Methotrexate-related lymphoproliferative disorder arising in the gingiva of a patient with rheumatoid arthritis.

Methotrexate (MTX) is the primary drug used in the management of rheumatoid arthritis (RA) and other immune-mediated inflammatory diseases. MTX is a strong immunosuppressive agent and has been reported to cause iatrogenic immunodeficiency-associated lymphoproliferative disorders (LPDs). Stomatitis caused by MTX-related cytotoxicity may occur, but gingival MTX-related LPDs are rare. In this article we present a case of gingival MTX-related LPD in a 60-year-old male with RA. The local findings of the gingival ulceration and alveolar bone exposure were similar to those of bisphosphonate-related osteonecrosis of the jaw. However, he had never received bisphosphonate therapy. The biopsy specimen of the gingival lesion was diagnosed as diffuse large B-cell lymphoma with Epstein-Barr virus positivity. Immediate withdrawal of MTX resulted in marked remission of the LPD.

Successful endovascular occlusion of an aneurysm of the cervical vertebral artery associated with neurofibromatosis-1.

A 30 year old female with a fusiform aneurysm of the cervical vertebral artery causing nerve root compression and associated with neurofibromatosis-1 was successfully treated with endovascular methods which resolved the mass effect. This is the first report demonstrating the reduction of the mass effect of an aneurysm on a cervical nerve root with endovascular treatment by using MR neurography.

Unsuspected small ameloblastoma in the alveolar bone: a collaborative study of 14 cases with discussion of their cellular sources.

BACKGROUND: Intraosseous ameloblastoma (IA) is the quintessence of epithelial odontogenic tumor and histologically and behaviorally defined as an undoubted neoplastic process. Current information must lead to the consensus that IA arises from the embryologic inclusions of odontogenic epithelium within the jawbone. Nevertheless, clinically oriented evidence is limited to this day. METHODS: The clinical and radiographic features, behavior, and pathology of 14 cases of small IA confined to the alveolar region were systematically examined. RESULTS: Six cases were a chance finding. There was no gender predilection and half of the lesions clustered in middle age (>40 years). The posterior region of the mandible (n = 7) and the anterior segment of the maxilla (n = 4) were favored. Five radiographic characteristics were recognized: interradicular (n = 5) and periradicular (n = 3), and periapical, residual and pericoronal (n = 2 each). They showed solid (n = 12) or unicystic (n = 2) growth pattern and 12 lesions were divided into seven follicular, three desmoplastic, and two plexiform subtypes. The main location of tumor was microscopically traceable in six cases; three interradicular type outside the periodontal ligament space and two periradicular and one periapical variants inside. CONCLUSION: By in-depth evaluation of the spatial relationship between tumor and its surrounding structure, the alveolar process, periodontal ligament space, and pericoronal area are all the likely starting points of IA. This report re-awakens the oral pathologist to the histogenetic significance of incipient IA as the only available human specimen for reappraisal of their origin.

Perivascular myoid tumors of the oral region: a clinicopathologic re-evaluation of 35 cases.

BACKGROUND: Myopericytoma (MPC) is a generic denomination to describe tumors showing differentiation toward perivascular myoid cells /myopericytes. It has been suggested that MPC forms a morphologic continuum with glomus tumor (GT), solitary myofibroma (SMF), and angioleiomyoma (ALM). This proposed relationship has not yet been assessed in the oral region. METHODS: We reviewed our 28-year experience with 35 oral tumors, originally diagnosed as ALM (n = 28), SMF (n = 4), GT (n = 2), and MPC (n = 1) to analyze their overlapping microscopic features, with the assistance of immunohistochemistry. RESULTS: Myopericytoma showed a wide range of growth patterns; concentric perivascular whorls, hemangiopericytomatous areas, glomangiopericytoma (GPC)-type vessels and leiomyomatous foci. Intravascular growth was also seen. Among 28 cases studied, three ALM were reclassified as MPC (n = 2) and SMF (n = 1), based on the present diagnostic criteria. Additional MPC-type components, at varying degrees, were similarly found in four ALM and three SMF, at least focally. One GT featured intravascular whorls of spindle cells. These four interrelated groups of tumors had in common GPC-type vasculature and intraluminal cellular proliferation was nearly ubiquitously present. Diffuse immunoreactivity for alpha-smooth muscle actin and less staining intensity of muscle-specific actin were observed in all tumors. Only ALM displayed desmin positivity of variable extent. Neither case tested expressed CD34. CONCLUSIONS: Our data matches with the recent results in extraoral sites that MPC, GT, SMF, and ALM exhibit histologic and immunohistochemical overlap with each other. A common perivascular myoid differentiation between these tumor types is further reinforced by the present oral series.

