some myoepithelial antigen expression

Background:  Odontogenic neoplasms have some morphologic overlap with salivary gland neoplasms, many of which show myoepithelial differentiation. In the 1980s, an ultrastructural study identified a population of myoepithelial-like cells in calcifying epithelial odontogenic tumor. Myoepithelial derived tumors have since been shown to have distinct immunohistochemical profiles.
Methods:  We examined a series of odontogenic neoplasms, including 11 ameloblastomas, four calcifying epithelial odontogenic tumors, five glandular odontogenic cysts (GOCs), and five keratocystic odontogenic tumors with a panel of myoepithelial-associated immunohistochemical stains. We also assessed representative control examples of oral mucosa, odontogenic rests, and dentigerous cysts.
Results:  All of the neoplastic and non-neoplastic oral epithelium-derived entities share a p63-positive, high molecular weight cytokeratin (CK5/6)-positive immunophenotype. Calponin reactivity was at least focally present in two of four calcifying epithelial odontogenic tumors, three of five GOCs, and 10 of 11 ameloblastomas; the sole completely non-reactive ameloblastoma represents a lung metastasis. One case of calcifying epithelial odontogenic tumor was focally positive for glial fibrillary acidic protein. However, other more definitive markers of myoepithelial differentiation, including S-100 and smooth muscle actin, were negative. Two of three calcifying epithelial odontogenic tumors and five of five GOCs were also positive for a low molecular weight cytokeratin (CK7).
Conclusions:  Ameloblastomas, GOCs, and calcifying epithelial odontogenic tumors show a distinctive immunophenotype which overlaps with that of myoepithelial-derived salivary gland neoplasms but does not provide definitive support for myoepithelial differentiation.     

牙源性角化囊性瘤恶变1例

牙源性角化囊性瘤（KCOT）有潜在的侵袭性和较高的术后复发率,但恶变却很罕见。本文报道1例KCOT术后多次复发及感染,最终恶变为鳞状细胞癌的病例,并结合文献对其进行临床病理分析。     

Mucinous adenocarcinoma with neuroendocrine differentiation of the mandibular ramus: report of a case

A rare case of mucinous adenocarcinoma with neuroendocrine differentiation of the mandibular ramus is presented. The patient, an 80-year-old man, was referred to our hospital with chief complaint of swelling and pain in the left buccal mucosa. CT and MRI examination showed an osteolytic tumor mass occupying the upper region of the left mandibular ramus. Macroscopically, the excised tumor was a relatively well-defined, solid mass with diffuse bone resorption, measuring 3 cm x 3.2 cm x 3 cm. Microscopical examination showed that the tumor forming glandular structures with abundant mucous production and high cellular atypia. Immunohistochemical studies demonstrated the positive reactivities for pan-keratin, cytokeratin 7, vimentin,alpha-amylase, alpha-smooth muscle actin, neuron-specific enolase, glial fibrillary acid protein, calcitonin, and somatostatin in tumor cells. These findings suggested that the tumor was originated from heterotopic or misplaced salivary gland in the mandible.     

Synchronous gubernacular canals with compound odontoma associated with a calcifying odontogenic cyst and transmigrated canine: An extremely rare event

This article reports the novel incidental concurrences of multiple oral lesions in a 13-year-old boy. Radiographically, an over-retained primary canine was seen with a subjacent mixed radiopacity and transmigrated permanent canine with a suspected dentigerous cyst (each with gubernacular canals). Treatment consisted of enucleation and removal of the impacted canine. The histopathologic findings were compound odontoma containing multiple tooth-like structures and calcifying odontogenic cyst with epithelium containing ghost cells, calcifications and subjacent fibrous connective tissue wall. All cases of odontomas should undergo microscopic assessment for diagnosis and ascertainment of associated pathosis.     

Clear cell odontogenic tumor in the mandible: report of a case with duct-like appearances and dentinoid induction

A case of clear-cell odontogenic tumor with unusual histological features is presented. A 61-year-old Japanese man was admitted because of swelling of the left premolar-molar region of the mandible. Radiological examination revealed a multilocular radiolucency with irregular margins. Histological examination of the resected specimen showed infiltrative proliferation of both clear and eosinophilic cells into the adjacent soft tissue without encapsulation, suggesting the malignant potential of the tumor. The tumor cells sporadically formed cystic lesions. In addition, several tumor cell nests showed duct-like characteristics, and many eosinophilic dentin-like structures were attached to the tumor cell nests, suggesting the potential for epithelial-mesenchymal induction. Histochemically, the clear tumor cells possessed cytoplasmic glycogen granules. Both clear and eosinophilic tumor cells showed positive immunoreactivities for cytokeratin 19, epithelial membrane antigen and filaggrin, indicating an odontogenic epithelial origin.     

Treatment of a transmigrated and an impacted mandibular canine along with missing maxillary central incisor: A case report

IntroductionTreatment of a case with impacted canines is complex and numerous options are available ranging from disimpaction to forced eruption.ObjectiveThe objective of this case report was to describe the treatment of a 19-year-old male, with a skeletal Class II, dental Class II Division 1 malocclusion, prognathic maxilla, proclined maxillary incisors, with missing left maxillary central incisor and bilateral mandibular canine impaction.MethodologyThe orthodontic treatment plan included forced eruption of the impacted canines and conversion of maxillary left lateral incisor to central incisor.ConclusionProper diagnosis and implementation of orthodontic biomechanics can minimize the amount of prosthetic and surgical intervention needed in the management of transmigrated canine and smile designing.     

Adenomatoid odontogenic tumor arising from the mandibular molar region: a case report and review of the literature

This case report describes a rare case of an adenomatoid odontogenic tumor (AOT) arising in the mandibular third molar region of a 20-year-old female. The tumor was a well-circumscribed intraosseous lesion with an embedded tooth. Histologically, the induction of extensive globular calcification was evident in addition to the characteristic histological features of AOT. The present case lends support to the categorization of AOT as an odontogenic tumor consisting of a disorderly mixture of odontogenic epithelium and odontogenic ectomesenchyme with calcification.     

Invasion of the intracranial space by a malignant odontogenic mixed tumor: Report of a case

A case of an odontogenic tumor which invaded the intracranial space from the mandible is reported. Judging from the radiographic images it was similar to a malignant tumor. The patient died 17 years after the first visit. According to the final pathological diagnosis, it was malignant odontogenic mixed tumor of low grade which did not belong to any of the WHO classification.