Diagnostic utility of cytology in a patient with Epstein-Barr virus-positive mucocutaneous ulcer in palatine tonsils: A case report.

Epstein-Barr virus-positive mucocutaneous ulcer (EBVMCU) is a newly established immunodeficiency-related disease. Herein, we report a case of EBVMCU and focus on its cytological usefulness for diagnosis. An 82-year-old man manifested pharyngalgia, dysphagia, and oral pain. His medical history included rheumatoid arthritis that had been treated with methotrexate. Clinically, peritonsillar abscess was suspected, but since neoplastic lesions, including malignant lymphoma (ML), could not be excluded, a series of cytohistological examination was attempted. Despite some alarming findings (e.g., frequent mitoses), fine-needle aspiration and touch imprint cytology consistently revealed a heterogeneous population of lymphoid and plasmacytoid cells with mild nuclear atypia. The final diagnosis of EBVMCU was established based on the permanent histologic specimen; however, retrospectively, cytology was more representative of the benign nature of the lesion than histology, helping a great deal to differentiate it from ML. Cytology can be a useful tool for the correct diagnosis of EBVMCU.

Congenital Membranous Stapes Footplate Producing Episodic Pressure-Induced Perilymphatic Fistula Symptoms.

Introduction: Recent third window syndrome studies have revealed that the intact bony labyrinth and differences in the stiffness of the oval and round windows are essential for proper cochlear and vestibular function. Herein we report a patient with a congenital dehiscence of the right stapes footplate. This dehiscence caused long-standing episodic pressure-induced vertigo (Hennebert sign). At the time of presentation, her increased thoracic pressure changes induced the rupture of the membranous stapes footplate. Perilymph leakage was confirmed by imaging and a biochemical test [perilymph-specific protein Cochlin-tomoprotein (CTP) detection test]. Case Report: A 32-year-old woman presented with a sudden onset of right-sided hearing loss and severe true rotational vertigo, which occurred immediately after nose-blowing. CT scan showed a vestibule pneumolabyrinth. Perilymphatic fistula (PLF) repair surgery was performed. During the operation, a bony defect of 0.5 mm at the center of the right stapes footplate, which was covered by a membranous tissue, and a tear was found in this anomalous membrane. A perilymph-specific protein CTP detection test was positive. The fistula in the footplate was sealed. Postoperatively, the vestibular symptoms resolved, and her hearing improved. A more detailed history revealed that, for 15 years, she experienced true rotational vertigo when she would blow her nose. After she stopped blowing her nose, she would again feel normal. Discussion: There is a spectrum of anomalies that can occur in the middle ear, including the ossicles. The present case had a dehiscence of the stapes, with a small membranous layer of tissue covering a bony defect in the center of the footplate. Before her acute presentation to the hospital, this abnormal footplate with dehiscence induced pathological pressure-evoked fluid-mechanical waves in the inner ear, which resulted in Hennebert sign. When patients have susceptibility (e.g., weak structure) to rupture, such as that identified in this case, PLF can be caused by seemingly insignificant events such as nose-blowing, coughing, or straining. Conclusion: This case demonstrates that PLF is a real clinical entity. Appropriate recognition and treatment of PLF can improve a patient's condition and, hence, the quality of life.

[ANALYSIS OF BACKGROUNDS AND LEVEL OF UNDERSTANDING OF TREATMENT OF POOR ADHERENCE AND DROPOUT CASES ON SUBLINGUAL IMMUNOTHERAPY FOR JAPANESE CEDAR POLLINOSIS IN THE FIRST FOLLOW-UP YEAR].

BACKGROUND: We considered the factors of poor adherence to and dropout from sublingual immunotherapy (SLIT) by verifying patient backgrounds 1 year after start of treatment. METHODS: We recruited 38 patients who began SLIT between November 2014 and September 2015. We analyzed their attributes and level of understanding of the treatment, and conducted a self-reported survey on factors behind dropout cases and poor adherence cases. RESULTS: Four patients dropped out 1 year after start of treatment. Three left for reasons related to anxiety about side effects. There were five cases of poor adherence. There was no significant difference between good adherence, poor adherence, and dropout regarding level of understanding of the treatment (p=0.59). In the comparison between good and poor adherence groups, except four dropout patients, the adherence tended to be poor in patients with short duration of disease, smoking patients, and young patients. Continuous rate of SLIT achieved about 90%, suggesting relatively high level of adherence. CONCLUSION: It appears possible that anxiety related to side effects could be a factor affecting dropout from SLIT. There was no significant difference regarding level of understanding of the treatment. The adherence tended to be poor in patients with short duration of disease, smoking patients, and young patients.