Hamartomatous proliferations of odontogenic epithelium within the jaws: a potential histogenetic source of intraosseous epithelial odontogenic tumors.

BACKGROUND: The jawbone is replete with a vestige of odontogenesis. The overall consensus is that intraosseous remnants of the enamel organ and dental lamina are the only histogenetic option for central epithelial odontogenic tumors. Curiously, incipient tumors or possible precursor conditions of residual odontogenic epithelium have rarely been reported in the literature. METHODS: We microscopically evaluated 39,660 biopsy samples to determine the presence of a tumor-like odontogenic epithelial nodule in the maxilla and mandible. RESULTS: Seven intraosseous specimens that associated with a focal proliferation of odontogenic epithelium were retrieved. Six hamartomatous processes showed four different morphologic patterns comparable with the tumor nests comprising ameloblastoma (n = 1), squamous odontogenic tumor (n=1), calcifying epithelial odontogenic tumor (n=2) and calcifying cystic odontogenic tumor (n=2). Among six lesions, four were the intrafollicular development. The remaining case of interest was multiple hyperplastic clear rests of Malassez in association with an impacted tooth. CONCLUSION: Although it is impossible to predict the fate of these microscopic structures of hamartomatous character, the present case series indicates that any of the dormant embryonic residues of odontogenic epithelium can return to an active state, capable of non-reactive, probably neoplastic proliferation of pathological significance.

Combined effects of a third-generation bisphosphonate, zoledronic acid with other anticancer agents against murine osteosarcoma.

Bisphosphonates (BPs) are widely used to treat bone diseases and also appear to possess direct antitumour activity. We have previously reported that third-generation BPs such as zoledronic acid (ZOL) and minodronic acid (YM529) synergistically augment the effects of anticancer agents in various cancer cells. Recently, we have also reported the antitumour effects of YM529 on murine osteosarcoma cells. As YM529 has not been clinically available, we herein focused on the anti-osteosarcoma effects of ZOL which is clinically available. In addition to ZOL alone, we evaluated the concurrent or sequential combined effects of ZOL with other anticancer agents against murine osteosarcoma cell lines. ZOL showed almost same anti-osteosarcoma activity compared with YM529 and more sensitive growth inhibitory effects against osteosarcoma cells than normal cells. Moreover, ZOL acted synergistically in vitro when administered concurrently with paclitaxel (PAC) or gemcitabine (GEM), not only in wild-type osteosarcoma cells but also in P-glycoprotein (P-gp)-overexpressing osteosarcoma cells, which were much less sensitive against each anticancer agent. Furthermore, 24 h of ZOL pretreatment significantly augmented the sensitivity of doxorubicin (DOX), PAC or GEM against osteosarcoma cells. These findings suggest that combined administration of ZOL with other anticancer agents may improve the osteosarcoma treatment.

Pacinian neuroma in adipose herniation of the buccal mucosa.

An interesting case of a trauma-induced tender mass of the buccal mucosa in a 45-year-old man was presented. Following surgery, the patient was relieved from pain. Microscopically, the mature adipose tissue is unique in that it contained a single enlarged Pacinian corpuscle near the deep margin. This is the hitherto undescribed intraoral lesion of Pacinian neuroma in the herniated buccal fat pad.

2D Thick-section MR digital subtraction angiography for the assessment of dural arteriovenous fistulas.

BACKGROUND AND PURPOSE: Although dynamic contrast-enhanced MR angiography studies for arteriovenous malformations (AVFs) and brain tumors have shown promising results, no formal attempt has yet been made to similarly evaluate dural AVFs. To assess the practical applicability of 2D thick-section contrast enhanced MR digital subtraction angiography (MRDSA) for the diagnosis and management of dural AVFs, MRDSA and intra-arterial digital subtraction angiography (IADSA) were comparatively evaluated. METHODS: We performed 80 consecutive MRDSA studies for 25 dural AVFs, including 11 cavenous sinuses, 9 sigmoid sinuses, 2 tentorial sinuses, one anterior condylar vein, one craniocervical junction, and one spine. MR images were continuously obtained following the initiation of a bolus injection of gadrinium chelates and subtraction images were constructed. We thereafter evaluated the imaging quality and hemodynamic information from all 46 MRDSA images performed in parallel with IADSA in either perioperative or follow-up studies. RESULTS: Most MRDSA images detected early venous filling, sinus occlusion, leptomeningeal venous drainage, and varices. It was difficult, however, to identify the feeding arteries because of both the partial volume effect and a low spatial resolution. Most important, MRDSA accurately detected aggressive lesions with leptomeningeal venous drainage and varices. CONCLUSION: Our MRDSA technique was found to have limited value for depicting all the anatomic details of dural AVFs, though it was able to identify important hemodynamic abnormalities related to the risk of hemorrhaging. MRDSA is therefore useful as a less invasive, dynamic angiographic tool, not only for perioperative studies but also for follow-up studies.

