Methotrexate-associated lymphoproliferative disorder (MTX-LPD) is defined as an “other iatrogenic immunodeficiency-associated lymphoproliferative disorder (LPD)” in the WHO Classification, 4th edition, that develops in patients with autoimmune diseases such as rheumatoid arthritis (RA) and who are under methotrexate (MTX) treatment. One of the characteristics of this disease is that remission may be achieved only with the cessation of MTX in some cases. We report herein on four cases of head and neck MTX-LPD. The patients were in their sixties to eighties, and comprised three females and one male. They were suspected of having malignant lymphoma with symptoms like sore throat, neck swelling and others, during treatment for RA with MTX. Three cases were diagnosed as having diffuse large B-cell lymphoma and another was diagnosed as having classical Hodgkin lymphoma. Epstein-Barr virus (EBV) is supposed to be associated with the pathogenesis and prognosis of this disease. Remission was achieved in three EBV positive cases only with the cessation of MTX. Chemotherapy was needed in another EBV negative case. The number of MTX-LPD cases has been increasing along with the increasing use of MTX. Taking a patient history is very important for the diagnosis of MTX-LPD. When this disease is suspected, we should examine the EBV-related protein and should consider the cessation of MTX.
Foreign bodies in the tongue occur relatively rarely and if they are buried in the tongue, it is difficult to diagnose those with inspection only. In addition, some cases require instruments to find foreign bodies because there are no landmarks in the tongue. We report herein on a case of a wire foreign body buried in the tongue muscle layer. A 70-year-old woman had noticed pain in the tongue and at the right submandibular area since eating croquettes. She was referred to our department because CT imaging showed an intraoral high density area. Our CT imaging taken with the mouth open revealed the position of the foreign body in her tongue muscle layer. We surgically removed the foreign body from her tongue. During the operation, we checked the position of the foreign body using fluoroscopic imaging with some X-ray non-transmissive landmarks. This is a rare case of a piece of wire buried in the tongue muscle layer, and intraoperative fluoroscopic imaging was useful in identifying the position of the foreign body.
Patients with rheumatoid arthritis (RA) are known to develop lymphoproliferative disorders (LPDs) during the course of their illness, particularly in cases treated with methotrexate (MTX) for long periods. We describe herein a case of an MTX-related Epstein-Barr-virus associated lymphoproliferative disorder. A 80-year-old woman presented to the ENT service with a one-month history of a pain in the oropharynx. The patient had a history of RA and had been treated with MTX. On endoscopic examination, a tumorous lesion was observed in the oropharynx. A positron emission tomography/computed tomography (PET/CT) scan showed abnormal accumulation in the same area. Histological analysis showed variant cells, which stained positive for CD3, CD4, CD5, CD8, CD20, and CD79 and negative for CD56. The variant cells also stained positive for EBER-ISH. Taking those findings together, we diagnosed the patient as having methotrexate associated diffuse large B-cell lymphoma related to Epstein-Barr virus infection.
A man in his 70s presented at our department with a four-month history of dysphagia. The patient had been diagnosed as having hypopharyngeal cancer in another hospital, and chemoradiation treatment was started. On endoscopic observation of the pharynx, the entrance to the esophagus was severely occluded, and the insertion of the endoscope was difficult. We performed examination with the endoscope under general anesthesia, but the pharynx was occluded. No recurrence of cancer was noted. Taking those findings together, we diagnosed the patient as having pharyngemphraxis associated with chemoradiotherapy. A pharyngoplasty was performed under general anesthesia and dysphagia improved on the same day. However, the dysphagia gradually deteriorated. Endoscopic balloon dilation was therefore performed on four occasions under local anesthesia and the dysphagia improved. At 3 months postoperatively, the patient in now taking food orally. Therefore, the pharyngoplasty and endoscopic balloon dilation are one of adaptation for pharyngemphraxis associated with chemoradiotherapy.
