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Streptococcus suis, a gram-positive facultative anaerobe commonly found in pigs, is an emerging zoonotic pathogen. Herein, we describe a case of a 45-year-old male Japanese meat wholesaler with S. suis meningitis and pyogenic ventriculitis. S. suis was isolated from his blood and cerebrospinal fluid culture, and sequence type (ST) and serotype were confirmed to be ST1 and serotype 2, respectively, by multilocus sequence typing and the Quellung reaction. Magnetic resonance imaging (MRI) revealed right labyrinthitis and pyogenic ventriculitis. The patient was treated with ceftriaxone and ampicillin for 24 days; the treatment was deemed successful based on negative blood cultures on day 4. However, the patient experienced hearing loss and a vestibular nerve disorder. S. suis is a rare pathogen in Japan but can cause severe infection and sequelae. To the best of our knowledge, this is the first report of a human case of pyogenic ventriculitis caused by S. suis. Our findings suggest that S. suis infection should be considered when hearing impairment is present in a patient with bacterial infection and that MRI can help detect ventriculitis, which can necessitate a prolonged treatment duration.

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IL-10-producing regulatory T cells (IL-10-producing Tregs) are one of the regulatory T cell subsets characterized by the production of high amounts of IL-10, the lack of FOXP3 expression and the strong immunosuppressive capabilities. IL-10-producing Tregs have been primarily reported as induced populations thus far, in part because identifying naturally occurring IL-10-producing Tregs was difficult due to the lack of definitive surface markers. Lymphocyte-activation gene 3 (LAG3) is a CD4 homologue that we have identified as being expressed on IL-10 producing Tregs. In human PBMC, LAG3 combined with CD49b efficiently identifies IL-10-producing Tregs. However, naturally occurring IL-10-producing Tregs in human secondary lymphoid tissue have not been described. In this report, we identified CD4+CD25-LAG3+ T cells in human tonsil. This T cell subset produced high amounts of IL-10 and expressed low levels of FOXP3. Surface markers and microarray analysis revealed that this is a distinct tonsillar CD4+ T cell subset. CD4+CD25-LAG3+ T cells expressed interleukin 10 (IL10), PR/SET domain 1 (PRDM1), and CD274 at high levels and chemokine receptor 5 (CXCR5) at low levels. CD4+CD25-LAG3+ T cells suppressed antibody production more efficiently than CD4+CD25+ T cells, and CD4+CD25-LAG3+ T cells induced B cell apoptosis. Moreover, analysis of humanized mice revealed that this cell subset suppressed a graft-versus-host disease (GVHD) reaction in vivo. Our study reveals the existence of naturally occurring IL-10-producing Tregs in human secondary lymphoid tissue and their function in immune regulation.

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A 74-year-old Japanese woman was admitted to our hospital because of fever, fatigue, and hearing loss associated with vertigo. She had a 1-year history of hearing impairment that got worse gradually and had been treated as otitis media with effusion, but without remarkable improvement. After admission, she developed renal dysfunction associated with hematuria and proteinuria. Laboratory tests showed leukocytosis and elevated C-reactive protein. Myeloperoxidase antineutrophil cytoplasmic antibody (MPO-ANCA) was elevated, but proteinase-3 antineutrophil cytoplasmic antibody (PR3-ANCA) was negative. Renal biopsy revealed pauci-immune focal necrotizing glomerulonephritis with crescents. She was diagnosed as having MPO-ANCA-associated polyangiitis. After treatment with 500 mg methylprednisolone applied intravenously for 3 days, followed by 40 mg prednisolone administered orally, renal function recovered completely. Her hearing also improved. Although otolaryngological symptoms are common in PR3-ANCA associated vasculitis, hearing loss is a rare manifestation of MPO-ANCA associated vasculitis (MPO-AAV). Our case suggests that AAV should be considered in the differential diagnosis of hearing loss.

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Distant metastasis from differentiated thyroid carcinoma is rare and metastasis to the paranasal sinus is extremely rare. We report a case of differentiated papillary thyroid carcinoma (PTC) with metastases to the sphenoid sinus presenting with epistaxis. An 81-year-old undergoing partial lobectomy for differentiated PTC 9 years earlier presented with intermittent epistaxis. Imaging showed a mass filling the sphenoid sinus and extending to the intracranial cavity. Histopathological diagnosis using endoscopic biopsy indicated a metastatic PTC tumor. Compared to the primary site, the metastatic tumor was poorly differentiated thyroid carcinoma. Despite external beam radiation therapy, her general condition worsened and she was moved to a hospice.

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We rarely encounter patients with proptosis, however, it is known that inflammation and extension of paranasal sinus tumors may cause this condition. We should consider the diseases that may cause proptosis to facilitate the diagnosis. Recently, we treated proptosis in 2 cases, one with inflammatory myofibroblastic tumor (IMT) and the other with malignant lymphoma. As these 2 diseases show various clinical features and these features frequently overlap, it is difficult to differentiate between IMT and malignant lymphoma without pathological examinations. In this study, we review the relevant literature reports and discuss the difficulties in establishing a clinicopathological diagnosis and present a slight distinction in magnetic resonance imaging.

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Multiple system atrophy (MSA) is a progressive neurodegenerative disease that is characterized by varying degrees of parkinsonism and cerebellar, corticospinal, and autonomic dysfunction. Vocal cord abductor paralysis (VCAP) is considered a sign of a poor prognosis in MSA, because it is a life-threatening complication that may cause nocturnal sudden death. This case report presents a patient who was treated for Parkinson's disease, and complained of dizziness and sleep apnea. We examined VCAP using fiberoptic laryngoscopy as the possible cause of sleep apnea. VCAP usually occurs in the advanced stages of MSA and is accompanied by a worsening of other symptoms. Optokinetic nystagmus was severely impaired and the caloric test response was bilaterally absent. Objective findings such as VCAP and abnormal neuro-otological results led to the diagnosis of MSA.

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We report a 67-year-old woman with small cell carcinoma of the hypopharynx, a very rare entity with few reports. Our treatment consisted of carboplatin (CBDCA) and etoposide (VP-16) in the same way as small cell carcinoma of the lung is treated. This achieved a complete response. Our case suggests that chemotherapy with carboplatin and etoposide is effective for small cell carcinoma of the hypopharynx.

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We report a patient with antithyroid drug-induced progressive bilateral sensorineural hearing loss associated with myeloperoxidase-antineutrophil cytoplasmic antibodies (MPO-ANCA). While antithyroid drugs have been linked to MPO-ANCA-associated small-vessel vasculitis, sensorineural hearing loss rarely was noted. A 36-year-old man treated for hyperthyroidism with propylthiouracil (PTU) developed progressive bilateral sensorineural hearing loss accompanied by fever and arthritis. MPO-ANCA were demonstrated in serum. Distortion product otoacoustic emissions test results suggested dysfunction of outer hair cells of the organ of Corti. Inner ear blood flow impairment from ANCA-associated small-vessel vasculitis presumably caused cochlear dysfunction. PTU withdrawal and high-dose methylprednisolone administration greatly improved hearing on both sides.

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A 63-year-old woman with rheumatoid arthritis sought medical assistance for dull and chronic pain in her left ear two and half years after her initial diagnostic examination. Otoscopic examination revealed that the posteroinferior wall of the bony external ear canal was eroded and that the small cavity was filled with squamous debris. The condition was diagnosed as external auditory canal cholesteatoma (EACC). The existence of EACC might suggest complications of bone disease, aging cerumen gland, or a low migratory rate of the epithelium.

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