Symmetric Lipomatosis Arising in the Tongue Presenting as Macroglossia and Articulatory Disorder.

Symmetric lipomatosis is definitively characterized by symmetric, tumorous lipomatous proliferation of adipose tissue that often develops in the head and neck, shoulders, and upper trunk. However, in the oral region, symmetric lipomatosis of the tongue (SLT) is an extremely rare condition related to generalized lipidosis that is often caused by chronic alcoholism. It is characterized by multiple symmetric lipomatous nodules and diffuse bilateral swelling located within the tongue. We report an extremely rare case of SLT arising in an 80-year-old man with a long history of alcoholic liver cirrhosis. He exhibited multiple soft nodular protrusions on the bilateral margin of the tongue presenting as macroglossia for years. Although MR imaging showed multiple fatty masses on both sides of the tongue, there was no elevated tumor mass on the bilateral margin. The patient underwent bilateral partial glossectomy under general anesthesia. Histopathologically, the resected tumor exhibited diffuse infiltration with mature adipose tissue lacking a fibrous capsule. Due to the lipidosis and the unusual presentation of multiple lesions, the lesion was ultimately diagnosed as SLT. At present, after surgery, the patient wears a full-denture and is in excellent condition, with no sign of recurrence, improved QOL, and recovery of masticatory, articulatory, and speech intelligibility functions.

Massive odontoameloblastoma arising in the maxilla: a case report.

INTRODUCTION: Odontoameloblastoma is an extremely rare mixed odontogenic tumor with both epithelial and mesenchymal components. The term odontoameloblastoma first appeared in the 1971 World Health Organization classification (Pindborg JJ., et al.) and is defined as "a neoplasm that includes odontogenic ectomesenchyme in addition to odontogenic epithelium that resembles an ameloblastoma in both structures and behavior." Because of the aggressive nature and risk of recurrence of the tumor, complete resection is essential. In this report, we describe an extremely rare case of a patient with massive odontoameloblastoma arising in the maxilla and occupying maxillary sinus. CASE PRESENTATION: In 2013, an 11-year-old Japanese boy was referred to our department for a painless and large mass of the right maxillary region. A panoramic X-ray showed a unilocular cystic lesion in the right maxilla containing a calcified mass in the lesion associated with an impacted tooth. Computed tomography showed a cystic lesion that included calcified structures and measured 3.6×3.1×2.7 cm. In 2013, the patient underwent tumor extirpation combined with impacted tooth extraction. The histopathological diagnosis was an odontoameloblastoma. No recurrence was noted 27 months after the operation. CONCLUSIONS: The patient has undergone postoperative occlusal guidance and functional orthodontic treatment, and his postoperative condition is excellent. However, postoperative recurrence or malignant transformation can occur in cases of odontoameloblastoma, and close long-term follow-up will be continued for our patient.

Massive Neurilemoma of the Hard Plate in Which Preoperative Diagnosis Was Difficult.

The patient was an 84-year-old man who was referred to our hospital in mid-December 2012 for a close examination of a mass arising from the left side of the hard palate that was found by a local dentist. The initial examination revealed the presence of a 3.0-cm elastic soft, dome-shaped mass in the left hard palate. CE-CT showed a lesion of size 1.8 × 1.4 cm in the right hard palate, which extended upward and invaded the nasal cavity. The mass was a solid tumor associated with resorption of surrounding bone and expansion of the greater palatine canal. CE-MRI indicated that the mass extended upward and invaded the nasal cavity, and the mass showed hypointensity on T1-weighted images, hyperintensity on T2-weighted images, and an irregular margin with internal enhancement. Abnormal uptake of FDG on PET-CT (SUVmax = 5.2) was observed in the left hard palate. The biopsy site lesion rapidly increased in size and biopsy was performed again in January 2013 due to suspicion of a malignant tumor. The histopathological diagnosis was a suspected malignant neurogenic tumor. Therefore, the patient underwent partial maxillectomy and a split-thickness skin graft in late February 2013. No recurrence was noted 29 months after the operation.

Methotrexate-associated lymphoproliferative disorder arising in the retromolar triangle and lung of a patient with rheumatoid arthritis.

