Clear cell Cystic Variant of Calcifying Epithelial Odontogenic Tumor

Calcifying epithelial odontogenic tumor (CEOT) is a solid, locally aggressive, benign odontogenic neoplasm characterized by sheets and nests of polyhedral epithelial cells exhibiting eosinophilic and less often clear cytoplasm, occasional nuclear pleomorphism without mitotic activity, calcifications, and deposits of amyloid. A cystic variant has been reported only twice. Herein, we present an additional example of cystic CEOT occurring in a 31-year-old male and featuring clear cell epithelial lining with deposits of amyloid and osteodentin.     

Multifocal calcifying epithelial odontogenic tumor.

The calcifying epithelial odontogenic tumor (CEOT), or Pindborg tumor, is a rare and benign odontogenic neoplasm that affects the jaw. The most common manifestation of CEOT is a unifocal or localized lesion of the involved jaw, which may appear clinically as a hard tissue swelling and radiographically as a mixed radiolucent-radiopaque mass. In this article, we present a unique case of CEOT affecting multiple sites in the maxilla and mandible of a 51-year-old white man. Though biopsy samples from all involved sites revealed similar histopathologic features consistent with CEOT, the fact that there was a multifocal presentation is an unusual phenomenon for CEOT and has never been reported. Multifocal odontogenic lesions are not typical but have been observed in conditions associated with known genetic mutations. For example, multiple odontogenic keratocysts are the most common feature of the inherited condition known as nevoid basal cell carcinoma syndrome. This case, however, is the first one to demonstrate that there may be a multifocal variant of CEOT that has not been previously recognized.     

Calcifying Epithelial Odontogenic Tumour with Clear Langerhans Cells: A Novel Variant,Report of a Case and Review of the Literature

Clear cell calcifying epithelial odontogenic tumour (CCEOT) is a rare variant of calcifying epithelial odontogenic tumor (CEOT). While it is not surprising to find clear cells in odontogenic lesions, the exact nature of the clear cells in CCEOT has not been elucidated. Herein, we report a case of peripheral CCEOT of anterior mandible in a 37 year old black female. Histologically, the tumour consisted of cords and small nests of clear cells surrounded by dense deposits of amyloid and basophilic calcifications. The cells possessed abundant clear cytoplasm and eccentrically located indented nuclei. Admixed with the clear cells were eosinophilic cuboidal to polyhedral cells. The clear cells were PAS negative and immunoreactive for S100 protein, CD1a and Langerin. The clear cells were negative for MNF-116, SMA, Desmin and CK-19. It is therefore recommended to recognize two variants of CCEOT, namely, CEOT with clear cell change and CEOT with clear Langerhans cells (LC). We further suggest that the contradictory term “non-calcifying variant of calcifying epithelial odontogenic tumour with LC” to be abandoned, as the current case clearly indicates that LC could be seen in CEOT irrespective of the presence or absence of calcifications.     

Clear cell variant of calcifying epithelial odontogenic tumor: Case report and review of the literature

The calcifying epithelial odontogenic tumor (CEOT) is a rare benign odontogenic neoplasm which was first described by Pindborg in 1955 and accounts for less than 1% of all odontogenic lesions. Recently, a clear cell variant of CEOT has been identified with only eight well-documented cases in the literature. We present an additional case of clear cell CEOT of the mandible and review the salient clinical, radiologic, and histopathologic features of this entity and CEOTs in general. The differential diagnosis of clear cell tumors in the mandible includes: clear cell odontogenic tumor, clear cell ameloblastoma (odontogenic carcinoma), metastatic clear cell adenocarcinoma, primary intraosseous mucoepidermoid carcinoma, acinic cell carcinoma, epithelial-myoepithelial carcinoma, clear cell salivary gland tumors, and clear cell variant of squamous cell carcinoma. Because of the belief that clear cell odontogenic tumors are locally aggressive neoplasms, definitive resection of the entire mass with tumor-free surgical margins and long-term follow-up are recommended. © 1994 John Wiley & Sons, Inc.     

Peripheral Clear Cell Variant of Calcifying Epithelial Odontogenic Tumor: Case Report and Review of the Literature

Calcifying epithelial odontogenic tumor (CEOT) is classified as an uncommon, benign, odontogenic neoplasm that is exclusively epithelial in origin. Of all reported cases, peripheral tumors account for 5–6 %, among which clear cell variants are still rare. A rare case of peripheral clear cell variant of CEOT occurring in the mandibular gingiva of a 47-year-old female is presented.     

