Intracavitary mass as the initial manifestation of primary pericardial mesothelioma: a case report

A 31-year-old woman presented with a 3-month history of progressing fatigue and effort dyspnea. Echocardiography depicted a tumor within the free wall of the right ventricle and right atrium, located on both sides of the tricuspid annulus. Computed tomography showed disseminated circular shadows sized up to 7 mm--most likely metastatic lesions--in both lungs. The diagnosis of low-grade mesothelioma bifasicum was confirmed with histopathologic and immunohistochemical studies of the samples taken by thoracoscopy from parietal pleura, lung tumor, and diaphragm region. Chemotherapy, which included gemcitabine and carboplatin, resulted in transient improvement of the clinical status of the patient and reduction of the tumor mass lasting several months followed by progression of the disease. Significant amounts of pleural fluid and huge tumors within both pleural cavities emerged. The patient died due to respiratory and circulatory insufficiency 11 months following the diagnosis.     

Oral inflammatory changes as an initial manifestation of Crohn's disease antedating abdominal diagnosis

The case of a 14-year-old boy who had oral ulcers with histologic proof of granulomatous disease nine months before the diagnosis of intestinal Crohn's disease is presented. Additional extraintestinal manifestations of this case were cheilitis, anal fissures, and “metastatic” disease to the umbilicus. The diagnosis was established after the onset of abdominal symptoms. All manifestations responded rapidly to systemic prednisone, sulfasalazine, and metronidazole.     

Septic arthritis as an initial manifestation of endocarditis

A 60-year-old female patient was admitted to the hospital with high fever and an acute onset of pauciarticular arthritis. Clinical examination and ultrasound imaging of the right knee demonstrated a severe synovitis with a large synovial effusion and a Baker's cyst. Arthrocentesis showed an opaque viscous synovial fluid with a highly elevated white blood cell count. In synovial fluid cultures and in serial blood cultures Aerococcus viridans (Av) was detected. Further examination revealed a mitral valve endocarditis as the origin of the septicemia. This case report is the first one to describe the association of an endocarditis and a septic arthritis due to Av.     

Pancreatic mass as an initial manifestation of polyarteritis nodosa:A case report and review of the literature

Classic polyarteritis nodosa(PAN) that targets mediumsized muscular arteries and microscopic polyangiitis(MPA),characterized by inflammation of small-caliber vessels and the presence of circulating myeloperoxidase anti-neutrophil cytoplasmic antibodies(MPO-ANCA),are distinct clinicopathological entities of systemic vasculitis.A 66-year-old woman presented with fever,cholestasis and positive MPO-ANCA.Radiological examination showed a pancreatic mass compressing the bile duct.Therefore,we performed pancreatoduodenectomy.Histopathological examination revealed that necrotizing vasculitis predominantly affecting the medium-sized vessels,spared arterioles or capillaries in the pancreas,a finding consistent with PAN.Unexpectedly,renal biopsy revealed small-caliber vasculitis and glomerulonephritis,supporting MPA.The initial manifestation of a pancreatic mass associated with vasculitis has only been reported in 7 articles.Its diagnosis is challenging because no reliable clinico-radiological findings have been observed.Clinicians should be aware of such cases and early diagnosis followed by immunosuppression is mandatory.Our findings may reflect a polyangiitis overlap syndrome coexisting between pancreatic PAN and renal MPA.     

Eosinophilic pneumonia as an initial manifestation of rheumatoid arthritis

Rheumatoid arthritis is a systemic disease that may have pulmonary manifestations. We describe a case of eosinophilic pneumonia as the primary presentation of rheumatoid arthritis. While several cases of acute and chronic eosinophilic pneumonia have been reported in patients with preexisting rheumatoid arthritis, this is the first case reported in which the eosinophilic lung disease was the initial manifestation of systemic rheumatoid arthritis.     

Sensorimotor polyneuropathy as an initial clinical manifestation of sarcoidosis

A 45 year-old Japanese woman developed numbness and tingling of both hands and feet. Electrophysiological examination revealed sensorimotor polyneuropathy. She was diagnosed as suffering from sarcoidosis on the basis of the pathological findings from dermal biopsy. Steroid therapy effectively improved the clinical symptoms. Although sarcoid neuropathy is rare, this case suggests sensorimotor polyneuropathy is an important symptom of sarcoidosis and can represent the initial clinical manifestation of the disease.     

Sensorimotor polyneuropathy as an initial clinical manifestation of sarcoidosis

Abstract

A 45 year-old Japanese woman developed numbness and tingling of both hands and feet. Electrophysiological examination revealed sensorimotor polyneuropathy. She was diagnosed as suffering from sarcoidosis on the basis of the pathological findings from dermal biopsy. Steroid therapy effectively improved the clinical symptoms. Although sarcoid neuropathy is rare, this case suggests sensorimotor polyneuropathy is an important symptom of sarcoidosis and can represent the initial clinical manifestation of the disease.     