[Effect of posterior nasal neurectomy combined with the inferior turbinate surgery for Japanese cedar pollinosis].

INTRODUCTION: Up to 30% of the Japanese population suffers from Japanese cedar pollinosis, and some of these patients also have severe perennial allergies or other pollen allergies. Posterior nasal neurectomy has recently been reported as effective treatment for severe perennial allergic and intrinsic rhinitis. However, the efficacy of this surgery for seasonal allergic rhinitis has not been shown. In this study, the effectiveness of posterior nasal neurectomy combined with inferior turbinate surgery for patients with Japanese cedar pollinosis with concomitant intractable perennial allergies was evaluated with a questionnaire. SUBJECTS AND METHODS: Nine patients with Japanese cedar pollinosis, who also had perennial allergies, and undergone posterior nasal neurectomy combined with inferior turbinate surgery from April in 2005 to July in 2008, were enrolled. A numeric rating scale (NMR) was used to evaluate clinical symptoms in the pre- and post-surgery periods. The frequency of medication (oral administration and nasal spray) during the cedar pollinosis season was also evaluated. RESULTS: The NMR scores for all nasal and eye symptoms during cedar pollinosis season were significantly reduced after the surgery. The frequency of medication during the season was dramatically decreased after surgery. CONCLUSION: The present study demonstrates that posterior nasal neurectomy combined with inferior turbinate surgery is a highly effective treatment for Japanese cedar pollinosis. Since the Japanese cedar pollinosis season is limited to 2 to 3 months, such surgery is indicated for patients who suffer from severe perennial allergies and are also sensitized to some allergens such as Japanese cedar and ragweed pollen.

[Effect of posterior nasal neurectomy combined with the inferior turbinate surgery for the patients with perennial allergic rhinitis or vasomotor rhinitis].

INTRODUCTION: Until recently, Vidian neurectomy had been applied mainly in intractable vasomotor rhinitis and severe perennial allergic rhinitis. Although the results were excellent, the operation has not been applied recently because of the adverse events such as xerophthalmia and trigeminal neuralgia. To resolve these problems, a new surgical technique, posterior nasal neurectomy, was developed. In this report, we examined the effectiveness of posterior nasal neurectomy combined with the inferior turbinate surgery for severe perennial allergic rhinitis and intrinsic rhinitis by questionnaire. PATIENTS AND METHODS: Twenty patients who had undergone posterior nasal neurectomy combined with the inferior turbinate surgery between April in 2005 and March in 2009 were enrolled. Numeric Rating Scale was used to evaluate clinical symptoms and quality of life (QOL) of pre- and postsurgery. Frequency of medication (oral administration and nasal spray) was also evaluated. RESULTS: Questionnaires were collected from 17 patients. As for all patients but one, the surgery significantly reduced rhinorrhea, nasal obstruction, and sneezing. Furthermore, QOL for many symptoms such as sleep disorder and malaise/feebleness was also significantly improved after the surgery. Eighty-one percent of patients were satisfied with the surgery. CONCLUSION: Posterior nasal neurectomy combined with the inferior turbinate surgery is effective in alleviating clinical symptoms and improving QOL in the patients with severe perennial allergic rhinitis and intrinsic rhinitis, although a longer follow-up is needed.

Efficacy of combined treatment with S-carboxymethylcysteine (carbocisteine) and clarithromycin in chronic rhinosinusitis patients without nasal polyp or with small nasal polyp.