We report an extremely rare case of massive methotrexate-associated lymphoproliferative disorder (MTX-LPD) arising in the retromolar triangle and lung of a patient with rheumatoid arthritis. The patient was a 75-year-old woman who was referred to our department because of severe pain associated with a unilateral ulcer on the left retromolar triangle. The tumor had an extranodal location in the retromolar triangle and in the right lung. A clinicopathologic examination found a lymphocytic infiltrate with increasingly atypical histopathologic features. Atypical large cells were strongly positive in Epstein-Barr virus-encoded small RNA in situ hybridization and in staining with CD20 antibodies. MTX-LPD was diagnosed based on the medical history and histopathologic results. The lesion responded well to withdrawal of MTX followed by R-CHOP (rituximab, cyclophosphamide, doxorubicin, vincristine, prednisone) chemotherapy. There have been no signs of recurrence for 4 years since withdrawal of MTX.

A case of Basal cell adenoma of the upper lip.

Basal cell adenoma is a rare type of benign salivary gland tumor found most commonly in the parotid gland. We present a rare case of basal cell adenoma arising in the minor salivary gland of the upper lip. The patient was a 59-year-old Japanese man who visited our department in December 2012 with a chief complaint of a mass in the upper lip, which had increased in size over several years. A mobile, elastic, and relatively soft mass without tenderness was palpable in the upper lip region. The mucosa of the upper lip covering the mass was normal. Tumor extirpation was performed under local anesthesia. Histologically, the tumor had a capsule and was composed of islands of relatively uniform, monotonous cells. Immunohistochemically, the inner tumor comprised tubuloductal structures that showed strong staining for CK7, while the outer tumor showed weak staining for CK7. The outer tumor cells also stained positively for CD10 and p63. The MIB-1 (Ki-67) labeling index was extremely low. Basal cell adenoma was diagnosed based on these results. The postoperative course was uneventful 12 months after surgery and there has been no recurrence.

Epidermoid cyst arising in the submandibular region.

Dermoid and epidermoid cysts in the oral cavity frequently develop in the midline or sublingual region of the floor of the mouth. Here, we report a rare case of an epidermoid cyst in the submandibular region. The patient was a 69-year-old man with a chief complaint of a mass in the right submandibular region. A mobile, elastic, relatively soft mass without tenderness was palpable in this region. The skin covering the mass was normal. MRI showed a cystic lesion measuring 3.5 × 3.0 cm under the platysma in the right submandibular region. Cystectomy was performed under general anesthesia. There was no adhesion to surrounding tissue and the right submandibular gland was preserved. The surgical specimen was cystic and contained soybean cord-like materials. Histopathologically, the cyst wall was lined by stratified squamous epithelium with no skin appendage, suggesting an epidermoid cyst. The postoperative course was uneventful and without recurrence after 28 months.

[Longitudinal assessment of articulatory and masticatory functions following glossectomy for tongue carcinoma].

The functions of articulation and mastication after glossectomy for tongue carcinoma were sequentially assessed. Longitudinal assessment of articulatory and masticatory functions was analyzed in 64 tongue cancer patients who underwent partial glossectomy (partial glossectomy group: 40 cases) or hemiglossectomy (hemiglossectomy group: 24 cases) in our department between January 2007 and August 2008. The mean age of the 64 patients (37 men, 27 women) was 56.9 years, ranging from 21 to 82 years. Tongue mobility was assessed using a tongue movement function test by Matsunaga's classification. Articulatory function was assessed using the monosyllable Japanese speech intelligibility test, while masticatory function was evaluated using color-changeable chewing gum and occlusal force score using Yamamoto's classification. The functions were evaluated preoperatively, and 1 month, 3 months, 6 months, and 12 months postoperatively. In all groups, both tongue movement and articulatory and masticatory functions were impaired 1 month after surgery, however they gradually recovered between 3 to 6 months and reached a plateau from 6 to 12 months. The values of the parameters in the partial glossectomy group and the hemiglossectomy group at 6 months postoperatively were respectively as follows : the tongue movement function scores were 6.9+/-0.4, 5.2+/-0.8; the speech intelligibility scores were 94.1+/-3.5%, 88.8+/-9.9%; the occlusal force scores by Yamamoto's classification were 5.3+/-0.9, 3.7+/-1.0; and the values of a* determined using the chewing gum were 13.8+/-2.4, 8.6+/-3.5. In addition, there was a correlation between speech intelligibility and the value of a*, and between speech intelligibility and occlusal force scores by Yamamoto's classification.