A case of anti-phospholipid antibody syndrome presenting chorea as an initial manifestation

A 20 year old female patient with anti-phospholipid antibody syndrome who presented chorea as an initial symptom is described. At the age of 14, she noticed involuntary movements of upper and lower extremities bilaterally. The CT scan revealed the presence of low density areas in the right caudate nucleus and left putamen. The result of the laboratory tests included antibodies to nuclear antigens (positive FANA and anti-DNA), prolonged PT and APTT, biological false positive for syphilis. Her serum contained antibodies to cardiolipin and phosphatidylserine as demonstrated by specific ELISA assay method. The clinical diagnosis of chorea was made and haloperidol was administered with partial symptomatic improvements. The patient reported here is the first well documented case of anti-phospholipid antibody syndrome presenting chorea as an initial manifestation in the Japanese literature.     

Hemophagocytic lymphohistiocytosis as an initial manifestation in an HIV-infected patient

A 48-year old man was admitted with idiopathic fever, and subsequently diagnosed as having hemophagocytic lymphohistiocytosis (HLH). Though an extensive series of laboratory examinations failed to demonstrate an apparent etiology, empirical use of steroids achieved remission. About two years later, the patient developed Pneumocystis Jiroveci pneumonia and was diagnosed as HIV-positive. Based on this case, HIV-screening tests would be performed whenever we encounter HLH in Japan, where the number of HIV-positive patients is increasing.     

Chylothorax as the initial manifestation of malignant pleural mesothelioma--a case report]

A case of chylothorax associated with diffuse malignant pleural mesothelioma in a 53-year-old woman was reported. Chest radiography in a mass examination revealed right pleural effusion. A thoracentesis yielded fluid with characteristics consistent with chyle. In spite of our suggestion that thoracoscopy for further examination was necessary, the patient and her husband had refused the operation for months. Chest CT scanning revealed a nodule behind the xiphoid process. Lymphoscintigraphy suggested an obstruction of the right parasternal lymphatic vessel. Repeated thoracentesis did not yield a diagnosis. As bilateral pleural effusion and chylous ascites appeared, dyspnea worsened. About 2 years after the mass examination thoracoscopy was performed. On thoracoscopic exploration, a nodule was found in the anterior mediastinum and a biopsy was performed. Histological analysis revealed that the patient had the epithelial subtype of malignant pleural mesothelioma. Thoracoscopic pleural biopsy is a useful means of establishing the nature of a chylous effusion. In adult patients with chylothorax, thoracoscopic exploration should be performed as soon as possible to rule out malignancy.     

Subcutaneous emphysema as the initial manifestation of perforation of a hollow abdominal viscus

The authors reports a case of cervical and abdominal subcutaneous emphysema, associated to pneumoretroperitoneum and pneumomediastinum, with no free air in the abdominal cavity, secondary to perforated diverticulitis of the sigmoid colon. We comment the rarity of this clinical presentation, the physiopathologic mechanisms of subcutaneous air presence in cases of non instrumental perforation of abdominal viscera and the ominous significance of the subcutaneous emphysema in such perforations.     

Paralytic ileus as an initial manifestation of malignant VIPoma of the pancreas--case report with review of the literature

A 57-year old patient with a paralytic ileus of unknown origin was admitted to the intensive care unit. Because of the laboratory findings with therapy resistant hypokalemia, hypercalcemia and metabolic acidosis a VIPoma was suspected. Therapy with somatostatin resulted in correction of laboratory abnormalities and in normalization of gastrointestinal motility. Plasma concentrations of VIP and PP were elevated, ultrasonography revealed a pancreatic tumor. Postsurgical examination of the removal tumor tissue confirmed the diagnosis of a malignant VIPoma. Clinical symptoms, laboratory findings with and without somatostatin-therapy and immunhistochemical properties are described.     

Gangrene as an initial manifestation of a catastrophic antiphospholipid syndrome

Catastrophic antiphospholipid syndrome (CAPS) is an unusual form of presentation of antiphospholipid syndrome with a poor prognosis, so early diagnosis and treatment are necessary. We report a patient who had gangrene as the initial manifestation of CAPS.     

Pneumothorax as an initial manifestation of obstructive sleep apnea syndrome

Case report  

A sixty-five-year-old man with bullous lung disease was admitted to emergency service with chest pain and dyspnea that developed during sleep. Pneumothorax was diagnosed both clinically and radiologically. After the chest drainage, the patient presented with a prolonged air leak that required thoracotomy. Further history and occurrence of pneumothorax during sleep suggested that obstructive sleep apnea might play a role in the development of pneumothorax. Nocturnal polysomnography later confirmed the diagnosis of severe obstructive sleep apnea syndrome.