OBJECTIVE: In Japan, fourteen-membered ring macrolides, antibacterial agents, and S-carboxymethylcysteine (SCMC; carbocisteine), a mucolytic, are commonly used to treat chronic rhinosinusitis (CRS), and they are also used in combination. However, no large-scale randomized study has examined the effects of these pharmacotherapies. The aim of this study is to evaluate the effect of combined administration of clarithromycin (CAM), a fourteen-membered ring macrolide, and SCMC, compared with CAM single therapy. METHODS: Patients with CRS were centrally registered and randomly assigned to treatment with CAM (200mg/day) alone (monotherapy group) or CAM (200mg/day) in combination with SCMC (1500mg/day; combination group) for 12 weeks. We assessed the clinical efficacy of the treatments using measures of subjective symptoms and objective findings, health-related quality of life (HRQOL) determined by the 20-Item Sino-Nasal Outcome Test (SNOT-20) score and computed tomography (CT) score. RESULTS: Four hundred twenty-five subjects were enrolled (combination group, 213; monotherapy group, 212). At week 12 of treatment, the rate of effectiveness was significantly higher in the combination group (64.2%) compared with the monotherapy group (45.6%; P=0.001). In addition, objective findings, including characteristics of nasal discharge (P=0.008) and post-nasal discharge (P=0.002) were significantly improved in the combination group. In both groups, SNOT-20 and CT scores were significantly improved from week 0 (P<0.001), and were not significantly different between groups. CONCLUSION: The results indicated that long-term combination therapy with SCMC at a dose of 1500mg/day and CAM at a dose of 200mg/day is effective for improving subjective symptoms and objective findings in adult patients with CRS.

[IgG4-related Mikulicz's disease: a report of 3 cases].

INTRODUCTION: We report three cases of hyper-IgG4 disease with synchronous or asynchronous lymphocytic infiltration onset, IgG4 positive plasma cell infiltration, and fibril formation in multiple exocrine glands and extranodal organs. IgG4-related sialadenitis attracting recent attention has yet to be clarified as a clinical entity. CASE REPORT: Case 1, a 61-year-old man, had a submandibular gland sample showing IgG4-positive plasma cell infiltration. Case 2, a 61-year-old man, was diagnosed with IgG4-related Mikulicz's disease confirmed by a sublingual gland sample. Case 3, a 57-year-old woman, had a diagnosis of IgG4-related Mikulicz's disease confirmed based on labial and sublingual gland samples. All reported oral dryness and bilateral submandibular swelling. Cases 1 and 2 recovered following Predonine administration tapered from 30 or 20 mg. DISCUSSION: IgG4-related Mikulicz's disease must be recognized as a clinical entity, together with its diagnostic criteria and treatment. Sublingual gland biopsy should be done to confirm its diagnosis following sublingual gland swelling.

Caspase 3 activation in nasal capillary in patients with epistaxis.

OBJECTIVE: We investigated whether an apoptosis of nasal microvessels contributes to probable mechanism of the onset of epistaxis. STUDY DESIGN AND SETTING: Nasal septal mucosa of Little's areas taken from patients without (n = 19) and with (n = 26) epistaxis were examined. Active caspase-3 in the mucosa was detected according to the methods of immunohistochemistry and Western blotting. On Western blot analysis of the homogenates of the mucosa, we also sought probable signaling factors after caspase-3 activation. RESULTS: Marked activation of caspase-3 was detected in the capillaries and its neighboring muscle cells of Little's area from patients with epistaxis, and the activation was due to enhanced expression of procaspase-3 protein and progressive cleavage of the precursor. As a result of Western blotting of signaling factors, enhanced expressions of caspase-9 and Bax protein in the homogenates of Little's area in epistaxis group were found compared with those in control group. Increased levels of cytochrome c released into a cytosol were also detected in the capillaries in epistaxis group. CONCLUSION: In the present study, caspase-3 activation was found in the capillaries of Little's area from patients with epistaxis, suggesting that an apoptosis of capillaries may contribute to a mechanism of the onset of epistaxis. Moreover, alterations of some apoptotic factors such as caspase-9, Bax, and cytochrome c in the tissues demonstrated participation of mitochondrial disturbance in one of the apoptotic mechanisms. SIGNIFICANCE: Further explorations of the pathobiologic mechanism of capillary apoptosis can lead not only to an identification of risk factors in the onset of epistaxis but also to the development of medical therapy of epistaxis.