Peripheral odontogenic tumor: a clinicopathologic study of 30 cases. General features and hamartomatous lesions.

BACKGROUND: Peripheral odontogenic tumors (POT), either neoplastic or hamartomatous, are rare. This study briefly summarizes the general features of POT and selectively reviews the histomorphologic spectrum of under-recognized hamartomatous lesions that we have designated peripheral odontogenic hamartomas (POH) in order to shed more light into the pathogenesis of POT. METHODS: Archival material accessioned at our institutions between 1970 and 2004 was systematically searched to identify examples of POT/POH. RESULTS: Among 39 660 biopsies, we retrieved 25 cases of 'classical' POT and five cases of 'unique' POH. Odontogenic fibroma and ameloblastoma were by far the most common. Of POH, two purely epithelial lesions showed multiple strands of basaloid rests [odontogenic gingival epithelial hamartoma (OGEH)] and a conglomerate of polyhedral epithelium, ghost cells and concentric calcifications (calcifying epithelial odontogenic tumor-like hamartoma), respectively. OGEH and peripheral squamous odontogenic tumor (PSOT) deserve to be a related entity. In two types of mixed POH, ectomesenchymal elements appeared juxtaposed to the squamous lining (gingival cyst-like organoid hamartoma) and ghost cells aggregated in the enamel organ of a microdont (peripheral odontoma). None of POH exhibited continuity with the surface epithelium. CONCLUSION: On the basis of this relatively limited series of cases, POH, to conceptualize a unified histogenetic source, are speculated to arise from the soft-tissue remnants of dental lamina. Gingival rests of Serres seem to retain the ability to pursue epithelial-ectomesenchymal interactions that are necessary leading to odontoma formation.

Collagenous fibroma (desmoplastic fibroblastoma): a new case originating in the palate.

A 49-year-old woman presented with a painless slowly growing but huge polypoid palatal mass. On magnetic resonance imaging (MRI), areas of low signal intensity were consistent with a fibrous tumour. T(1) and T(2) weighted MRI after Gd-DTPA administration highlighted a well-circumscribed, purely soft tissue process. Histological analysis of the operative specimen detected collagenous fibroma (desmoplastic fibroblastoma). The patient remains disease-free 4 years after excision.

Suppressive effect of functional drinking yogurt containing specific egg yolk immunoglobulin on Helicobacter pylori in humans.

Helicobacter pylori is a human pathogen that infects over 50% of the population worldwide. It is the most important etiologic agent of gastroduodenal ulcers and malignancies. Helicobacter pylori urease enzyme is considered the main factor for the organism's colonization in the gastroduodenal mucosa. Hens immunized with the purified urease produce a highly specific anti-H. pylori urease immunoglobulin (IgY-urease) in their egg yolks. Immunoglobulin Y-urease was stable at 60 to 65 degrees C for 30 min and at pH 4.0 for 7 h. Its activity was lost at 80 degrees C for 20 min and at pH 2 for 4 h. Specially designed functional drinking yogurt containing Lactobacillus acidophilus and Bifidobacterium spp. with 1% egg yolk IgY-urease was produced commercially. Immunoglobulin Y-urease activity showed stability in the product up to 7 d, and then decreased to 85% after 3 wk of storage. A clinical study was conducted to determine the effectiveness of IgY-urease yogurt to suppress infection in humans. Forty-two volunteers who tested positive for H. pylori using a 13C-urea breath test were recruited. A total of 450 mL of IgY-urease (test group) or IgY-urease-free yogurt (control group) was consumed in 150-mL portions 3 times daily for 4 wk. Volunteers were tested after 2 and 4 wk; urea breath test values significantly decreased in the test group compared with the control group. The results indicate that suppression of H. pylori infection in humans could be achieved by consumption of drinking yogurt fortified with IgY-urease.

Subpontic cartilagenous hyperplasia of the mandible.

We report here a case of subpontic cartilagenous hyperplasia of the lower first molar of a 65-year-old woman. A sessile mass of calcifying hyaline cartilage firmly attached to the alveolar ridge was found below the pontic of a fixed partial denture. This is a novel type of reactive cartilagenous metaplasia in the subpontic tissue.

Basaloid squamous cell carcinoma of the oral mucosa: a new case and review of 45 cases in the literature.

