A case of intestinal T-cell lymphoma with repeated episodes of perforation

A case of intestinal cytotoxic/suppressor T-cell lymphoma with repeated episodes of perforation was seen in a 77-year-old Japanese male who was admitted complaining of severe abdominal pain accompanied by high grade fever. Surgery revealed diffuse peritonitis due to perforation in the sigmoid colon. After a loop sigmoid colostomy the patient recovered to somedegree. Seven days later, however, another perforation occurred. Several aneurysmal swellings accompanied by perforation in the small intestine, forming a mass of 5 X 5 cm, were seen. Although the patient tolerated this operation, generalized emaciation proceeded and the patient died 10 days late. Histological examination at the second operation showed diffuse medium-sized lymphoma of the Lymphoma Study Group (LSG) classification infiltrating the entire depth of the intestinal wall. Destruction of muscle cells was prominent. An immunohistochemical study using fresh frozen material was positive for CD3, and CD8. This marked destruction of the muscle layer by lymphoma cells may be associated with repeated episodes of perforation.     

A case of intestinal T-cell lymphoma with repeated episodes of perforation.

A case of intestinal cytotoxic/suppressor T-cell lymphoma with repeated episodes of perforation was seen in a 77-year-old Japanese male who was admitted complaining of severe abdominal pain accompanied by high grade fever. Surgery revealed diffuse peritonitis due to perforation in the sigmoid colon. After a loop sigmoid colostomy the patient recovered to some degree. Seven days later, however, another perforation occurred. Several aneurysmal swellings accompanied by perforation in the small intestine, forming a mass of 5 x 5 cm, were seen. Although the patient tolerated this operation, generalized emaciation proceeded and the patient died 10 days late. Histological examination at the second operation showed diffuse medium-sized lymphoma of the Lymphoma Study Group (LSG) classification infiltrating the entire depth of the intestinal wall. Destruction of muscle cells was prominent. An immunohistochemical study using fresh frozen material was positive for CD3, and CD8. This marked destruction of the muscle layer by lymphoma cells may be associated with repeated episodes of perforation.     

A case of primary Sj?gren syndrome with repeated purpura]

In Sj?gren syndrome, purpura is one of its various well known eruptions. Although this disease state is assumed to be based on hypergammaglobulinemia, the details of its mechanism are unknown. We experienced a case involving a female patient with primary Sj?gren syndrome showing repeated purpura on the legs, and examined her blood viscosity and histopathology. This girl developed Sj?gren syndrome and was admitted to our hospital at 12-years-old. She underwent steroid treatment because of aggravation of the xerosis state and prominent purpura on the legs. Hypergammaglobulinemia was improved during the course; however, purpura appeared repeatedly. Although her blood viscosity was slightly higher than normal, this had no relation to purpura and serum gamma globulin values. Skin biopsy revealed necrotizing angiitis. These results suggest that the purpura of this case was caused not only by hyperviscosity from the hypergammaglobulinemia but also involvement of vasculitis by the primary disease.     

A case of mumps pneumonia complicated with acute respiratory failure]

A 38-year-old male with swollen parotid glands was admitted to our hospital with dyspnea on effort. Positive serum IgM antibody for mumps supported a diagnosis of mumps. Computed tomography (CT) scan showed a ground-glass appearance in both lower lungs. On the third day of hospitalization, bronchoalveolar lavage demonstrated an elevation of both the total cell (2.0 x 10(6)/cc) and the lymphocyte count (83%), as well as a decrease of the CD 4+/CD 8+ ratio (0.4). Bronchial biopsy specimens revealed infiltration of lymphocytes in both the bronchiolar walls and the alveolar septa. These data were suggestive of mumps pneumonia. The patients suffered acute respiratory failure on the seventh day of hospitalization, which improved after 3 days of high-dose methyl prednisolone therapy. This is the first report of adult mumps pneumonia in Japan. As a result of the examination of the bronchoalveolar lavage and a bronchial biopsy of viral pneumonia which had been reported in the past, mumps pneumonia was a corresponding opinion to other viral pneumonia.     

A case of primary lung cancer complicated with hypothyroidism by diffuse thyroid metastasis]

A 56-year-old woman with hypothyroidism was admitted to our hospital because of an abnormal chest X-ray film. The chest CT showed a 3-cm spiculated tumor in the left lingual branch, and multiple nodular shadows with random distribution. Cervical CT showed diffuse enlargement of the thyroid gland with low density. The cytology of the tumor in the left lingular branch revealed adenocarcinoma, and ultrasound-guided fine needle aspiration cytology of the thyroid gland revealed adenocarcinoma, cytologically identical to the cells from bronchoscopic examination. Immunochemically, specimens obtained from both bronchoscopy and the thyroid gland were negative for thyroglobulin. Therefore, we diagnosed this case as primary lung cancer complicated with hypothyroidism by diffuse metastasis of the thyroid gland. Metastatic thyroid tumor is rarely diagnosed clinically. Furthermore, metastatic thyroid tumor complicated with thyroid dysfunction is extremely rare. We conclude that in patients with both diffuse thyroid swelling and thyroid dysfunction, the possibility of metastatic thyroid tumor should be considered.     