Multi-recurrent invasive ameloblastoma: A surgical challenge

IntroductionAmeloblastomas are rare head and neck tumors, and yet the most common odontogenic neoplasms. They account for 1% and 11% of all head and neck and odontogenic tumors respectively. Embryologically, they originate from remnants of odontogenic epithelium. Their aggressive, destructive nature, as well as their anticipated high rate of recurrence, even after en bloc resection, poses a surgical predicament.PresentationWe present a case of a 56 year-old Asian female with a multi-recurrent invasive ameloblastoma. Initially, the lesion was mandibular in location for which she underwent a mandiblectomy. Later on, she presented with a maxillary ameloblastoma with invasion of both the anterior wall of the maxillary sinus and the floor of the orbit. The patient was operated twice and histopathology confirmed a cystic type recurrent ameloblastoma. A year later, she came with recurrent maxillary ameloblastoma and a maxillectomy was done. However, histopathology revealed a follicular ameloblastoma. Three years later, she presented with a retro-orbital ameloblastoma with infiltration to the temporal muscles. The patient was operated and the histopathologic examination revealed a partially cystic lesion with no malignant transformation.ConclusionThis case discusses available treatment options and emphasizes on the importance of long-term patient follow-up due to the biological behavior of ameloblastoma.     

Glandular odontogenic cyst: A case report and clinicopathologic analysis of the relationship to central mucoepidermoid carcinoma

Glandular odontogenic cyst (GOC), an unusual multilocular cystic lesion of jaws exhibiting the features of both botryoid odontogenic cyst and mucoepidermoid tumor has been reported recently as a new entity with uncertain histogenesis. The World Health Organization named GOC as an independent pathologic entity and classified it as a developmental odontogenic epithelial cyst. We report an additional case, describe its clinicopathologic features, and discuss the similarity to the central mucoepidermoid carcinoma. © 1997 John Wiley & Sons, Inc. Head Neck 19: 227–231, 1997.     

Peripheral Calcifying Odontogenic Cyst: A Case Report and Review of the Literature

The peripheral calcifying odontogenic cyst (PCOC) accounts for less than 25% of the cases of calcifying odontogenic cysts and most commonly appears as a nodule on the gingiva. This paper aims to present both a case report of a PCOC located in the left vestibular maxilla as well as a review of the English-language literature. An 11-year-old female patient presented a swelling in the vestibular region of teeth 12 and 13. Periapical and panoramic radiographs demonstrated irregular calcification. Surgical excision was performed. Microscopic examination showed an odontogenic cystic lesion lined by ameloblastoma-like epithelium, containing numerous ghost cells. Areas of calcification associated with ghost cells could also be observed. The patient was diagnosed with PCOC. The patient has been disease-free for 36 months. The review of the cases of PCOC showed 44 well-defined cases. The mean age was of 49.4 years at the time of diagnosis. The reported cases appeared as a painless swelling, with a slight predilection for females, and were more frequently located in the anterior region of the maxilla or mandible. Surgical excision is the treatment of choice, and recurrence is rare.     

A rare case report of an adenomatoid odontogenic tumor associated with odontoma in the maxilla

IntroductionThe adenomatoid odontogenic tumor (AOT) associated with odontoma has been described as a new entity with overlapping features of adenomatoid-like structures and odontoma. Presentation of the case: We report the second case of AOT associated with odontoma in the posterior maxillary region. A 22-year-old patient presented expansion of the vestibular and palatine cortical wall of maxilla. Radiographic examination and computed tomography confirmed erosion of maxillary walls, root resorption, displacement of a neighboring tooth and radiopacities within the lesion. Histopathological analysis showed a cystic cavity lined by odontogenic epithelium, organized as duct like structures and tubular dentin. The diagnosis established was AOT associated with odontoma.DiscussionAs fewer cases have been described, the aim of this study is to report clinical behavior and evolution of adenomatoid odontogenic tumor associated with odontoma, as it have not yet been described and its origin is not completely established. The hamartomatous or neoplastic origin of this tumor is under debate.ConclusionsTherefore, it is necessary similar cases to be published to increase the knowledge about the clinical behavior and evolution of this tumor, to enable such lesions to be more clearly defined in the next classification of odontogenic tumors.     

