A case report of rheumatoid arthritis complicated with rapidly progressive interstitial pneumonia, multiple bullae and pneumomediastinum, which was successfully treated with tacrolimus.

A 64-year-old woman had been treated with prednisolone (PSL) for interstitial pneumonia (IP) of unknown origin since 1988. The IP progressed gradually, however, and home oxygen therapy was instituted in 1993. In 2002, persistent arthritis of the hands appeared and diagnosis of rheumatoid arthritis (RA) was finally established based on radiological and pathological findings. Salazosulfapyridine was given with only partial effect. On October 2002, she was hospitalized because of back pain followed by dyspnea. Chest X-ray revealed multiple giant bullae on bilateral upper lung fields, accompanied by deterioration of IP. Methyl-prednisolone pulse therapy followed by 30 mg/day of PSL was instituted and the bullae were diminished with gradual improvement of IP and synovitis. On the 55th hospital day, she complained of chest oppression, and chest X-ray revealed a complication of pneumomediastinum. Since IP was still active and serum KL-6 remained high, 3 mg/day of tacrolimus was added to control IP further and to reduce the dosage of PSL which was recognized as one of the aggravation factors of pneumomediastinum. As a result, pneumomediastinum disappeared gradually along with amelioration of IP. PSL was successfully tapered to 15 mg/day by the 87th hospital day and the patient was discharged. Although the efficacy of tacrolimus on IP complicated with polymyositis / dermatomyositis and other autoimmune diseases has been reported, this case first suggests its efficacy on IP associated with RA.     

Case of minocycline-induced pneumonitis with bilateral pleural effusion]

A 51-year-old man was admitted to our hospital with fever, dry cough and dyspnea. He had taken minocycline for 11 days because of urinary tract infection. Chest X-ray on admission showed diffuse reticular shadows in bilateral lung fields with bilateral pleural effusion. Cessation of minocycline led to spontaneous improvement of symptoms and radiographic findings. The lymphocyte stimulation test for minocycline with peripheral blood and pleural effusion were negative. After provocation test with minocycline, he developed fever and dry cough and bilateral ground glass opacity appeared on his chest X-ray. He was diagnosed as minocycline-induced pneumonitis and recovered rapidly following corticosteroid therapy.     

Case of hemophagocytic syndrome associated with active dermatomyositis]

We report a case of dermatomyositis (DM) with hemophagocytic syndrome (HPS). The patient is a 60 year old male admitted to our hospital with muscle weakness, high fever, weight loss and pancytopenia. On physical examination, proximal muscle weakness and skin rash on the back were noted. Laboratory data revealed elevated serum levels of muscle enzymes, lactate dehydrogenase and ferritin. Serum levels of M-CSF, TNF-alpha, soluble IL 2 receptor were remarkably increased. Bone marrow aspiration showed histiocytosis of 7 to 10% with prominent hemophagocytosis. Gastroendoscopic examination revealed II-a gastric cancer. He was treated with methylprednisolone (m-PSL) pulse theraphy (1 g/ day x 3 days) followed by 60 mg/day oral prednisolone. He quickly responded to the treatment and laboratory data returned to normal in 20 days despite the remaining gastric cancer which was removed successfully 3 months later. HPS is a rare complication of DM and only three cases have been documented so far in the literature. Augmented cytokine in his serum is considered to be closely related to HPS in this case.     

Hypogammaglobulinemia in steroid-dependent asthmatics correlates with the daily dose of oral prednisolone

