Oral dysesthesia is associated with a variety of abnormalities in the oral cavity, however the pathogenesis of the disease remains largely unknown. In order to help clarify this, we retrospectively investigated the characteristics and treatment effects in 51 oral dysesthesia patients who visited our department from 2011 to 2018. The investigation items were age, gender ratio, route of consultation, duration of symptoms, nature of abnormal sensations, systemic diseases, and treatment effects. The average age of the patients was 63 years (range of 20 to 86 years), and women accounted for 78%. The patient referral rate was 69%, and the highest number of referrals was from dental practitioners. The median duration of symptoms was 12 months (range of 1 to 360 months). More than half of the patients were aware of abnormalities in the entire oral cavity. There were 41 patients with systemic diseases, 6 patients had a history of reflux esophagitis, and 13 patients had some kind of psycho-neurologic disease. There were 13 cases of psychiatric neurological disorders; depression was the most common (8 cases). Many of the patients were elderly women, and the treatment methods used in our department were mainly brief psychotherapy and pharmacotherapy. Traditional Chinese medicine (kampo) was used in many cases, and many kinds of drugs such as anxiolytics were also used. The improvement rate was significantly higher in patients treated with Hangekobokuto than in those not treated (p = 0.04). However, the effect of the treatment was refractory in 31% (16/51) of cases. The effect of treatment was examined in terms of gender, age and disease duration. The improvement rate was compared between the two groups at the median of the disease duration and age. Although there was a tendency for men to have a lower rate of improvement than women, no statistically significant differences were found, including for the other two items. Comparison between the improved group and unimproved group using the Mann-Whitney U test for the three items of mean treatment period, disease duration, and age, showed a significant difference in the mean treatment period (p = 0.003).
Inflammatory myofibroblastic tumors (IMT) are spindle cell proliferations with a characteristic fibro-inflammatory appearance, originally referred to as an inflammatory pseudotumors or plasma cell granulomas. We report a case of IMT in the floor of the mouth of a 41-year old female. The patient had a tumor in the floor of her mouth, measuring 35 × 30 × 20 mm. Magnetic resonance imaging (MRI) showed a lesion with high signal intensity on T1-weighted images. We performed resection under local anesthesia. Histological examination of the surgically removed lesion showed that the proliferative spindle cell was found inside the lipoma. There was no evidence of recurrence after five years. IMT are usually benign, however there are also reports of recurrence and malignant transformation. Careful long-term follow-up using MRI etc. is thought to be necessary.
Oral hairy leukoplakia has been regarded as a lesion characteristic of human immunodeficiency virus (HIV) infection; however, many cases are reported in HIV-negative immunocompromised patients. A EBV-positive mucocutaneous ulcer was first reported by Dojcinov et al. in 2010 and was categorized as an EBV-associated lymphoproliferative disorder. The cause is EB virus reinfection associated with iatrogenic or age-related immunosuppression. We report a case in which OHL and EBVMCU coexisted. The case involved a 75-year-old woman who visited our department for the purpose of examining a white lesion on the right tongue margin. A smooth, thin, uniform white lesion with a shallow, flat, well-defined, painful ulcer was found on the right lingual margin. She had a medical history of rheumatoid arthritis, which was treated with oral methotrexate therapy. The white lesion was accompanied by an ulcer. We suspected leukoplakia, intractable ulcer, and methotrexate-related lymphoproliferative disorder (MTX-LPD), and performed a biopsy of her tongue. Results confirmed that the lesions were overlapped with the oral hairy leukoplakia and EBV-positive mucocutaneous ulcer. We considered iatrogenic immunosuppression as the cause, and methotrexate treatment was discontinued. The ulcer healed 4 weeks after methotrexate was discontinued, and the hairy leukoplakia thinned.
Rhabdomyoma is a rare benign tumor classified as either cardiac or extracardic rhabdomyoma. Extracardiac rhabdomyoma is histopathologically categorized into adult, fetal, and genital types. Here we report a case of a 61-year-old man with adult rhabdomyoma occurring in the tongue. He consulted us after noticing a mass in his tongue. Since the tumor was small (diameter: 5mm), an excisional biopsy was performed under local anesthesia. Histopathologically, cross-striation and jackstraw-like crystals were clearly observed in the H-E and PTAH stained specimen. Also proliferation of round and polygonal tumor cells with eosinophilic granules and glycogen-rich cytoplasm were observed in the H-E stained specimen. As for the immunohistochemical findings, the tumor cells were positive for desmin, vimentin and HHF-35, while negative for myoglobin. On the basis of these results, a diagnosis of adult rhabdomyoma was established. He has not suffered a recurrence in the six years following surgical resection.
The number of patients undergoing wrinkle removal using skin filler is increasing. A 71-year-old woman was referred to our hospital because of swelling of the right cheek, which was suspected of being an odontogenic infection. However, no obvious odontogenic infection was observed at the first visit, and the cause of the swelling was unknown. MRI revealed a T2 hyperintensity region in the bilateral buccal subcutaneous tissue. Therefore, her medical history was revisited, and a history of facial filler injection in both cheeks was discovered. A foreign body considered to be the facial filler was observed in an abscess in the buccal mucosa, suggesting that the phlegmon had been caused by an infection in the facial filler injected into her cheek.
Myasthenia gravis (MG) is an autoimmune disease in which autoantibodies against the nicotinic acetylcholine receptor (AChR) at the neuromuscular junction (NMJ) cause impaired neuromuscular transmission. The patient was a 64-year-old man with a history of thymectomy who underwent a partial glossectomy 14 months ago, with the chief complaints of dysarthria and dysphagia. We initially suspected tumor recurrence or brain metastasis. But he showed blepharoptosis, and we suspected MG thus performed an examination. Diagnostic tests (edrophonium test and ice test) and a laboratory test (serum anti-ACh receptor antibody titer) were performed at our hospital. Both diagnostic tests returned positive for MG, and the serum anti-ACh receptor antibody titer was 34.0 nmol/L. Based on these results, the patient was diagnosed with post-thymectomy myasthenia gravis (PTMG). His symptoms improved after prescription of prednisolone (corticosteroid) and pyridostigmine bromide (cholinesterase antagonist).
A 42-year-old woman diagnosed with acquired von Willebrand syndrome (AvWS) was referred to our department for pain in the left mandibular third molar. vWF-containing concentrates (Confact F®) were transfused before and after tooth extraction for hemostatic management. Although initial hemostasis was achieved, persistent postoperative bleeding was observed 3 days later. Therefore, additional hemostatic management using a mouth splint with gingival packing and increased transfusion of Confact F® was required. After this, no further bleeding was observed, and the administration of Confact F® was ceased 14 days post-operation. The stitches were removed 24 days post-operation. Two years and 11 months after the operation, the clinical course is good. The pathophysiology of AvWS has not been elucidated, and an effective treatment has not yet been established. Therefore, reliable hemostatic management in cooperation with hematology is necessary.