Chronic otitis media or deep sensorineural deafness often develops due to ANCA (antineutrophil cytoplasmic antibody)-associated vasculitis such as granulomatosis with polyangiitis (GPA) and microscopic polyangiitis (MPA), and similar diseases of the ear has recently been termed otitis media with ANCA-associated vasculitis (OMAAV). The OMAAV-working group was subsequently organized in the Japan Otological Society and studied 90 cases with OMAAV. All patients complained initially of ear symptoms such as hearing loss, ear pain and/or otorrhea, which were not improved by treatments with antibiotics and tympanic tube insertion. At the initial visit, granulomatous lesions in the tympanic cavity and/or mastoid cavity were seen in 74 (89.2%). Serum PR3- or MPO-ANCAs were detected in 66 (78%) patients. Eighty three (92%) patients had sensory hearing loss. Facial nerve palsy, hypertrophic pachymeningitis, lung lesions and kidney lesions developed in 25 (28%), 18 (21%), 8 (9%) and 6 (7%) patients, respectively. Three (3%) patients died because of subarachnoid hemorrhage due to vasculitis of the basilar artery. When a lesion progresses, it is easy for concomitant complications to occur alongside OMAAV such as facial paralysis and hyperplastic pachymeningitis. The current diagnostic criteria of ANCA-related vasculitis cannot be used to diagnose cases of OMAAV. Even if a diagnosis is not made of an intractable upper respiratory tract lesion including the ear, it is necessary to bear OMAAV in mind when thinking about the AAV (ANCA-associated vasculitis)-related lesions. OMAAV cases are being accumulated by the OMAAV working group, and pathologic elucidation currently anticipated.
The opsoclonus-myoclonus syndrome (OMS) is characterized by subacute onset of opsoclonus, a disorder causing of saccadic eye movements causing involuntary, chaotic saccades that occur in all directions. We report herein on a 36-year-old man with mainly horizontal chaotic saccades after truncal ataxia. Intravenous corticosteroids pulse therapy was very effective and his opsoclonus disappeared. No malignant tumors were detected with positron emission tomography. We analyzed eye movement using electronystagmography (ENG) and considered this case to be one of non-paraneoplastic OMS.
Since Streptococcus pneumonia has severe pathogenicity and the incidence of penicillin-resistant S. pneumoniae has been increasing, it is important to identify S. pneumonia rapidly as the pathogenic bacterium to enable appropriate antimicrobial treatment. However, especially in the case of pediatric rhinosinusitis, S. pneumoniae is one of the normal inhabitants of the nasopharynxes, so it is difficult to identify it as a pathogenic bacterium. We investigated the usefulness of a pneumococcal antigen detection kit, RAPIRAN®S. pneumoniae HS (HS kit) for the detection of pathogenic bacteria associated with acute rhinosinusitis. We have evaluated 134 nasopharyngeal secretions (72 children, 62 adults), and have examined the HS kit, culture test and microscopic examination (Gram stein), and made comparisons among them. The positive rate of the HS kit was 38.9% in children, and 6.5% in adults, with a sensitivity of 65.2%, a specificity 97.7% and a concordance rate of 86.6%. Furthermore, in the culture test, the HS kit(+) dominant bacterium was S. pneumoniae, whereas the HS kit(-) dominant bacterium were Haemophilus influenzae and Moraxella catarrhalis. Based on our results, we concluded that the HS kit could be useful for the detection of pathogenic bacteria associated with acute rhinosinusitis, so that if the HS kit is positive, the pathogenic bacterium is probably S. pneumoniae, and if the HS kit is negative, the pathogenic bacteria are probably others, with the exception of S. pneumoniae.
The blood boil is a benign hemorrhagic pseudotumor that usually arises in the sinonasal tract, particularly the maxillary sinus and blood boils in the frontal sinus are very rare. We experienced a rare case of a blood boil in the frontal sinus. A 55-year-old female visited our hospital because of a swelling in her forehead and lateral deviation of the left eye. CT and MRI showed a large mass extending from the left frontal sinus to the orbital roof with bone erosion. Because of poor improvement with conservative medical treatment, we performed a frontal sinus operation via the transcranial approach and removed the tumor. Histopathologically, the tumor was composed of necrotic tissue with hemosiderin deposition and no malignant tissue, and it was diagnosed as a blood boil in the frontal sinus. No recurrence has been seen for about 1 year after the operation.