BACKGROUND: Steroid-induced adverse effects including suppression of humoral immunity should be considered in steroid-dependent severe asthma. Only a few studies have determined the exact steroid dose that could potentially suppress humoral immunity in asthmatics. METHODS: Randomly selected 100 adult asthmatics treated with inhaled beclomethasone dipropionate (BDP) were classified into three groups based on the dose of steroid to determine the serum IgG, IgA and IgM levels by radioimmunoassay. Relationships between serum immunoglobulin levels and the daily dose and duration of oral prednisolone (PSL) therapy were examined. RESULTS: None of the patients on inhaled corticosteroid alone had hypogammaglobulinemia. Patients on oral PSL at a dose >12.5 mg/day for at least 1 year had low serum IgG. There was no significant correlation between the duration of oral PSL therapy and serum IgG. CONCLUSIONS: Oral PSL can potentially suppress humoral immunity in severe asthma. In asthmatics, hypogammaglobulinemia could develop in those on a daily dose of PSL >12.5 mg, but is independent of the duration of such treatment. No suppression of humoral immunity was noted on inhaled corticosteroid therapy alone, either at low or high dose.     

Undiagnosed pleural effusion treated with traditional Chinese medicine: A case report

The main clinical manifestations of pleural effusion are exertional dyspnea, predominantly dry cough, and pleuritic chest pain. To treat pleural effusion appropriately, it is important to determine its etiology; which however, remains unclear in nearly 20% of cases.A 73-year-old man with a history of invasive pulmonary tuberculosis (TB), had been experiencing chest congestion and dyspnea with undiagnosed pleural effusion for six years. After a series of clinical examination and laboratory tests, there was still no clear diagnosis. Despite administering diuretics and intermittent draining, the patient's condition aggravated progressively. He sought further treatment at Dongzhimen Hospital Respiratory Outpatient Clinic. The patient was treated with Zanthoxylum and Trichosanthes Decoction (Jiao Mu Gua Lou Tang). After one and a half years, his symptoms greatly improved and ultrasound revealed that the pleural effusion had apparently absorbed.It is suggested that TCM herbal formulas can play a critical role in preventing the progression of complicated, undiagnosed pleural effusion, especially in cases of poor response to conventional therapy and thoracentesis. Additional studies on the functions and mechanisms of the medicinals are warranted.     

Hemothorax with high number of eosinophils following warfarin overdose

Some drugs are known to induce pleural effusion. Drug-induced pleural effusion is often associated with pleural fluid eosinophilia. Anticoagulant therapy may induce pleural effusion by at least two different mechanisms: bleeding complication (haemothorax) and allergic or toxic reaction. Authors describe 76-yr-old male with warfarin-induced pleural effusion. Since INR was 15.5, and the value of pleural effusion Hct exceeded significantly 50% of Hct value in blood, spontaneous haemothorax due to warfarin overdose was diagnosed. Pleural fluid analysis revealed relatively high percentage of eosinophils (13%), but it was probably secondary to the presence of numerous red blood cells in the effusion. The authors discuss different mechanisms of drug-induced pleural effusion, with special attention to eosinophilic pleural effusion and review the literature on the spontaneous haemothorax as a complication of anticoagulant therapy.     

Effects of a vitamin D analog, alfacalcidol, on bone and skeletal muscle in glucocorticoid-treated rats

Glucocorticoids cause secondary osteoporosis and myopathy. The efficacy of vitamin D on osteoporotic fractures is thought to be through direct effects on bone and indirect effects on muscles that help to prevent falls. However, effects of vitamin D on muscles under glucocorticoid treatment remain unclear. Six-month-old female Wistar rats were randomized to four groups: vehicle-treated controls; a prednisolone (PSL)-administered group (PSL group); an alfacalcidol-administered group (D group); and a group administered both PSL and alfacalcidol (PSL+D group). After a 4-week treatment period, maximum contractile strength and strength decrement index (SDI), an indicator of muscle fatigue, were measured in the calf muscle by electrical stimulation of the sciatic nerve. Cross-sectional area (CSA) of muscle fibers in the tibialis anterior muscle and bone mineral density (BMD) of the femur were evaluated. The PSL group showed significantly lower muscle strength, BMD and CSA of muscle fibers, and significantly higher SDI compared to the other three groups (P < 0.05). No significant differences were observed in any of these parameters among control, D, and PSL+D groups. These results suggest that in glucocorticoid-treated rats, alfacalcidol preserved not only BMD, but also muscle strength and muscle volume, and prevented muscle fatigue.     