Basaloid squamous cell carcinoma (BSCC) of the oral mucosa other than the tongue is uncommon. We report a case of a 67-year-old man who diagnosed with Stage I BSCC in the floor of the mouth. This early stage presentation carries a considerably better prognosis. Clinical summary of 46 cases of oral BSCC indicated that the tongue base was the most preferred site (61%). The patients were 19 males and 15 females with the mean age of 61 years (n=34). Most presented with Stage III or IV disease (62%). Even at the initial presentation, 47% had cervical lymph node metastases. Its aggressive clinical behaviour was characterized by a high incidence of local recurrence (32%), regional lymph node metastases (52%), and mortality rate (38%). Because of the advanced stage at presentation, oral BSCC is prognostically worse.

Asymptomatic brain tumor detected at brain check-up.

Brain check-up was performed in 4000 healthy subjects who underwent medical and radiological examinations for possible brain diseases in our hospital from April 1996 to March 2000. Magnetic resonance imaging revealed 11 brain tumors which consisted of six meningiomas, three pituitary adenomas, one astrocytoma, and one epidermoid cyst. The detection rate of incidental brain tumor in our hospital was 0.3%. Nine patients underwent surgery, with one case of morbidity due to postoperative transient oculomotor nerve paresis. The widespread use of brain check-up may increasingly detect asymptomatic brain tumors. Surgical indications for such lesions remain unclear, and the strategy for treatment should be determined with consideration of the patient's wishes.

[Cerebral arteritis and cerebritis caused by subdural empyema: two cases report].

We report two cases of cerebral angitis and cerebritis caused by subdural empyema. A 22-year-old man, who complained of a headache and high fever, suddenly developed unconsciousness and right hemiparesis. CT and MRI demonstrated left subdural empyema with diffuse brain swelling. CT angiography showed diffuse narrowing of the left internal carotid artery, middle cerebral artery, and anterior cerebral artery. Although we performed craniotomy, continuous irrigation with drainage, systemic injection of antibiotics for subdural empyema, antiplatelet therapy, and hyperbaric oxygen therapy for angitis, his condition remained unchanged. A 67-year-old man who had previously undergone burr hole surgery presented to our hospital for the treatment of scalp infection. He suddenly developed unconsciousness and right hemiparesis. CT and MRI demonstrated left subdural empyema with diffuse brain swelling, but MR angiography did not show any abnormal findings. Hemiparesis improved after the surgery and systemic injection of the antibiotics. Subdural empyema with sinusitis or meningitis around the skull base sometimes causes cerebral angitis. We considered that the angiographical evaluation for the subdural empyema was necessary to detect angitis.

Adenomatoid hyperplasia of the palate mimicking clinically as a salivary gland tumor.

This report describes an illustrative case of adenomatoid hyperplasia (AH) of the minor salivary glands on the palate of a 31-year-old man. The clinical features of the present lesion corresponded with those of pleomorphic adenoma, but histologically large lobules of normal-appearing mucous acini were found. The cell proliferative activity demonstrated in histological sections, by an immunohistochemical staining of proliferating cell nuclear antigen and Ki-67, showed no statistically significant differences among AH and a matched control group of normal palatal salivary glands. This case suggests that AH apparently exhibits an idiopathic, focal hypertrophic lesion of intraoral mucous glands with limited growth potential.

Traumatic herniation of the buccal fat pad.

Two new cases of traumatic herniation of the buccal fat pad are presented. The herniated fat pad was repositioned in case 1 and was excised in case 2. There was no recurrence after surgery in each case. Reviewing the literature, the lesion usually occurred in infants or young children under the age of 4 years as a result of traumatic penetration of the buccal mucosa and buccinator muscle with a foreign object.

Primary intraosseous carcinoma associated with impacted third molar of the mandible: review of the literature and report of a new case.

This study investigated the clincopathologic features of primary intraosseous carcinoma (PIOC) related to the third molar of the mandible. Six cases (5 from the literature and 1 new case; mean patient age 73 years) of PIOC associated with completely impacted lower third molar were analyzed. For comparison, 8 cases of PIOC presenting after extraction of the lower third molar were also evaluated, with a significantly lower mean patient age of 56 years. Most lesions were deceptively similar to those related to the lateral type of dentigerous cyst, and half of the impacted cases revealed an asymptomatic fracture of the mandible. In all patients except for the present case, metastasis had not been recorded. Surprisingly, no patients are known to have died of PIOC. In our experience the prognosis of PIOC associated with a completely impacted third molar is much better than commonly believed, although the number of cases involved is too small to make a general conclusion. However, delay in establishing the correct diagnosis followed by extraction without awareness of the carcinoma is know to adversely affect the prognosis.

Cellular hemangioma in an adult.

This report describes an adult case of cellular hemangioma arising in the lower lip. A 39-year-old healthy woman presented with a polypoid mass of 4 months duration. The tumor imparted little color to the overlying mucosa and was misdiagnosed as a mucous granuloma preoperatively. The lobular proliferation of plump endothelial cells with inconspicuous vascular spaces was a cardinal morphologic feature of the present tumor.