Metastatic neoplasms in the thyroid gland are rare in clinical practice. Clear cell renal carcinoma is the most frequent site of origin of thyroid metastases and represents 12% to 34% of all secondary thyroid tumors. A man in his 70s presented to the ENT service with a one-month history of hoarseness and swelling of the heck. He had a history of renal clear cell carcinoma of the right kidney, which had been resected 20 years previously. CT showed an irregular region in the thyroid and tumor embolism in the jugular vein and brachiocephalic vein. A positron emission tomography/computed tomography (PET/CT) scan showed accumulation in the same area with a 4.8 standardized uptake value (SUV). Ultrasonography showed a mobile tumor embolism in brachiocephalic vein. Renal cell carcinoma was diagnosed with aspiration biopsy. A total laryngectomy and thyroidectomy were performed. The tumor embolism in the brachiocephalic vein was extracted and the jugular vein was removed. A histological examination after total thyroidectomy revealed clear cell renal carcinoma metastasis to the thyroid. Preoperative diagnosis of a secondary thyroid neoplasm is difficult to achieve. The diagnosis of metastatic disease should be taken into account if patients have a history of clear cell renal carcinoma.
Objective: In recent years, the opportunities to detect hypopharyngeal cancer in the early stage have increased thanks to advances in optical instruments. In this report, we discuss regional differences and the process of recent hospital visits in patients with hypopharyngeal cancer who visited our hospital. Methods: The subjects were patients with hypopharyngeal cancer who were referred to the department of Otolaryngology, Kagoshima University Medical and Dental Hospital during the period from January 2009 to December 2015. Results: Among 45 hypopharyngeal cancer patients from Kagoshima City, 4 (8.8%), 3 (6.6%), 13 (28.8%), 5 (11.1%), and 20 patients (44.4%) were diagnosed as having phase 0, I, II, III and IV, respectively. Among 75 patients from the mainland, other than Kagoshima, 6 (8.0%), 5 (6.6%), 12 (16.0%), 12 (16.0%), and 40 patients (53.3%) were diagnosed as having phase 0, I, II, III and IV, respectively. Among 23 patients from isolated islands, 0 (0%), 3 (13.0%), 2 (8.7%), 3 (13.0%) and 15 patients (65.2%) were diagnosed as having phase 0, I, II, III and IV, respectively. In the comparison between the groups, a statistically significant difference (p=0.04) was confirmed between the patients from Kagoshima City vs. those from the isolated islands. In the phase IV group alone, the number of patients who were referred by a department of otolaryngology was higher, while many patients with phase 0 to III were referred by other departments. Conclusion: In a statistical comparison, the number of patients with advanced cancer from the isolated islands was significantly higher than that of patients from Kagoshima City, suggesting a regional difference. Regarding the department that referred patients to our hospital, in the phase IV group alone, the number of patients referred by the department of otolaryngology was higher, while many patients in earlier stages were referred from other departments.
We report herein on a case of an intraparotid facial nerve schwannoma in a 62-year-old female. The patient had no facial nerve palsy or other specific findings before the operation. We treated the intraparotid facial nerve schwannoma with an intracapsular resection for preservation of the nerve function. After the operation, the patient developed facial nerve palsy, but 3 month later, the facial nerve function improved to the preoperative function. Intracapsular resection is therefore a useful technique for preservation of the nerve function in an intraparotid facial nerve schwannoma. Since intraparotid facial nerve schwannomas enlarge over the long term, we consider not only a follow up but also surgery after informed consent.
We report herein on a case of a fish bone foreign body that could be confirmed after a palatal tonsillectomy. A 71-year-old man presented with the main complaint of a sore throat after having eaten some fish. He underwent a medical examination at another hospital, but the presence of a foreign body could not be confirmed. However, because the sore throat persisted, he was admitted to our hospital after three days. We could not find any foreign body on normal examination. However, based on the CT imaging, the patient was diagnosed as having a fish bone foreign body in his throat. Since the fish bone foreign body seemed to be piercing the palatine tonsil, the patient underwent a palatal tonsillectomy. However, the foreign matter was not observed in the palatine tonsil. We then observed the operative field in detail, we were able to find the fish bone foreign body penetrating into the posterior pharyngeal wall, and we removed it.