Myelomatous effusion with poor response to chemotherapy

While pleural effusion in multiple myeloma is relatively infrequent, myelomatous pleural effusion is extremely rare. We experienced a 61-year-old woman with IgD-lambda multiple myeloma and pleural effusion. The diagnosis was made originally by pleural biopsy, pleural fluid cytology and immunoelectropheresis of pleural fluid. Transient improvement of the pleural effusion was observed after administration of combination chemotherapy of vincristine, melphalan, cyclophosphamide, prednisone (VMCP)/vincristine, cyclophosphamide, adriamycin, prednisone (VCAP). Two months later, myelomatous pleural effusion recurred and no response to salvage therapy was observed. We reviewed the clinical feature of this case and literature concerning myelomatous pleural effusion.     

Unusual clear cell variant of epithelioid mesothelioma.

Clear cell mesothelioma is an extremely rare neoplasm of the pleura, which can easily be mistaken for a metastasis of clear cell carcinoma to the pleura. We report here the histochemical, immunohistochemical, and ultrastructural aspects of a new case of clear cell pleural mesothelioma in a 52-year-old man with no known asbestos exposure. He was admitted to the hospital for recurrent pleural effusion, which was negative for neoplastic cells at the cytologic examination. A partial decortication of the right pleura was performed. The morphologic, immunohistochemical, and ultrastructural features reported for this case are consistent with the diagnosis of clear cell mesothelioma. The differential diagnosis and immunohistochemical features in comparison with other clear cell neoplasms are discussed.     

Rhabdomyosarcoma in a child with massive pleural effusion: cytological diagnosis from pleural fluid.

We report on a case of embryonal rhabdomyosarcoma in an 11-yr-old boy investigated for mild chest pain after trauma. Chest radiography showed a massive right pleural effusion. Cytological analysis of the pleural fluid demonstrated the presence of malignant small undifferentiated cells. The rhabdomyoblastic nature of the cells was confirmed by positive immunostains of HHf35 actin, desmin, and skeletal muscle myosin; histological examination of a core biopsy confirmed the diagnosis of rhabdomyosarcoma. Computed tomography and gallium scan revealed the presence of an extensive anterior and lower chest wall mass involving the mediastinum, as well as retroperitoneal lymphadenopathy. Massive pleural effusion is a frequent presentation in malignant disease, but is rare in rhabdomyosarcoma. The immunochemical stain studies performed on cytological smears in this case proved to be very useful for making the definitive and accurate diagnosis. Diagn. Cytopathol. 1999;21:125-128.     

Effectiveness of prednisolone/aspirin therapy for recurrent aborters with antiphospholipid antibody.

Seventeen women with recurrent fetal loss associated with anti-phospholipid antibody were treated by prednisolone and low-dose aspirin (PSL/ASP) therapy from the early gestational period. The success rate of pregnancy in the treated patients was significantly higher than that in 12 untreated patients of similar background, including age, number of previous fetal losses and anti-cardiolipin titre (76.5 versus 8.3%, P less than 0.01). The degree of fetal growth retardation evident in previous pregnancies was also improved by the therapy, suggesting that PSL/ASP itself has a beneficial effect against placental damage. These clinical improvements were accompanied by a reduction in anti-phospholipid antibody titre, especially that of anti-phosphatidylserine antibody (anti-PS), to within the normal range within 8 weeks after PSL/ASP administration in most of the treated patients. There was no reduction of antibody titre in the untreated patients during pregnancy. It was concluded that PSL/ASP therapy, when started in the early gestational period (prior to 8 weeks gestation), was effective for the achievement of successful pregnancy and the prevention of fetal growth retardation and that the anti-PS titre was a good clinical marker for evaluating the effect of PSL/ASP therapy.     