Metastatic malignant ameloblastoma of the kidneys

Ameloblastoma is an uncommon disease in the urological field. The resulting tumors or cysts are of odontogenic epithelial origin, are usually benign in nature and rarely metastasize to distant organs. We describe a case of metastatic ameloblastic carcinoma in both kidneys of a 38-year-old Japanese man, who had a history of malignant ameloblastoma and was referred to us for evaluation because of gross hematuria and left flank pain. Computed tomography showed irregular cystic masses in both kidneys. After we confirmed that the primary lesion and the lung metastatic lesion had not recurred, we treated the patient surgically. Approximately 4 months postoperatively the patient suffered a local recurrence of tumors that was very invasive and aggressive. The patient died 2 months later and the autopsy showed local metastasis only, without any metastatic lesion in the lungs or other organs. The present case showed that malignant ameloblastoma is highly aggressive, and in the case of metastases the prognosis is usually extremely poor.     

Differenzialdiagnosen einer schmerzlosen wurzelresorbierenden Raumforderung des Oberkiefers

A 41-year-old male patient was referred because of a root-resorbing lesion in the left upper jaw. The intra-oral examination showed a fistula palatinal and close to tooth 23. The CT investigation confirmed an osteolytic lesion with thinning and partial break through of the vestibular corticalis of the left upper jaw and with resorption of several tooth roots. A biopsy was performed but unspecific. Therefore the lesion was removed completely followed by a detailed histological examination which revealed the presence of an odontogenic fibroma. Odontogenic fibromas are rare odontogenic tumors. Enucleation is the treatment of choice. Relapses have been reported rarely.     

A case of adenomatoid odontogenic tumor with intracranial extension

A 49-year-old female of adenomatoid odontogenic tumor with intracranial extension was reported. She had 3 months history of left nasal obstruction and visual disturbance. Endocrinological examinations revealed a low basal level of serum cortisol and low level of urinary 17-OHCS. Neurological examinations revealed left anosmia and left visual disturbance (hand movement, marked afferent pupillary defect, and lower hemianopsia). Plain X-ray films and frontal tomograms of the skull showed a mass of soft tissue density in the left upper maxillary, left posterior ethmoid, and sphenoid sinuses with extensive destruction of the sella turcica, dorsum sellae, and anterior clinoid process. Plain axial CT scans demonstrated a low density mass filling up the left maxillary, left posterior ethmoid, and sphenoid sinuses and extending into the left middle fossa with bony destructions. Contrast-enhanced coronal CT scans demonstrated an extradural extension of the tumor into the left middle fossa and upward displacement of the pituitary complex. Left carotid angiograms showed a faint narrowing of the left internal carotid artery between the cisternal portion and the origin of the ophthalmic artery. Subtotal removal of the tumor and decompression of the optic canal and superior orbital fissure were performed via a left subtemporal extradural approach. Adenomatoid odontogenic tumor is a comparatively rare benign lesion which exhibits a slowly expansive growth without recurrence or metastasis after the surgical removal. No other case that recurred several times and extended into the intracranial space has been reported in the literature.(ABSTRACT TRUNCATED AT 250 WORDS)     

Eosinophilic granuloma of the hand

The first reported case of eosinophilic granuloma of the hand is presented. A slowly expanding lesion of the right third metacarpal shaft was treated by biopsy and curettage. Histological examination confirmed the presence of eosinophilic granuloma. A bone scan showed that it was a solitary lesion. There has been no evidence of recurrence of the lesion during follow-up. Eosinophilic granuloma is a rare disease of the mononuclear phagocyte system, representing one of the clinical manifestations of Langerhans-cell histiocytosis. The lesions are usually associated with the skull, femur, mandible and ribs; this condition has not been previously reported in the hand.     

Squamous Odontogenic Tumor

A 24 year male presented with a radiolucent lesion associated with the right maxillary canine. Histologic examination of the biopsy material resulted in a diagnosis of squamous odontogenic tumor a rare, benign epithelial odontogenic neoplasm. The clinical, radiographic and morphologic features of the squamous odontogenic tumor are discussed.     

Orthokeratinized Odontogenic Cyst of the Mandible with Heterotopic Cartilage

Cartilaginous metaplasia is a rare but well-documented phenomenon occurring in the wall of odontogenic keratocyst. The mural cartilage not associated with odontogenic keratocyst has been reported only once in a maxillary teratoid cyst of congenital origin to our knowledge. A case presented is a 38-year-old man with intraosseous keratinizing epidermoid cyst in the mandible, the wall of which contained a nodule of mature hyaline cartilage. The present lesion likely represents a previously undescribed, histologic hybrid consisting of orthokeratinized odontogenic cyst and cartilaginous heterotopia.     