Castleman’s disease was first reported by Castleman et al. in 1954 as localized large benign hyperplastic mediastinal lymph nodes histologically characterized by hyperplasia of the lymphoid follicles and capillary proliferation with endothelial hyperplasia. We report herein on a rare case of Castleman’s Disease arising in a left parotid gland. A 11-year-old male patient complained of swelling of the left parotid lesion. Preoperative CT scan, MRI and echo findings suggested that the tumor was very hypervascular. As this lesion was clinically suspected to be a benign tumor of the parotid gland, superficial layer segmental resection of parotid gland was performed. After surgery, the tumor was diagnosed as hyaline-vascular type of Castleman’s disease. There is no evidence of recurrence in the patient 40 months after the operation.
Facial palsy is generally recognized as a symptom of malignancy among parotid tumors. We report herein on multiple Warthin’s tumors involving the parotid and parapharyngeal region accompanied with facial palsy. A 45-year-old man visited our clinic complaining of right parotid swelling. No other symptom was observed except the swelling. CT revealed multiple tumors involving the right parotid and parapharyngeal region. Fine needle aspiration cytololgy indicated Warthin’s tumor. He did not accept the proposal of surgical removal of the tumor. Six years and 8 months later, he had acute onset of right facial palsy and severe pain of the right half of the face. We performed surgery suspecting malignant transformation because of the accompanying facial palsy. The tumors were removed visibly by the cervical and parotid approach with a midline mandibulotomy (i.e.; mandibular push up method). The right facial nerve was preserved because of the lack of malignancy as demonstrated by the intraoperative rapid histopathological diagnosis. The postoperative histopathological examination failed to show malignant transformation of the Warthin’s tumors. No evidence of recurrence of the disease was observed and the facial palsy had almost regressed a year after the surgery.
We report herein on a case of minocycline-induced black thyroid with papillary thyroid carcinoma. A 60-year-old female, who had taken oral minocycline intermittently for osteomyelitis of the mandible for two years, noticed a palpable thyroid nodule. Fine needle aspiration of the nodule revealed atypical cells and the serum thyroglobulin level was high. Under the diagnosis of suspected thyroid carcinoma, a hemithyroidectomy with central compartment neck dissection was performed. The color of the tumor, which was diagnosed as a papillary carcinoma, was white. On the other hand, the color of the normal thyroid gland was dark red to black with minocycline pigmentation. Minocycline therapy may induce headache, nausea, vomiting, liver dysfunction and dental hyperpigmentation in children. But a minocycline-induced black thyroid is uncommon. Recently, there have been some reports that the risk of malignancy in black thyroid glands is higher than in non-black thyroids.
A 76-year-old man had been aware of the swelling of the posterior neck on both sides for a year. When he underwent health screening, an abnormal shadow in the mediastinum was found on CT imaging. The local findings at the first visit, revealed multiple swelling in the lateral and posterior neck with no tenderness. Lymphadenopathy from the mediastinum to the neck in the cervicothoracic was suspected from the CT findings. We removed a lymph node under general anesthesia, but the specimen lacked diagnostic characteristics. The lymph nodes of the submandibular gland were also removed. The specimen showed no structure of the lymph node but an eosinophilic lesion was observed. Congo red staining was performed. Based on the results of the general examination, a diagnosis of primary macroglobulinemia was made. Amyloidosis is a pathological condition in which amyloid with a fiber structure is deposited in the extracellular tissue of various organs. The present patient was diagnosed as having amyloidosis with underlying primary macroglobulinemia. Deposition of amyloid material is frequently found in the heart, tongue, skeletal muscle, and intestinal tract, but very rarely in the neck. The present case had no subjective symptoms other than cervical lymphadenopathy, and the diagnosis was made by biopsy. Reports of amyloidosis that was discovered through swelling of the neck are very scarce with only two cases in Japan, and five cases overseas.
The objective of the present study was to clarify the efficacy of proton pump inhibitor (PPI) therapy with esomeprazole on symptoms, signs, sleep disturbance and work productivity associated with laryngopharyngeal reflux disease (LPRD). Twenty one patients with LPRD were enrolled in present study and received esomeprazole 20 mg daily for two months. These patients with LPRD were investigated by comparing the frequency scale of gastroesohageal reflux disease (GERD), the laryngopharyngeal reflux symptom score, the objective laryngopharyngeal reflux finding score, the Pittsburg sleep quality index questionnaire and the work productivity and activity impairment questionnaire before and after treatment. We found that the scores of these parameters after treatment were significantly lower than those before treatment. These findings indicate that LPRD might have an influence on the quality of life of affected patients including sleep disturbance and work productivity. PPI treatment might improve the symptoms and signs of LPRD also reduce the sleep disturbance and restores the work productivity of patients with LPRD. Taken together, esomeprazole was shown to be useful for the treatment of patients with LPRD.