A case of suspected allergic granulomatosis and angiitis with a rapid clinical course of paraplegia

A 68-year-old man with suspected allergic granulomatosis and angiitis is reported. He had received 10 mg of prednisolone daily since July 1988 for asthma. He abruptly developed muscle weakness of the lower extremities, followed two days later by paraplegia. Six days after the onset of the muscle weakness, he was hospitalized. He showed disturbance of recent memory, disorientation, neck rigidity, paraplegia, mild muscle fasciculation and hypesthesia. He also showed paralytic ileus. Laboratory findings showed leukocytosis (24580/mm3), eosinophilia (56% of the peripheral white blood cells and 19% of the cells in the cerebrospinal fluid), on erythrocyte sedimentation rate of 31 mm/h, and the IgE level of 1200 IU/ml. The ECG showed loss of the r-wave in V1 and V2. A granulomatous lesion anterior to the spinal cord was found on myelography and MRI. Prednisolone was given at a dose of 60 mg daily resulting in improvement of the clinical symptoms and eosinophilia. There was disappearance of the granuloma on MRI performed after prednisolone therapy. Despite the severe manifestation of allergic granulomatosis and angiitis, prednisolone therapy had a marked effect in this patient. The granulomatous lesion anterior to the spinal cord shown by MRI suggested an eosinophilic granuloma, and may have been the etiology of some of the neurological symptoms.     

Amebic lung abscess with coexisting lung adenocarcinoma: a unusual case of amebiasis

Amebic lung abscess with concurrent lung cancer, but without either a liver abscess or amebic colitis, is extremely uncommon. Here, we report a 70-year-old man presenting with pulmonary amebiasis and coexisting lung adenocarcinoma. During his first-time hospitalization, the diagnosis of lung amebiasis was confirmed by morphological observation and PCR in formalin-fixed and paraffin-embedded sediments of pleural effusion. Almost four months later, the patient was readmitted to hospital for similar complaints. On readmission, lung adenocarcinoma was diagnosed by liquid-based sputum cytology and thought to be delayed because coexisting amebic lung abscess. This case demonstrated that sediments of pleural effusion may be used for further pathological examination after routine cytology has shown negative results. At the same time, we concluded that lung cancer may easily go undetected in the patients with pulmonary amebiasis and repetitive evaluation by cytology and imaging follow-up are useful to find potential cancer.     

Primary malignant lymphoma of the central nervous system presenting with ascites and pleural effusion

A 70-year-old woman was admitted to our hospital owing to ascites and pleural effusion. Though malignant cells (B-cell type lymphoma) were detected in both the ascites and pleural effusion, neither lymph node swelling nor a tumor was detected upon chest, abdominal and pelvic computed tomography (CT). After weekly THP-COP therapy for 8 weeks, the ascites and pleural effusion completely disappeared. Two years after the first admission, she was re-admitted because of a disturbance of consciousness, and a brain tumor was detected on CT scan. The immunohistological and genetic data for the brain tumor were identical to those of the malignant cells in the pleural effusion and ascites detected 2 years previously. Whereas the symptoms at onset of a primary lymphoma of the central nervous system (CNS) are usually neurological ones, in this rare case of primary CNS lymphoma, the symptoms at onset were the ascites and pleural effusion without neurological symptoms.     

胸膜腔穿刺抽液并注入尿激酶治疗结核性胸腔积液

目的探讨穿刺抽液并注入尿激酶治疗结核性胸腔积液的临床价值。方法对30例结核性胸腔积液(多房性14例,包裹性6例,脓胸10例)患者,经穿刺抽液后并注入生理盐水50ml稀释的尿激酶12.5万IU,24h后抽尽液体,如仍有分隔或积液重复上述治疗。结果30例患者共穿刺了85次,成功率92.5%,3例并发气胸,1例并发血气胸。24例患者仅经1次尿激酶治疗后有效,总有效率96%。1例无效。注入尿激酶之后第1次抽液量较用药前明显增多(P<0.05)。而第2~4次用药后抽液量增多不明显(P>0.05)。结论穿刺抽液并注入尿激酶是治疗结核性胸腔积液的一种安全、有效的方法,使绝大多数患者避免了外科手术。     

