A 77-year-old man with a painless and bilateral submandibular swelling was referred to our hospital. He had already been treated with systemic prednisolone for retrobulbar neuritis. Laboratory examinations showed high serum IgG4 concentrations, and a biopsy of the submandibular gland revealed the infiltration of IgG4-positive plasmacytes. Abdominal CT demonstrated tumor formation in the tail of the pancreas. These findings suggested the patient to have Mikulicz's disease associated with localized autoimmune pancreatitis. Treatment with prednisolone improved the swelling of the submandibular gland. As a result, when a paticent is suspected to have Mikulicz's disease, it is important to consider the possibility of systemic IgG4-related plasmacytic disease.
We report a patient who was admitted because of a nail stuck in the mouth. The patient was a male carpenter in his twenties. While making a bookshelf at home, he accidentally shot a nail from his nail-gun into his left cheek. He came to the hospital 3 weeks after this accident, when the head of the nail protruded from his left cheek. On an X-ray film, the nail was found to be 90mm long. It was subsequently removed with the patient under general anesthesia. There have been no previous reports of nail injury to the cheek via the oral cavity and no reports about a nail 90 mm in length entering the cheek. The patient did not notice that a nail had penetrated his cheek at the time of injury. It was an unusual case because of the time that had elapsed until the foreign body was detected. The causes of foreign body injury are changing. We must acquire new knowledge to meet such changing needs.
Osteochondroma is an outgrowth of medullary and cortical bone, covered with a cartilaginous cap that projects from the cortical bone surface of the involved bone. In the mandibular region, solitary osteocartilaginous outgrowths of the coronoid or articular processes were reported previously. In this report, we describe the clinicopathologic characteristics of a case of osteochondroma arising in an articular process of the mandible. A 31-year-old man visited our clinic because of trismus. A radiographic examination showed a well-circumscribed ovoid radiopacity in the left temporomandibular joint. Computed and three-dimensional tomographic scans showed a large bony mass at the medial surface of the left condyle. The mass extended superiorly towards the cranial base. The patient underwent surgical extirpation with condylectomy. The resected specimen measured 45×30×21mm, and the histological diagnosis was osteochondroma. His occlusion was stable, and no functional disturbances have occurred as of 8 years after surgery.
Cavernous hemangiomas rarely arise in the submandibular gland. We report a case of cavernous hemangioma in the submandibular gland that had atypical clinical symptoms and imaging findings. A 27-year-old man presented with intermittent pain and swelling in the right submandibular region in August 2007. Three years 6 months ago the patient had had colicky pain during a meal and was given a diagnosis of submandibular sialolithiasis at another university hospital. Extraoral examination revealed an elastic soft mass with well-demarcated borders in the right submandibular region. On oral examination, salivation from the right Wharton's duct was normal, and other findings were unremarkable. MRI demonstrated a well-demarcated tumor in the submandibular gland, which showed low-signal intensity on Tl-weighted images and heterogeneous highsignal intensity on T2-weighted images. Enhanced CT showed a poorly-defined intraglandular lesion and enlarged submandibular and upper jugular lymph nodes. On the basis of these preoperative findings, a malignant tumor was included in the differential diagnosis. The submandibular gland was extirpated to remove the tumor with intraoperative frozen section examination in October 2007. The tumor was histopathologically diagnosed as a cavernous hemangioma with chronic sialadenitis. Factors such as the characteristic blood flow of the cavernous hemangioma and the intraglandular location of the tumor may have caused the atypical features of this case.
Acanthosis nigricans (AN) is an uncommon dermatosis with systemic brownish hyperpigmentation, velvety thickening of the skin, and verruca-like papules on the oral mucosa. It was classified into benign, pseudo, and malignant types by Curth in 1952. Generally, the benign and pseudo types are associated with endocrinopathies and obesity. The symptoms of these types are not as severe as those of malignant type. Malignant type is related to visceral malignant tumors. In addition, malignant type is frequently associated with oral mucous lesions. Although oral lesions have been reported in 25% to 50% of all AN cases, only a few articles have reported oral lesions associated with AN in the oral and maxillofacial region. In this report, we describe a case of AN with oral pallipomatous lesions without a visceral malignant tumor.
Extranodal NK/T cell lymphoma, nasal type, rarely arises in the oral cavity. We report the case of a 52-year-old man in whom the lesion originated in the left lower gingiva. The patient had slight pain and swelling at the left lower third molar region. Under a diagnosis of pericoronitis, the left lower third molar was extracted at a dental clinic. Because of a poor postoperative course, he was referred to our clinic. Intraoral examination revealed an ulcer and necrotic mucosa around the socket of the extracted third molar. CT scanning showed no bone resorption in the region around the extraction socket. Although the clinical findings suggested necrotizing ulcerative gingivitis, a biopsy revealed accumulation of atypical lymphocytes around the blood vessels. The biopsy specimen was positive for cytoplasmic CD3ε, Epstein-Barr virus encoded small RNA 1 in situ hybridization (EBER 1/ISH), and granzyme B, and negative for CD56 and CD20. Genetic screening of the frozen specimen was negative for T cell receptor gene rearrangement (TCR). On the basis of these findings, a definite diagnosis of extranodal NK/Tcell lymphoma, nasal type, was confirmed. The patient was treated with 50 Gy of radiation and 3 courses of DeVIC (CBDCA, VP-16, IFO, DEX) chemotherapy. His general condition remarkably improved, and there were no signs of recurrence after treatment. Because of the poor prognosis for patients with this type of tumor, prompt diagnosis and treatment are very important.
Idiopathic thrombocytopenic purpura (ITP) is a common hemorrhagic disorder in childhood but rarely occurs in infancy. We report on a 7-month-old girl who presented with swelling of the cheek. ITP was diagnosed on the basis of the history, physical examination, complete blood count, and CT scans. Because the platelet count was 0.4×104/μl, we referred her to a pediatrician. She was hospitalized immediately and was given human immunoglobulin (2g/kg). On the fourth day in the hospital, her platelet count returned to the normal range. She left the hospital on the seventh day. At present, 1 year has passed since treatment, and no recurrence has been detected. When ITP is discovered in infants with cheek traumatic injury, immediate diagnosis and treatment according to the acute childhood ITP guidelines are necessary.
Stimulant abuse is a social problem in recent years. We describe a patient with a history of stimulant abuse who had flashback phenomena during hospitalization for the treatment of a maxillofacial phlegmon caused by intraoral self mutilation. The patient was a man in his forties who continued to take stimulants and served a sentence in prison since 1 year before admission to our hospital. He had pain in the left cheek for 1 month before admission to our hospital and was referred to a certain hospital. He was given a diagnosis of maxillofacial phlegmon with intracranial abscess on computed tomography. The patient was referred to the Department of Emergency and Critical Care Medicine in our hospital for further examination and treatments. Because the maxillofacial phlegmon was apparently caused by intraoral infection, he was referred to our department. With the patient under general anaesthesia, we performed drainage of the left fossa infratemporalis, intraorbital region, fossa pterygopalatine, and the submandibular space on the day of admission to our hospital. Incomprehensible speech and self mutilation occurred after the drainage. Amphetamine psychosis with flashback phenomenon was diagnosed by a psychiatrist in our hospital. Antipsychotic drugs and a tranquilizer were prescribed, and his symptoms improved. His prognosis was good. He returned to his original hospital 20 days after surgery. In conclusion, it is very important to recognize the clinical condition of amphetamine psychosis. The treatment of such patients requires close cooperation with a psychiatrist.