Rare primary effusion lymphoma associated with HHV-8 in Japan.

We describe a 62-year-old man infected with human immunodeficiency virus (HIV)-1 who developed primary effusion lymphoma (PEL). Pleural effusion contained atypical lymphoid cells with human herpesvirus (HHV)-8 latent nuclear antigen (LANA)(+). Radiological examination revealed pleural and pericardial effusion, but no evidence of tumor mass or lymph node enlargement. The patient was administered with highly active anti retroviral therapy (HAART) and THP-COP therapy, resulting in complete remission. The prevalence of HHV-8 infection among HIV positive individuals is higher than in the general population in Japan. Although PEL is extremely rare in Japan, the incidence might increase in the future.

Multiple tiny granulomatous lesions with eosinophils in a patient with smoldering-type adult T-cell leukaemia: the possibility of a new type of bronchioloalveolopathy.

We herein describe the first case, to our knowledge, of pulmonary lesions characterized by necrotizing granuloma formation with eosinophils, in a patient with smoldering-type adult T-cell leukaemia. A 74-year-old man, diagnosed with smoldering-type adult T-cell leukaemia 1 year previously, was admitted due to repeated pyrexia and concurrent identification of diffuse small pulmonary nodules. A surgical lung biopsy specimen revealed inflammatory nodular lesions measuring up to 3 mm in size, which contained necrotizing and non-necrotizing granulomas, intraluminar organization and eosinophils, and were of unknown aetiology. The patient's condition, including the radiological findings, improved gradually without any treatment. This case suggests the possibility of a new type of human T-lymphotropic virus type-1 associated bronchioloalveolar disorder.

[Allergic bronchopulmonary mycosis induced by Schizophyllum commune--case report and review of the literature].

A 55-year-old man was admitted to our hospital because of pyrexia, cough and sputum. He suffered from bronchial asthma. Chest X-ray showed infiltrates in the left upper and right lower lung fields. Chest CT scans showed mucoid impaction and consolidation predominantly in the left upper lobe. Laboratory tests showed peripheral eosinophilia, elevated level of serum IgE, and the increased eosinophils in his sputum. Schizophyllum commune was isolated from the bronchoscopically-removed mucous plug. A diagnosis of allergic bronchopulmonary mycosis (ABPM) due to S. commune was made. Simultaneous daily administration of 400 mg itraconazole (ITCZ) and corticosteroid (prednisolone; 30 mg daily) provided sufficient improvement. However recurrence was recognized on chest CT scan findings one year later. There are not enough case reports concerning S. commune-induced ABPM to establish a therapeutic approach to the condition.

A Japanese family with multiple lung cysts and recurrent pneumothorax: a possibility of Birt-Hogg-Dubé syndrome.

We herein report a Japanese family lineage, possibly demonstrating Birt-Hogg-Dubé (BHD) syndrome. A 29-year-old nonsmoking woman was admitted to our hospital due to spontaneous pneumothorax. A chest CT showed multiple lung cysts, and breast cancer was simultaneously detected that needed priority surgical treatment. In the family history, the patient's father and half brother also experienced recurrent pneumothorax, and both had similar findings in their chest CT. In a genetic analysis of her half brother, the mutation of the BHD gene was identified. BHD syndrome is a rare autosomal and dominantly inherited disorder, which has three characteristics: multiple lung cysts that may be associated with pneumothorax, skin fibrofolliculomas, and renal neoplasm. For multiple-cystic disease of the lungs with an unknown etiology, or pneumothorax, as seen in a family history, it is necessary to consider the possibility of BHD syndrome.

[Sarcoidosis with epididymal and testicular lesions: case report and a review of the literature].

A 47-year-old man was admitted for further examination of uveitis. He had noticed scrotal swelling before his admission. A computed tomographic scan of the chest showed hilar and mediastinal lymphadenopathy, multiple micronodules and thickening of the interlobular septum, and these findings were consistent with sarcoidosis. Bronchoalveolar lavage fluid showed lymphocytosis. Gallium-67 scintigraphy revealed an abnormal accumulation in the hilar and mediastinal lymph nodes and in the bilateral scrotum. The resected and biopsied specimens of the epididymis and testis demonstrated numerous noncaseating epithelioid cell granulomas but no evidence of neoplasm. Therefore, systemic sarcoidosis was diagnosed. A review of the Japanese literature found most cases to be associated with a history of painless scrotal swelling with chest roentgenogram findings of stage I or II, while also indicating it was important to perform biopsy or surgically resect any epididymal and testicular lesion.

[Pulmonary adenocarcinoma with facial nerve palsy as the first sign of onset due to a metastatic temporal bone tumor].

A 70-year-old woman was admitted because of left facial nerve palsy. Brain magnetic resonance imaging showed a mass lesion with a well-enhanced margin in the left temporal bone. A computed tomographic scan of the chest showed a lung mass in the left lower lobe, which was thereafter diagnosed as adenocarcinoma. An immunohistochemical examination of the tissue specimen obtained from the left temporal bone revealed evidence of metastatic adenocarcinoma from the lung. No other metastatic lesions including separate site of bone were seen. This is a very rare case of lung cancer with facial nerve palsy as the first sign of onset due to a metastatic temporal bone tumor.

[Familial summer-type hypersensitivity pneumonitis in a husband and wife].

We encountered a family in which two of four members, the husband and his wife, had summer-type hypersensitivity pneumonitis at the same time, about two months after they moved to the residence. A 45-year-old man had cough, fever and exertional dyspnea. Chest computed tomography showed diffuse centriloblar ground-glass attenuation in both lung fields. His 43-year-old wife had chest small nodular shadows and similar symptoms to his husband. Serum anti-Tricosporon cutaneum (T. asahi: serotype II and T. mucoides: serotype I) antibodies of both patients were at the positive level. They were given diagnosis as summer-type hypersensitivity pneumonitis by radiological, serological and histological examinations. The symptoms in both cases were improved immediately after administration of systemic corticosteroid. Summer-type hypersensitivity pneumonitis was assumed to be caused for about two months duration of expousure to antigen.

[Case of toxocariasis showing migratory nodular shadows with halos].

A 30-year-old man, who had kept a dog for nine years and often ate raw beef liver, visited a hospital because of a chest nodular shadow in the left lung field found on a checkup examination. Chest computed tomography obtained 8 days after the checkup showed no abnormal shadow in the left lung but two nodular shadows with halos in the right upper and lower lobes. Peripheral blood eosinophil counts and serum IgE values were elevated. Immunological examination including microplate ELISA showed a high titer of specific antibody against Toxocara canis in the serum. He was successfully treated with albentazole. Parasitic disease, especially toxocariasis, is an important consideration in the differential diagnosis of migratory nodular shadow with a halo on chest computed tomography, and serology is useful in diagnosis screening.