乙型肝炎后肝硬化合并原发性甲状腺功能减退症1例

肝硬化患者常可出现低T3综合征或低T3、T4综合征, 但肝硬化合并原发性甲状腺功能减退症者文献报道很少. 本文对1例静止性肝硬化合并腹水, 同时并发原发性甲状腺功能减退症, 因甲减临床表现不典型而误诊2年的少见病例报道如下.     

A case of pulmonary embolism following acute respiratory failure with hypercapnia]

A 18-year-old boy was admitted to hospital in an unconsciousness state as a result of taking a large dose of several psychotropic drugs simultaneously in an attempt to commit suicide. Blood studies revealed hypoxia (55.7 mmHg) and hypercapnia (59.7 mmHg). Hypoxia (74.3 mmHg) and hypercapnia (46.7 mmHg) were still present on the fourth day after admission, and the patient was becoming lethargic. Reduced vascular markings in the right upper lung field on chest roentgenogram in spite of hypercapnia suggested that the persistent hypoxia was the result of a pulmonary embolism. This diagnosis was supported by a perfusion defect on 99mTc-MAA scintigram and arterial obstruction in right pulmonary angiogram. Hypercapnia is an unusual finding in pulmonary embolism, and in this case was considered due to depression of respiration by psychotropic drugs.     

A case of miliary tuberculosis associated with acute respiratory failure during pregnancy

A case of miliary tuberculosis associated with acute respiratory failure during pregnancy was reported. A 39-year-old, 29-week pregnant woman was admitted to our hospital with complaints of nonproductive cough and fever on June 12. On admission, her temperature was 38.2 degrees C; pulse rate was 90/min., and blood pressure was 120/76 mmHg. Physical examination revealed moist rales at right lung basis. Chest X-ray showed small nodular infiltrates in right lower lung field. Laboratory data revealed positive CRP, accelerated ESR and increased level of alpha 2-globulin. The number of T-cells was markedly decreased (14/mm3). The PPD skin test was negative, and the sputum smears for acid-fast bacilli were negative. Suspected of bacterial or viral pneumonia, the patient was treated with antibiotics (CPM, EM and CAZ), which had no effects for her. On June 16, the Chest X-ray showed infiltrates throughout bilateral lung fields, and the patient became increasingly dyspneic. On June 18, the results of arterial blood gas, analysis under room air were: PaO2 26.7 Torr, PaCO2 29.0 Torr, pH 7.505. Because of severe hypoxemia, she was intubated and placed on a volume-cycled respirator. Hydrocortisone (1000 mg, daily) was added to treatment because ARDS was suspected. Since the smears of tracheobronchial secretions showed acid-fast bacilli on June 24, she was diagnosed to have miliary tuberculosis. Then the intensive therapy with antituberculosis drugs (isoniazid 400 mg, rifampicin 450 mg, and streptomycin 1g, daily) was started. The non specific antibiotics were discontinued; hydrocortisone was tapered and stopped. The next week, she became afebrile and hypoxemia steadily improved.(ABSTRACT TRUNCATED AT 250 WORDS)     

A case of nemaline myopathy with associated dilated cardiomyopathy and respiratory failure

Nemaline myopathy is a representative form of congenital myopathy, and is characterized by nemaline bodies in muscle fibers. Here we report a 47-year-old man with congenital nemaline myopathy complicated with dilated cardiomyopathy-related heart failure, and restrictive respiratory failure. The complication of dilated cardiomyopathy in nemaline myopathy has rarely been reported. In this case, nemaline bodies were detected in the cardiac muscle fibers, demonstrating the presence of underlying disease-related myocardial degeneration. The patient responded to the combination of conventional therapy for heart failure including β-blocker and noninvasive continuous positive-pressure ventilation for respiratory failure. His general condition has been stable during a 10-month follow up period.     

A case of sarcoidosis presenting with high fever and acute respiratory failure

A 55-year-old man was admitted with complaints of remittent fever (39 degrees C) and dyspnea on exertion which began ten days previously. His family and past histories were non-contributory for diagnosis except his occupation as a stone mason for 26 years. The chest X-ray film taken on admission showed diffuse small nodular shadows associated with small amounts of pleural effusion and bilateral hilar adenopathy. Arterihl blood gas analysis showed severe hypoxemia and hypocapnea (Pao2 32.2 Torr, Paco2 31.6 Torr). The serum level of LDH was 985 IU/L and ACE was 49.0 IU/L, lysozyme was 28.8 micrograms/ml. Biopsied materials of the lung obtained by TBLB, liver and bone marrow showed non-caseating epithelioid granuloma without caseating necrosis. T-lymphocyte ratio increased in BALF. The patient was diagnosed to have sarcoidosis. The administration of prednisolone was initiated, which resulted in a marked improvement of clinical data including chest X-ray films, BGA, LDH, ACE and lysozyme.