内科胸腔镜在提高胸腔积液诊断率中的价值

目的评价内科胸腔镜在提高胸腔积液病因诊断中的价值及内科胸腔镜的检查时机。方法连续收集渗出性胸腔积液患者311例,其中男性194例,女性117例;年龄17～90岁,平均年龄58.3岁。评估胸腔积液细胞学对恶性胸水的诊断率。对27例经胸水细胞学未能诊断的患者和2例胸水细胞学恶性胸腔积液但未准确分型的患者进行了内科胸腔镜检查,评估胸腔镜检查联合胸水细胞学检查对胸腔积液诊断率及恶性胸水的准确诊断率的影响。结合不同次数的胸腔积液细胞学的阳性率及相关支出,评估内科胸腔镜的检查时机。结果311例渗出性胸腔积液患者经胸水细胞学诊断恶性胸腔积液的患者为80例;231例胸水细胞学未确诊的患者结合各自情况,经过内科胸腔镜检查和/或支气管镜、或肺动脉CT造影等检查,并结合临床资料系统分析后,诊断如下:恶性胸腔积液106例,良性胸腔积液91例,未确诊病例34例。胸水细胞学检查的确诊率为28.9%(80/277)。29例接受了内科胸腔镜检查患者中,25例患者直接确诊,诊断率为86.2%。内科胸腔镜检查使该组病例的确诊率提高到37.9%(105/277),与单纯胸腔积液细胞学检查结果比较差异有统计学意义(P=0.024)。3次及以上的胸腔积液细胞学检查不能较2次胸腔积液细胞学检查提高更多的诊断率(P=0.156),且其支出超过内科胸腔镜的支出。结论内科胸腔镜不仅较胸腔积液细胞学检查能提高恶性胸腔积液的诊断率,更能提高胸腔积液总体诊断率。对2次胸腔积液细胞学检查无结果者,如无禁忌证,应积极进行内科胸腔镜检查。     

HER2/HER3‐positive metastatic salivary duct carcinoma in the pleural effusion: A case report

Salivary duct carcinoma (SDC) is an aggressive form of salivary gland tumor, and SDC patients tend to be older men, more commonly in advanced stage with a poorer prognosis. Although the cytological characteristics of SDC on fine‐needle aspiration cytology have been well‐described at the primary site, they have not been explored in metastasis. Here we reported a case of HER2/HER3‐positive metastatic SDC in the lung and pleural effusion. The patient was a man in his 50s who had undergone extended total parotidectomy in 2008. He was originally diagnosed as having HER2‐positive left parotid SDC. Six years later a mass was discovered in the left lung by chest computed tomography (CT) and was diagnosed as metastatic SDC by both bronchial biopsy and cytology. Subsequently he had a recurrent SDC in the left pleural effusion and died of respiratory failure. Cytological findings from bronchial brushing smear showed small sheet clusters in a slightly necrotic background. In the pleural effusion cytology, tumor cells appeared as ball‐like clusters of epithelioid cells with apocrine‐like findings. In immunocytochemistry, HER3 of SDC cells in pleural effusion was significantly overexpressed relative to the matched primary tumor, even though HER2 amplification did not change. Cytological findings and HER family receptors differed between the primary and metastatic SDC. Therefore, molecular tests, such as protein expression and gene amplification using cytological specimens, may become important in future when determining therapy strategies in patients with distant metastasis.     

Role of the Neutrophil-Lymphocyte Ratio in the Differential Diagnosis of Exudative Pleural Effusion