A case of recurrent parotid gland swelling seen in characteristic sialography findings is reported. A 60-year-old woman consulted us with a complaint of recurrent swelling of the right parotid grand. The patient had bronchial asthma and was under medication prescribed by a local doctor. The bilateral parotid glands were swollen, with the right predominating. There were no abnormal findings in oral cavity but a jelly-like viscid saliva flowed from the bilateral Stensen's ducts. After the jelly-like saliva was excreted, the swelling of the parotid glands disappeared. Hematological examination showed no abnormalities in the levels of eosinophils and serum IgE, and did not suggest any autoimmune disease including Sjögren's syndrome and IgG4-related disease. MRI showed segmental dilatation of the bilateral parotid ducts. A sialogram actually revealed the right parotid duct was dilated, resembling a bunch of grapes and the left was segmentally dilated. We searched the literature, but were unable to find any characteristic sialogram resembling that of the patient's right parotid duct. As for the right parotid gland, sialoangiectasis was suspected from the sialogram, but did not lead to a definitive diagnosis. On the other hand, although secretion of fibrinous plugs containing numerous eosinophils was not observed, the possibility of sialodochitis fibrinosa was considered because the jelly-like saliva disappeared following the prescription of antiallergic drugs. This unique pathology requires further examination.
We investigated a nasopharyngeal carcinoma which showed cranial nerve symptoms at the first visit. The frequency of cranial nerve symptoms and problems regarding the treatment were examined. From 2002 to April 2014, among the 24 patients with nasopharyngeal carcinoma, 5 cases with cranial nerve symptoms at the first visit were treated for the first time, 3 men and 2 women, ages ranging from 28 to 74 years (median 49). The observation period was 2-42 months (median 29). Lower cranial nerve symptoms were seen in 4 cases and an optic nerve failure was found in 1 case. In 4 cases there were lower cranial nerve symptoms. The complication of pneumonia occurred in 3 cases except for 1 case in which anti-cancer therapy was not carried out. In these 3 cases anti-cancer therapy could be performed by nutritional management via a Percutaneous Endoscopic Gastrostomy (PEG). It is better to perform PEG prior to the start of treatment when we treat patients with nasopharyngeal carcinoma who demonstrate lower cranial nerve symptoms.
Squamous cell carcinoma is the most common malignant tumor of the tongue base, but minor salivary gland tumors are rare. The first-line treatment for minor salivary gland tumors, such as mucoepidermoid carcinomas, is radical resection. Many surgical approaches, including the transoral or external approach, are available for resecting tumors of the tongue base. Surgeons select the most suitable approach depending on the size, histological composition, and degree of invasion of the tumor. In the present case, a 77-year-old man with a 2-month history of dysphagia presented with a comparatively large tumor on the tongue base, which was biopsy-confirmed to be a mucoepidermoid carcinoma. CT and PET-CT revealed no lymph node swelling; therefore, the patient underwent tumor resection with a median glossotomy. The tumor was successfully removed by bisecting the tongue along the median raphe. The patient was discharged 18 days after the surgery. No adjuvant therapy was offered because histological examination of the specimen revealed it was a low-grade, mucoepidermoid carcinoma. At the 16-month follow-up, the patient reported no functional disorders or tumor recurrence. The median glossotomy is a very useful technique to approach tumors of the tongue base. Compared to other procedures, it is less invasive and does not require any special surgical support devices.
Neurofibromatosis type 1 (NF1), also known as von Recklinghausen disease, is an autosomal dominant neurocutaneous disorder characterized by neurofibromas, café-au-lait spots, etc. with an incidence of 1 in approximately 3000 individuals. NF1 also affects mesodermal tissues including blood vessels, and vascular abnormalities have been reported to be complications of NF1. These include aneurysms, arteriovenous malformations, and arteriovenous fistulae. We report herein on a rare case of a nasopharyngeal arteriovenous fistula (AVF) in a patient with NF1. A 17-year old male with a known case of NF1 presented with a refractory nasopharyngeal hemorrhage. 3D-CT angiography revealed an AVF from left ascending pharyngeal artery. The AVF was successfully treated with endovascular coil embolization. Postoperatively, there has been no hemorrhage and adverse effect for 1 year.
