An adult case of cervico-mediastinal tuberculous lymphadenitis]

We reported a case of cervico-mediastinal tuberculous lymphadenitis followed by the development of pulmonary lesions and recurrent laryngeal nerve palsy. A 48-year-old man was admitted to our hospital due to fever, dry cough, and loss of body weight. He had no medical history of tuberculosis or other significant diseases. On admission, a right cervical tumor was palpable and chest X-ray films revealed widened superior mediastinum. Chest computed tomography showed multiple swollen mediastinal lymph nodes, including multiple low-density areas and contrast medium-enhanced septa and margins. Gallium-67 scintigraphy demonstrated abnormal uptake in the right cervix and mediastinum. A PPD skin test was strongly positive and ribosomal RNA of tubercle bacilli was detected in aspirated gastric juice. Although anti-tuberculous chemotherapy was initiated, fever and cough persisted, and hoarseness due to left recurrent laryngeal nerve palsy developed. One month later, chest X-ray films showed abnormal infiltration in the left upper lung field. The patient was sero-negative for HIV. To confirm the diagnosis, right supraclavicular lymph node biopsy was performed. Microscopic examination of the biopsy specimen revealed acid-fast bacilli and granuloma with central caseous necrosis surrounded by Langhans' giant cells and epithelioid cells. After 9 months of extended anti-tuberculous chemotherapy, the cervical and mediastinal masses receded and the abnormal chest X-ray shadows disappeared. Because the incidence of tuberculosis in Japan is gradually increasing among young people as well as the elderly, the differential diagnosis of this disease will become more necessary.     

肺放线菌病一例并文献复习

目的 提高对肺放线菌病发病机制、诊断、临床特点及治疗的认识.方法 报道2007年3月解放军总医院附属第一医院呼吸科收治的1例肺放线菌病病例并进行相关文献复习.结果 患者女,66岁.反复出现咳嗽、咳痰、低热4年,多次X线胸片均示右中叶肺炎,经支气管镜检查及病理诊断为肺放线菌病.经林可霉素联合磺胺甲嚼唑/甲氧苄啶治疗6个月后痊愈.结合本例并复习近20年国内外文献报道的187例肺放线菌病(国外162例,国内25例)资料,结果 显示本病男性多见,发病年龄为12～87岁;咳嗽、咳痰、胸痛为常见症状,咳黄色颗粒样物为典型表现,但并不多见.胸部CT可表现为实质团块影、片状影、实变影伴支气管充气征、空洞、纵隔淋巴结肿大、胸腔积液及胸膜增厚等.病变部位以左下叶及右中叶居多,与肺癌、肺结核易混淆.确诊主要依靠经支气管镜活检病理及手术病理检查,痰涂片及培养阳性率低.青霉素、四环素、红霉素、磺胺类或林可霉素等抗生素治疗及手术切除是主要的治疗手段.结论 肺放线菌病是一种少见病,早期诊断和治疗可获得良好疗效.     

A case of smoldering adult T-cell leukemia complicated by various pulmonary infections]

A 46-year-old man was admitted to Oita Medical College Hospital on October 16, 1987, because of cough and sputum. Chest X-ray and chest CT films showed diffuse reticulonodular shadow. The specimens obtained by transbronchial lung biopsy revealed cysts of pneumocystis carinii. Abnormal lymphocytes with lobulated nuclei were found 2-7% of peripheral leucocytes. The anti HTLV-I antibody was positive. According to these data, we diagnosed the patient as smoldering adult T cell leukemia with pneumocystis carinii pneumonia. The abnormal shadow on chest X-ray disappeared after SMX-TMP and pentamidine treatment. After about 1 year, he was again admitted for high fever. Chest X-ray showed infiltration with cavity in right upper lobe. Streptococcus pneumoniae was isolated from the sputum. The infiltration shadow on chest X-ray disappeared after antibiotics treatment. However, multiple nodular shadow appeared on the chest X-ray and ATL cell infiltration was found in the specimens of transbronchial lung biopsy. ATL cells in peripheral blood also increased and serum LDH and Calcium levels were markedly high. According to these data, we diagnosed the patient as having a ATL crisis. Although chemotherapy for ATL was started, the ATL, cell infiltration shadow on the chest X-ray enlarged, and bilateral diffuse patchy shadows was appeared on the chest X-ray. He died of respiratory failure on April 26, 1989. Cytomegalovirus pneumonia and ATL cell infiltration were revealed by necropsy.     