Repair of a thoracoabdominal aortic aneurysm in the presence of a left-sided inferior vena cava

Venous anomalies are not infrequently encountered during aortoiliac reconstruction, because of the complexity of development of the venous system. Retroaortic left renal veins, duplicate inferior vena cava (IVC), and left-sided IVC are occasionally found. Left-sided IVC has been reported with infrarenal aortic aneurysms. We report successful repair of a thoracoabdominal aneurysm in a patient with a left-sided IVC. The embryology and intraoperative management are discussed.     

Noninflammatory pseudotumor simulating venous thrombosis after metal-on-metal hip resurfacing

Metal-on-metal hip resurfacing has become a widespread procedure, especially in young, physically active patients. Pseudotumor is a new complication that can occur after hip resurfacing and metal-on-metal total hip arthroplasty (THA). This article presents a case of a 37-year-old woman who underwent metal-on-metal resurfacing of the left hip for symptomatic osteoarthritis. Twelve months following implantation, the patient reported painless swelling of the left lower leg. There was no clinical evidence of a deep venous thrombosis. Ultrasound and computed tomography showed a solid cystic lesion in the iliopsoas muscle, which communicated with the hip joint and compressed the external iliac vein. As a consequence, the cystic lesion was resected marginally. A few months later, the patient reported some discomfort in the groin and symptoms of instability, metallic clicking, and a restricted range of motion. Clinical and radiological examination revealed normal findings. Determining the serum concentration of cobalt and chromium revealed high increased levels of these metal ions. Ten months following excision of the pseudotumor, the patient reported recurrent swelling of the left lower leg. Computed tomography of the affected area showed a lobulated cystic formation; therefore, a relapse was suspected. At the second revision, the mass was excised and the implant was revised to a conventional ceramic-on-ceramic THA. At 30-month follow-up, the patient was doing well and there were no signs of local recurrence. Additionally, the metal ion levels of cobalt and chromium in the blood had significantly decreased.     

Intraosseous ganglion with impending fracture of the glenoid

Intraosseous ganglia are benign cystic and often multiloculated lesions located in the subchondral bone. Only 13 cases around the shoulder have been described in the literature. These lesions are often asymptomatic, but, in cases located close to neurovascular structures or articular surfaces, they can be symptomatic, causing pain, neurologic dysfunction, or articular fractures. This article reports the clinical, radiographic, and pathological findings of a 34-year-old man reporting shoulder pain during heavy working activities. At physical examination, his left shoulder showed complete active and passive range of motion but was painful in external rotation and extension. Tests for the evaluation of the rotator cuff were all negative. The anterior apprehension test provoked pain. The Constant-Murley score was 59 points. Radiographs, computed tomography (CT) scan, and magnetic resonance arthrography showed an elliptical-shaped osteolytic area in the anteroinferior portion of the glenoid extending to the anteroinferior glenoid margin. Leakage of the intra-articular contrast medium showed that the cyst had eroded the articular surface and was about to cause an intra-articular fracture of the glenoid. The patient was operated on with the suspicion of an intraosseous ganglion. The cyst was curetted carefully and packed with autologous bone from the proximal tibia and artificial bone chips. At 1-year follow-up, the patient was completely asymptomatic and reported no pain during his working activities. Physical examination was negative. Radiographs and CT scan showed a cystic lesion in the anteroinferior margin of the glenoid that was partially filled by bone.     

Squamous-cell carcinoma arising from an odontogenic cyst--the importance of computed tomography in the diagnosis of malignancy.

Primary intraosseous carcinoma of the mandible is rare, and when it occurs often arises within or closely associated with an odontogenic cyst. The purpose of this article is to show the role of computed tomography (CT) in the early detection of carcinomatous changes, because of its specificity in establishing the nature of the lesion. An unusual case of a squamous cell carcinoma that arose in an odontogenic cyst is described. The appearance on the panoramic radiograph was suggestive of a benign cystic lesion in the left side of the mandible, but when a CT scan was performed, the appearance of the lesion was compatible with malignancy. Histopathological analysis confirmed the suspicion of a squamous cell carcinoma, but without CT investigation it would be impossible to demonstrate that carcinomatous change had developed from a cystic lesion.