Xanthogranulomatous cholecystitis expressing high levels of DUPAN-II

We report a case of xanthogranulomatous cholecystitis (XGC) showing high levels of serum DUPAN-II in a 65-year-old woman. Preoperative radiologic examination showed no abnormal findings except in the gallbladder. Endoscopic ultrasonography was effective for differentiating chronic cholecystitis from gallbladder cancer before the operation. Cholecystectomy was performed by laparotomy, and the diagnosis of XGC was confirmed intraoperatively by examining a frozen section. Histologically, no cancer lesion was observed in the gallbladder, while immunochemical reactivity to DUPAN-II was demonstrated in the brush-border area of the epithelium and in histiocytes in the gallbladder. The half-life of serum DUPAN-II in our patient after cholecystectomy was approximately 1 month, and finally dropped to within the normal range after cholecystectomy.     

A case of tuberculous aneurysm of subclavian artery occurred in the course of treatment for miliary tuberculosis

This case is a 56-year old woman. Steroids were being administered perorally after a thymectomy for myasthenia gravis. A fever of 38-39 degrees Celsius appeared during night, an abnormal shadow showed up on a chest X-ray and the patient was hospitalized. Gaffky No. 2 acid-fast bacilli were detected in the patient's sputum and the chest CT showed diffuse granular-like shadow, the patient was diagnosed as miliary tuberculosis and treatment with combined use of INH, RFP, EB, and PZA was started. Subsequently, fever started to subside and the miliary shadow on chest X-ray improved, however, six weeks after the start of treatment, hoarseness and dysphagia appeared. From the cervical CT and cervical angiography findings, the diagnosis of right subclavian artery impending ruptured aneurysm was made. Because the patient's sputum was acid-fast bacilli positive and because the patient had undergone thymectomy, it was decided that it would be difficult to treat her by a thoracotomy again. Therefore, a right subclavian artery stent insertion, right subclavian artery-right common carotid artery bypass creation operation was carried out with the objective of blocking the flow of blood to the aneurysm. The hoarseness and dysphagia improved post-operatively and the patient's progress is being monitored. Tuberculous aneurysms are a rare affection and they are mostly discovered when the autopsy is done, however, this case was diagnosed due to the manifestation of subjective symptoms. While this case was not diagnosed histopathologically, it is envisaged from the clinical progress that this was a tuberculous subclavian aneurysm complicated during the treatment for miliary tuberculosis.     

Huge brain abscess and invasive pulmonary disease by fungal infection during chemotherapy for infantile acute myeloid leukemia

The authors encountered a 7-year-old girl with a huge brain abscess and invasive pulmonary lesion due to fungus, who had been treated for acute myelogenous leukemia (AML). Although she was administered voriconazole to prevent fungal infection, she developed partial seizure and paralysis of the left side because of the huge brain abscess. Fungus culture and serum fungal markers, including Aspergillus antigen, were all negative. She underwent drainage and surgical resection of necrotic tissue after antifungal agents, including liposomal amphotericin B (L-AMB). Resection pathology revealed localized fungal infection, suspected as due to zygomycosis. The cerebral lesion reduced after the operation and the pulmonary lesion also vanished. We discontinued AML treatment because of the severe fungal infection; however, she has remained in continuous remission. Although Lipo-AMPH and itraconazole are comparatively effective for zygomycosis, progressive disseminated zygomycosis is extremely intractable. Our case underlines the feasibility and successful application of combined conventional antifungal agents and surgical resection in such a patient.     

Infusion therapy at outpatient clinic in chronic end-stage heart failure

OBJECTIVES: To determine whether drug infusions at ambulatory clinic in patients with end stage congestive heart failure are safe and reduce the period of hospitalization. METHODS: Between May 2000 and November 2006, 21 ambulatory patients with end stage congestive heart failure were treated with infusions of the natriuretic peptide, carperitide (6 patients, 43 infusions of mean 0.033 microg/kg/min for mean 3.7 hr), the phosphodiesterase inhibitor, olprinone (19 patients, 75 infusions of mean 0.11 microg/kg/min for mean 3.8 hr), or the catecholamines, dopamine or dobutamine(5 patients, 89 infusions of mean 3.3 microg/kg/min for mean 3.2 hr). RESULTS: Systolic and diastolic blood pressure was lower after infusion of carperitide, whereas catecholamines increased systolic blood pressure and heart rate (all differences from baseline p < 0.0001). Olprinone changed neither blood pressure nor heart rate. No adverse effect was observed, including arrhythmias or change in blood pressure requiring cessation of drug infusion. Mean urinary output per infusion was 979 ml for carperitide, 720ml for olprinone, and 594ml for catecholamines. There was no correlation between mean urinary output and dose of furosemide administered during intermittent infusion therapy. There was a close correlation between pre-infusion blood pressure and urinary output(systolic: p < 0.05; diastolic: p < 0.0001). Infusion therapy reduced the length of hospitalization (p < 0.05) in 7 patients from April 2005. CONCLUSIONS: Ambulatory, low-dose infusion therapy may not decrease the mortality of patients in end-stage congestive heart failure, but was safe and might represent an acceptable end-of-life therapeutic option.     

Multiple brown tumors in primary hyperparathyroidism caused by an adenoma mimicking metastatic bone disease with false positive results on computed tomography and Tc-99m sestamibi imaging: MR findings

We encountered an unusual case of hyperparathyroidism with both hemosiderin deposits on the ribs and low intensity on T2-weighted magnetic resonance imaging (MRI) caused by a parathyroid adenoma with multiple brown tumors that mimicked metastatic bone tumor due to false positive results on computed tomography (CT) and Tc-99m sestamibi (MIBI) imaging. The patient, a middle-aged woman, had very high serum levels of calcium (14.1 mg/dl), alkaline phosphatase (9,369 IU/l) and intact-PTH (12,400 pg/ml), and a large tumor (2.5 cm in diameter) in the lower portion of the left lobe of the thyroid. Plain X-ray revealed a soft tumor in the left chest wall. On CT scan, there were multiple destructive masses in the ribs, including large intramedullary masses on both 3rd ribs. On MIBI scintigraphy, there was strong late uptake in the lower portion of the left cervical region, both 3rd ribs, and the left 7th, 8th, and 10th ribs. T2-weighted image MRI scans showed that both 3rd ribs had a low intensity with hemosiderin deposits. These findings suggested that the patient had hyperparathyroidism with multiple bone metastases due to carcinoma of the parathyroid gland. However, on pathology, the resected tumor of lower portion of the left lobe of thyroid was diagnosed as a parathyroid adenoma, and the tumors of the left 3rd and 7th ribs, as well as the right 2nd rib, were shown to be brown tumors. After resection, the patient's serum levels of calcium, alkaline phosphatase, and intact-PTH normalized. At 1.5-years follow-up, CT, MIBI, and MRI scans showed no abnormal findings. It is necessary to determine whether MRI can be used to distinguish between brown tumors and metastases caused by carcinoma of the parathyroid gland.