An adult case of cervico-mediastinal lymph nodes tuberculosis followed by the development of pulmonary lesions during the treatment with antituberculous drugs

A 27-year-old man was admitted to our hospital due to a painful mass in the right neck and fever. Cervical and superior mediastinal computed tomography showed an enlargement of right supraclavicular lymph node and multiple swollen mediastinal lymph nodes, including low-density areas and contrast medium-enhanced septa and margins. Smears of the pus obtained from right supraclavicular lymph node showed acidfast bacilli identified as Mycobacterium tuberculosis by PCR method. He was treated with antituberculous drugs with INH, RFP, EB, and PZA. PZA was given for initial two months. Six months later, productive cough developed and chest X-ray films showed infiltrative shadow in the right upper lung field. One month after the onset of cough, bronchoscopy revealed a polypoid lesion with a white coating in the right main bronchus. Microscopic examination of the specimen obtained by transbrochial biopsy revealed many epithelioid cell granulomas, consistent with tuberculosis. From these findings, pulmonary lesion was suggested to be due to invasion of the mediastinal lymph node into the bronchus. After one year of antituberculous chemotherapy, the swelling of the cervical-mediastinal lymph nodes was reduced and the abnormal chest X-ray shadows disappeared.     

A possible mechanism of primary ciliary dyskinesia: a case of a segmental defect in ciliary microtubules.

We report here a 13-year-old woman with cough, sputum and fever. The patient had both chronic sinusitis and bronchitis. Chest X-ray and computed tomographic scan of the chest revealed mucous bronchial filling and bronchiectasia in bronchi of bilateral lower lobes, right middle lobe and left upper lobe. Aerosol inhalation scintigraphy with 99mTechnetium demonstrated delays of the discharged tracer. On the basis of these findings, primary ciliary dyskinesia was suggested. This was confirmed by the findings from nasal biopsy with transmission electron microscopy where all of the microtubules were segmentally defected near the basal body in the cilia. On the basis of these findings, we diagnosed the patient with primary ciliary dyskinesia which may be due, at least in part, to segmental defect of ciliary microtubules.     

Eosinophilia and cough induced by resumption of cigarette smoking in a beginning smoker recovering from acute respiratory failure]

A 23-year-old man was admitted to the hospital because of acute respiratory failure accompanied by high fever and severe cough. He had started smoking about 3 weeks earlier and had gradually increased the number of cigarettes he smoked each day. After the number of cigarettes reached 10 per day, the patient experienced cough with serous sputum, high fever (38 degrees C) and dyspnea. Chest X-ray films revealed diffuse, peripheral shadows predominantly in the right lung. The patient was treated with an intravenous drip infusion of antibiotics (FMOX). On the second day of hospitalization, the dyspnea and chest X-ray shadows rapidly resolved. Although the cause of respiratory failure and diffuse shadows on chest X-ray films remained unclear, the patient was discharged. However, on the day of discharge, he smoked cigarettes and experienced cough with serous sputum for a few hours the following morning. He was readmitted for evaluation. We previously reported a case of smoking-induced acute eosinophilic pneumonia (AEP). We suspected that the present case was AEP induced by cigarette smoking but did not perform bronchoalveolar lavage or transbronchial lung biopsy for a definitive diagnosis. Although we did perform a provocation test, the patient demonstrated no respiratory symptoms without cough and respiratory failure. Blood gas analysis and pulmonary function tests also revealed no abnormalities. However, the eosinophil count (3066/mu/l) and percentage (42%) in peripheral blood increased after the patient resumed cigarette smoking, and returned to normal ranges after smoking cessation. These results pointed to a correlation between cigarette smoking and eosinophilia. This case suggested that in some patients, AEP may be induced by cigarette smoking, but does not recur after the resumption of smoking.     

A case of pulmonary eosinophilic granuloma with involvement of the ocular fundus]

A 37-year-old man was admitted to our hospital because of abnormal shadows on chest X-ray film at routine medical examination. The chest X-ray showed diffuse granular shadows in the bilateral upper and middle lung fields. Chest CT showed multiple cystic lesions and nodular lesions. Bronchoalveolar lavage (BAL) and transbronchial lung biopsy (TBLB) were performed. Electron microscopic examination of BAL fluid revealed Langerhans' cells, and lung tissue specimen obtained by TBLB showed granuloma with infiltration of histiocytes and eosinophils. During the clinical course, the patient complained of cough and decrease of right visual acuity in association with an increase of abnormal shadows on chest X-ray. The lung disease gradually resolved after the cessation of smoking in spite of no therapy. In the right eye, an exudative lesion with retinal elevation was noted in the ocular fundus. Corticosteroid administration was effective for the treatment of this lesion. There was no pathological evidence of the right ocular lesion. However, the correlation of its clinical course with the lung disease suggests a relationship between the ocular lesion and pulmonary eosinophilic granuloma.     