OBJECTIVES:Pleural effusion is a common diagnostic and clinical problem. The differential diagnosis of pleural effusion may be difficult and may require several procedures, including invasive ones. Certain studies have investigated biochemical parameters to facilitate the diagnosis of exudative pleural effusion; however, it remains a challenging problem in clinical practice. We aimed to investigate the potential role of the neutrophil-lymphocyte ratio, which can be easily obtained by determining the cell count of the pleural fluid, in the differential diagnosis of exudative pleural effusion.METHODS:Records from patients who underwent thoracentesis and pleural fluid analysis between May 1, 2013, and March 1, 2015, were obtained from the electronic database of our hospital. The patients who met the inclusion criteria were divided into five groups according to their diagnosis: malignant pleural effusion, para-malignant pleural effusion, para-pneumonic effusion, tuberculosis-related effusion or other. The neutrophil-lymphocyte ratio value was calculated by dividing the absolute neutrophil count by the absolute lymphocyte count. The patient groups were compared according to the given parameter.RESULTS:A total of 465 patients who met the inclusion criteria among 1616 patients with exudative pleural effusion were included in the study. The mean neutrophil-lymphocyte ratio value was significantly lower in tuberculosis-related pleural effusion compared to malignant, para-pneumonic and para-malignant effusions (p=0.001, p=0.001, p=0.012, respectively). The areas under the curve for tuberculosis pleurisy compared to malignant, para-pneumonic and para-malignant effusions were 0.38, 0.36, and 0.37, respectively. Lower cut-off values had higher sensitivity but lower specificity for tuberculosis pleurisy, while higher cut-off values had higher specificity but lower sensitivity for this condition.CONCLUSION:The pleural fluid neutrophil-lymphocyte ratio, which is an inexpensive, reproducible, and easily calculated hematological parameter, may facilitate the differential diagnosis of pleural effusion.     

A case of rheumatic fever complicating carditis detected by a newly-developed systolic murmur in an adult woman

A 62-year-old woman presented to a primary care doctor on January 2006 due to a sore throat and high fever, and had received medication for a common cold. She was referred to our hospital in February 2006 because of additional manifestations such as painful rashes on the lower limb similar to erythema nodosum and polyarthralgia on her feet, shoulder and finger joints. She was initially treated with an anti-inflammatory drug (NSAID) for polyarthritis but the symptoms did not improved. In addition, the serum level of anti-streptolysin O antibody (ASO) was elevated at the second visit more than that at the first visit. She was diagnosed to have rheumatic fever (RF) based on the polyarthritis, inflammatory data and an increase of the ASO level. She was treated with 10 mg a day of prednisolone (PSL) and sultamicillin tosilate. However, a systolic murmur that had been never noticed by previous auscultation appeared after the third hospital day and the mitral regurgitation was also detected on echocardiogram. She was then treated with 40 mg a day of PSL because of an appearance of the carditis due to RF. The increased PSL dose promptly improved the systolic murmur as well as the arthritis. This report presented an RF case with carditis detected by an development of the systolic murmur in an adult female.     

A case of amyopathic dermatomyositis with acute interstitial pneumonia (DAD pattern)]

A 61-year-old man was admitted to our hospital because of edematous erythema on his upper eyelids and dry cough. No subjective nor objective findings suggestive of skeletal muscle involvement, such as muscle weakness and elevated levels of aldolase and creatine phosphokinase were noted. Chest high-resolution computed tomography revealed a ground glass opacity and consolidation of his lower lung. Skin biopsy findings were compatible with dermatomyositis. Therefore, he was diagnosed as amyopathic dermatomyositis (ADM) with acute interstitial pneumonia and treatment with steroid pulse therapy was started. Since histological evaluation showed diffuse alveolar damage during the initial treatment, the treatment was changed into the combination therapy of prednisolone and cyclosporine. However, his acute interstitial pneumonia did not respond to this treatment and passed away by aggravation of a breathing state and concurrence of disseminated intravascular coagulation. Japanese patients with ADM have been shown to be more frequently associated with intractable acute interstitial pneumonia than Caucasian patients, suggesting that the racial difference influences the occurrence of acute interstitial pneumonia in ADM. Since autoantibodies specific for ADM have not been detected, we performed immunoprecipitation analysis using 35S methionine-labeled K562 cells to identify them. His sera immunoprecipitated a polypeptide of 140 kDa. The 140 kDa polypeptide might be one of autoantibodies specific for ADM with acute interstitial pneumonia, although future analysis using a larger number of patients with ADM will be required to confirm this result.