Objective: Hemorrhage is one of the most common and serious complications after tonsillectomy. The aim of the present study was to investigate the rate of post-tonsillectomy hemorrhage and to evaluate the clinical risk factors. Methods: We retrospectively analyzed 93 patients who underwent bilateral tonsillectomy in Hirosaki University Hospital. We defined post-tonsillectomy hemorrhage as detection of bleeding or coagulation in the tonsillar fossa. We investigated the clinical risk factors such as age, sex, body mass index (BMI), smoking habit, history of hypertension or diabetes, anticoagulant agents, surgeons and operative time. Results: The total incidence of post-tonsillectomy hemorrhage was 13 cases (14.0%), and 5 of those (5.4%) required reoperation under general anesthesia to stop the bleeding. The hemorrhages of the present study most frequently occurred on postoperative day 6, and half of them were after discharge from hospital. The rate of hemorrhage in males was significantly higher than in females, and male gender correlated with the rate of hemorrhage. There was no significant difference between the rate of hemorrhage and any other risk factors. Conclusions: In the present study, the rate of hemorrhage was 14.0%. Our study suggests that the clinical risk factor for post-tonsillectomy hemorrhage is male gender.
[Objective] Cronkhite-Canada syndrome (CCS) is a serious protein-losing digestive disease caused by polyposis in the digestive tract. Patients with CCS complain of symptoms of cavities, dysgeusia, onychodystrophy, and diarrhea. [Subjects and Methods] Four patients with CCS who were admitted to our hospital because of dysgeusia are reported in this study, consisting of 2 men and 2 women with a mean age of 61.0 years. Two taste tests and blood examination were performed and the course was observed in all cases. [Results] In 3 cases, assident symptoms except dysgeusia (e.g., weight loss, diarrhea, and general fatigue) appeared. Only dysgeusia was reported in one patient. Zinc levels in serum were 58.0, 64.3, 72, and 108.7μg/dL, respectively. All patients were taking polaprezinc. The results of the taste disc test were severe degradation in 3/4 patients. Alopecia, skin pigmentation, and onychodystrophy appeared in one patient who had complained of only dysgeusia within the 3 months since the first consultation. In the other patients, bloody stools and onychodystrophy also appeared within 3 months. All patients were treated with steroid therapy, 1-2 months after starting which their symptoms improved markedly. Polyposis was also markedly improved. However, the symptoms reappeared on the reduction of the steroid in 2/4 patients. Their first symptom on exacerbation was dysgeusia. [Conclusion] In previous reports, the first symptom of CCS was dysgeusia in 80% of patients with CCS. The symptom of dysgeusia could reflect the therapeutic effect and be an indicator of exacerbation.
We report herein on a case of intraoral penetration injury in an infant caused by a toothbrush entering the neck subcutaneously from the pharynx. A 1-year-7-month-old boy who was brushing his teeth before bedtime suffered intraoral penetration with his toothbrush after falling from an indoor jungle gym for infants. The patient was transferred to the emergency room in our hospital. CT scanning showed the toothbrush handle near the common carotid artery in the parapharyngeal space and its head situated subcutaneously near the neck. There was a problem in that the handle of the toothbrush prevented mask ventilation during general anesthesia. Under sedation performed with spontaneous respiration, the toothbrush head was cut and removed from the skin side with a nipper. The toothbrush handle was pulled out from the oral cavity. We were then able to introduced general anesthesia and perform skin dissection. The subcutaneous tissue was irrigated with warm saline. Considering that even a toothbrush can induce serious penetration injuries, it is important to perform treatment at a facility that is able to use a multidisciplinary approach.
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