A case of plasma cell granuloma showing rapid growth and elevation of serum CEA

A 67-year-old man was admitted with complaints of cough and hemosputum. Chest X-ray examination revealed enlargement of a coin lesion in the right upper lobe, which had been pointed out about one year previously and had been followed up. Although the histology of TBLB specimens and the cytology of sputum and materials showed no malignancy and chest CT showed calcification at the edge of the coin lesion, the mass shadow in the right upper lobe rapidly enlarged and the serum level of CEA gradually elevated. Therefore, it seemed to be impossible to neglect the possibility of lung cancer and right upper lobectomy was performed. The dissected specimen was diagnosed as plasma cell granuloma. Because the histology of the plasma cell granuloma is multifarious, TBLB shows various results. It is therefore difficult to diagnose such inflammatory tumors by TBLB. The increase of the mass shadow in size and the elevated serum level of CEA made it difficult to diagnose this case.     

Pulmonary tuberculosis with cerebellar lesion in a patient after anti-TNF-alpha (infliximab) treatment for rheumatoid arthritis]

We report a case of pulmonary tuberculosis in a 51-year-old Japanese woman, who received treatment with infliximab for active rheumatoid arthritis. She had cough and sputum after the second infusion of the drug, small nodular lesions of right lung field and left lower lobe on her chest CT and a small nodular lesion of right cerebellar lobe on her cranial MRI were identified. Mycobacterium tuberculosis was cultured from her sputum. Therefore, we diagnosed her illness as pulmonary tuberculosis with a cerebellar lesion. The patient was treated with anti-tuberculosis drugs and showed marked improvement in lesions of the lung and brain. We considered this case a tuberculosis reactivation after infliximab treatment because of the short interval between the administration of infliximab and the occurrence of tuberculosis, and the complication of extrapulmonary lesion that suggested brain tuberculoma.     

Aortitis syndrome associated with ulcerative colitis, preceded by pulmonary infarction involvement]

A 21-year-old woman with a 6-year history of ulcerative colitis admitted to our hospital with chest pain, cough and fever of unknown origin in August 1998. On admission, laboratory data showed positive inflammatory signs. A chest radiograph and chest computed tomogram (CT) revealed nodular shadows in the right upper lung field. Fifty days after admission, hypertension developed and a bruit was audible in the neck and the upper abdomen. Digital subtraction angiography showed stenosis in carotid, renal and right upper pulmonary arteries. On the basis of these results, a diagnosis of aortitis syndrome was made. Moreover, these findings indicated pulmonary infarction in the right upper lobe due to aortitis syndrome. Aortitis syndrome preceded by pulmonary infarction involvement is very rare. Autoimmune disorders may have been involved in this case because of the association with ulcerative colitis.     

Pulmonary Langerhans' cell histiocytosis presenting with an endobronchial lesion

A 19-year-old man visited our hospital complaining of cough, sputum and low-grade fever. Chest radiograph and computed tomography findings suggested that he was suffering from pulmonary Langerhans' cell histiocytosis (PLCH). Bronchoscopy revealed a whitish elevated lesion at the bifurcation of the right upper lobe bronchus, and a specimen of this lesion showed the same pathological findings as pulmonary parenchymal lesions. Although there have been only a few reports of endobronchial LCH without pulmonary parenchymal lesions, this is, to our knowledge, the first case of PLCH with an endobronchial lesion, which was confirmed by bronchoscopy, and disappeared several months later.     

A case of primary pulmonary sporotrichosis

We report the first case of primary pulmonary sporotrichosis in Japan. A 53-year-old man was admitted to our hospital for further examination of the abnormal shadows on chest X-ray film. Six months before admission, he was admitted to another hospital because of alcoholic liver disease and diabetes mellitus. Since the initial chest film showed cavities with infiltration in the left upper lung field, he was treated with antituberculous drugs despite negative sputum cultures for mycobacterium. In spite of the medication, his chest X-ray film revealed another cavitary lesion, so he was referred to our hospital. He had been asymptomatic during this period. Chest X-ray on admission disclosed multiple cavities in the left upper lobe and a cavity in the right lower lobe. Repeated sputum specimens, bronchial washings and brushings for cytology and cultures were all negative. In an attempt to clarify the pathogen, percutaneous lung aspiration (PLA) was performed. The PLA sample yielded a positive culture of Sporothrix shenckii. After the diagnosis, S. schenckii was also cultured from sputa. A sporothrix skin test and yeast agglutination test for S. schenckii were positive. In the absence of a history for skin lesion, the patient was diagnosed as a primary pulmonary sporotrichosis. As iodide therapy was ineffective, he was started on a regimen of intravenous amphotericin B. However his renal function progressively deteriorated, so amphotericin B was discontinued. Now he receives miconazole intravenously and is still under careful observation. As far as we know, this is the first report of primary pulmonary sporotrichosis in Japan. The possibility of sporotrichosis should be considered in any cases of undiagnosed cavitary lung diseases.     

A case of lymphoma which developed bone, liver, and skin lesions two years after the diagnosis of lymphoid interstitial pneumonia

We reported a case of lymphoid interstitial pneumonia (LIP) in 1985. The patient, a 37-year-old housewife, had suffered from cough and dyspnea. Her chest roentgenograms revealed bilateral diffuse micronodular shadows. After open lung biopsy in 1983, the lesion was interpreted as LIP in the premalignant state. After pulse therapy her condition remarkably improved. She was readmitted because of fever and shoulder pain in 1985. X-ray films revealed punched out lesions on the extremities. As liver dysfunction and skin eruptions had been recognized to wax and wane since the first admission, transcutaneous liver biopsy and skin biopsy was done. The diagnosis was middle- to large-sized T cell lymphoma. CHOP therapy seemed effective. However there appeared dyspnea and cotton-like patchy shadows in both lung fields. Despite chemotherapy, she died of pulmonary fungal and cytomegaloviral infection in 1986. As a result of the reevaluation of the open lung biopsy specimen, we concluded that this case should have been considered as lymphoma at onset.     

A case of diffuse panbronchiolitis complicated by peripheral T cell lymphoma not otherwise specified

We report a case of diffuse panbronchiolitis (DPB) complicated by peripheral T cell lymphoma not otherwise specified. A 40-year-old Chinese man presented with intermittent fever, cough and significant white sputum production for more than 9 years, in addition to dyspnea and chest congestion that worsened after exercise. A chest CT scan indicated diffuse centrilobular fine nodular opacities with a 'tree-in-bud' appearance in both lungs. An open-lung biopsy was performed, and DPB was diagnosed by histopathological analysis. Three months later, the patient's pulmonary symptoms worsened. A chest CT of both lungs revealed multiple patchy opacities as well as enlargement of the hilar, mediastinal and multiple superficial lymph nodes. A whole-body bone scan revealed multiple osteolytic lesions located in the thoracic, lumbar and sacral spine. A biopsy of the right supraclavicular lymph node was performed, and peripheral T cell lymphoma not otherwise specified was diagnosed histopathologically. Cases of DPB complicated by non-Hodgkin's lymphoma are a rare occurrence. To our knowledge, there is only one earlier report of such a case in the literature (in Japanese). However, the prevalence of DPB complicated by T cell tumors is relatively high, indicating a possible association in pathogenesis of T cell disorders and DPB.     

A case of lung tuberculosis showing no chest radiograph findings with recurrent hemoptysis

A 59-year-old male was referred to our hospital because of hemoptysis. A chest X-ray film and 7 mm-slice CT scan showed no abnormal finding. Bronchoscopy revealed hemorrhage in the right upper bronchus (B1a) Bronchial lavage of the lesion was performed, but Mycobacterium tuberculosis was not detected. Because of repeated hemoptysis, he was admitted to our hospital. Right bronchial artery angiograph showed vascular hyperplasia in the peripheral part of the upper lobal branch, and this lesion was suggested to be a bleeding point. There were no vascular malformations. Thin slice (0.5 mm-thick) CT scan showed mild infiltrative shadow in the right upper lobe. After admission, sputa smear for mycobacteria and PCR for M. tuberculosis became positive, and he was diagnosed as pulmonary tuberculosis. After starting antituberculous chemotherapy, hemoptysis disappeared, and sputa smear and culture for mycobacteria converted to negative. This case suggests that lung tuberculosis should be suspected in patients having hemoptysis, even though they had no chest X-ray film abnormality.     

A case of secondary pulmonary cryptococcosis complicating diabetes mellitus]

In this case, a 65-year-old man complained of fever and productive cough while being treated for diabetes mellitus at the outpatient clinic. His chest CT scan revealed multiple infiltrative lesions in both the right and left lower pulmonary lobes. He was therefore given an antibiotic on suspicion of having bacterial pneumonia, and he also received nutritional instruction in relation to diabetes mellitus, and remission resulted. However, he could not maintain sufficient glycemic control thereafter, and his pulmonary lesions persisted. Because his lesions changed into cavitied multiple nodular lesions, as seen on a chest CT scan, a transbronchial lung biopsy was performed. Histopathological examination of the biopsy specimen demonstrated Cryptococcus organisms, and the Cryptococcus antigen titer was high, which led to a diagnosis of pulmonary cryptococcosis. After oral treatment with fluconazole for 1 year and 4 months, only a small nodule in the right lower lobe and a funicular lesion in the left lower lobe remained on a chest CT scan, and the patient had neither subjective symptoms nor evidence of inflammation, although he still had a positive antigen titer for Cryptococcus. Thus, the treatment was terminated. Improvement of the clinical symptoms and of the laboratory and radiological findings demonstrated the therapeutic efficacy of this treatment.     

Rapid remission of pulmonary eosinophilic granuloma in a young male patient after cessation of smoking]

A 24-year-old asymptomatic man was referred to our hospital in October 7, 1997 for further examination of abnormal shadows detected on chest X-ray films that had been obtained during an annual health examination on September 12, 1997. The patient had suffered productive cough since the end of August 1997. A chest X-ray film obtained one year earlier showed no abnormalities. The patient had smoked 20 cigarettes per day since the age of 14. The findings of a physical examination and laboratory tests were unremarkable. A chest X-ray films showed micronodules 2-5 mm in size that were predominantly distributed in the upper and middle fields of both lungs. A chest computed tomogram (CT) showed multiple cysts measuring about 10 mm in diameter, with thick walls (2 to 3 mm) in addition to small nodular shadows. A transbronchial lung biopsy and a thoracoscopic lung biopsy revealed several stellate nodular fibrotic lesions containing some S-100 protein-positive, large mononuclear cells and lymphoid cells. An electron microscopic study found several Langerhans' cells with Birbeck's granules in their cytoplasm. A chest CT scan obtained 2 months after the patient stopped smoking (the day of admission) showed marked regression of the cystic shadows. Evidence suggests the pathogenesis of the disease is closely linked to smoking, and some case reports have documented clinical and radiographic improvements after patients stop smoking. Pulmonary small nodular lesions and cystic lesions with thick walls tend to undergo regression. Although surgical lung biopsy specimens disclosed several stellate nodular fibrotic lesions of eosinophilic granuloma that seemed to be histologically irreversible, our patient experienced remarkable regression of his pulmonary lesions within 2 months after the cessation of smoking.     

A case of Wegener's granulomatosis with a giant solitary pulmonary mass]

A 44-year-old woman was admitted to hospital with a history of high fever, cough, and weight loss. Her chest X-ray film showed a giant solitary pulmonary mass in the left lower lobe. After admission, she developed bilateral eyeache and left exophthalmos. CT film of the head showed a massive lesion in the sinuses. Biopsy specimens obtained from both massive lesions showed either epithelioid granulomas with Langhans' giant cells or necrotising vasculitis. Antineutrophil cytoplasmic antibodies, which have high sensitivity and specificity for active WG, were found. The diagnosis of WG was thus made.     

Mycobacterium abscessus infection complicated with diabetes mellitus]

We report a case of Mycobacterium abscessus infection complicated with diabetes mellitus. A 38-year-old man with diabetes mellitus as an underlying disease, was admitted to our hospital because of a productive cough. He had had pulmonary tuberculosis two years before. Chest radiography revealed infiltration in both lung apices and chest CT showed a cavitary lesion in the left upper lobe. Gaffky 2 was found on a sputum smear. However, in the examination of PCR on sputum, not only M. tuberculosis but M. avium complex was negative, and repeated cultures of sputum were positive for M. abscessus. On the basis of the diagnosis of an M. abscessus infection, the patient was initially treated with amikacin, imipenem/cilastatin and levofloxacin during hospitalization while receiving insulin for diabetes mellitus. The smear and culture of sputum became negative for Mycobacterium, and the findings of chest radiography and chest CT improved. After discharge, treatment was continued with clarithromycin and levofloxacin. It is considered that the choice of effective drugs and the additional treatment of an underlying disease are very important for the treatment of a Mycobacterium abscessus infection.     

Pulmonary tuberculosis case with consistant findings of endobronchial spread in chest roentogenography for about three years: a case report

We reported a case of pulmonary infection by Mycobacterium tuberculosis complicated by endobronchial spread. Chest roentogenography and CT for an 85-year-old male complaining of cough showed endobronchial spread in right upper lung field. His sputum culture for eight weeks showed 10-20 colonies of Mycobacterium tuberculosis. Transbronchial lung biopsy revealed granulomas with caseous necrosis. Findings in chest XP and CT after the therapy with INH, RFP and EB for six months showed much improvement.