All-trans retinoic acid (ATRA) is the standard induction treatment for acute promyelocytic leukemia (APL). Quite many ATRA-related side effects, including retinoic acid syndrome, were reported. So far, it has rarely been reported that Sweet's syndrome, characterized by fever, neutrophilia, painful erythematous cutaneous plaques, dense dermal infiltrates of mature neutrophils and rapid response to steroid therapy, is associated with ATRA. In the case that Sweet's syndrome associated with ATRA is found, physicians will have to face a great challenge over the possibility of infectious conditions. We present here a case of Sweet's syndrome associated with ATRA. A 35-year-old female with APL developed fever, painful erythematous cutaneous plaques on both cheeks, right wrist and both shins during induction chemotherapy with ATRA. A skin biopsy revealed a dense dermal infiltrate, consisting of mature neutrophils without vasculitis or cutaneous immunoglobulin deposits, which is compatible with Sweet's syndrome. Oral prednisone was administered and the lesions started to improve within 48 hours
Adult ; Biopsy, Needle ; Case Report ; Female ; Follow-Up Studies ; Human ; Leukemia, Promyelocytic, Acute/diagnosis/*drug therapy ; Prednisone/administration & dosage ; Risk Assessment ; Sweet's Syndrome/*chemically induced/drug therapy/*pathology ; Tretinoin/*adverse effects/therapeutic use

Glomerulonephritis associated with visceral abscess is being increasingly recognized. The association of glomerulonephritis with visceral suppuration in the absence of endocarditis was first described by Whitworth and associates. Abscesses were most frequently located in the respiratory tract but have been reported at numerous other sites, including appendix, uterus, aorto-femoral bypass graft and cutaneous wound. This report documents the apparently rare occurrence of glomerulonephritis with acute renal failure in association with pyogenic liver abscess. The need for awareness of glomerulonephritis as a cause of acute renal failure in pyogenic liver abscess is highlighted.
Aged ; Antibiotics/administration & dosage ; Biopsy, Needle ; Case Report ; Drainage/methods ; Follow-Up Studies ; Glomerulonephritis/*complications/*diagnosis/therapy ; Human ; Liver Abscess/*complications/*diagnosis/therapy ; Male ; Pseudomonas Infections/*complications/*diagnosis/therapy ; Tomography, X-Ray Computed ; Treatment Outcome

Retinal vascular lesions are the most common ophthalmologic manifestation of systemic lupus erythematosus (SLE), occurring in 3% to 29% of cases, generally late in the disease. More rare is the severe vaso-occlusive disease, often termed "retinal vasculitis", which includes central retinal artery occlusion, multifocal arteriolar occlusions, extensive capillary nonperfusion and central venous occlusion. Patients with SLE and raised serum concentrations of anticardiolipin antibodies (ACA) have a higher risk of developing occlusive ocular vascular disease. We report a case in which retinal involvement was an earlier manifestation of SLE in a patient without ACA.
Adolescent ; Adrenal Cortex Hormones/administration & dosage ; Angiography ; Case Report ; Diagnosis, Differential ; Female ; Follow-Up Studies ; Human ; Lupus Erythematosus, Systemic/*diagnosis/drug therapy ; Ophthalmoscopy ; Retinal Vein Occlusion/*diagnosis/drug therapy ; Treatment Outcome ; Visual Acuity

Spontaneous medialstinal emphysema (pneumomediastinum) and pneumopericardium may be defined as the presence of free air or gas in the mediastinal structures and in the pericardial sac without an apparent precipitating cause. It most frequently occurs in young healthy adults without serious underlying pulmonary disease. Although pneumomediastinum and pneumopericardium is often asymptomatic, it may cause pain in the neck and chest, dysphonia and shortness of breath. Treatment is supportive unless the patient has a history of trauma from foreign body aspiration. The course of spontaneous pneumomediastinum and pneumopericardium is usually benign and self-limited. A case of spontaneous pneumomediastinum, pneumopericardium and subcutaneous emphysema in a 20-year-old male is reported in this paper
Adult ; Blood Gas Analysis ; Case Report ; Follow-Up Studies ; Human ; Male ; Mediastinal Emphysema/*complications/*radiography ; Pneumopericardium/*complications/*radiography ; Radiography, Thoracic ; Remission, Spontaneous ; Tomography, X-Ray Computed

Malignant peripheral nerve sheath tumor (MPNST) is defined as any malignant tumor arising from or differentiating toward the cells of the peripheral nerve sheath. MPNST accounts for about 5-10% of all soft tissue tumors and is often associated with neurofibromatosis type I (NF-1, von Recklinghausen's disease). It is one of the malignant tumors associated with von Recklinghausen's disease. Its common site is the lower and upper extremities, trunk, head and neck. But intrathoracic manifestations are very rare. We report a case of a 40 year-old man with multiple neurofibromatosis who was presented with an intrathoracic malignant peripheral nerve sheath tumor
Adult ; Case Report ; Diaphragm/pathology ; Human ; Lung/pathology ; Male ; Nerve Sheath Tumors/complications/diagnosis/*pathology ; Neurofibromatosis 1/complications/diagnosis/*pathology ; Prognosis ; Severity of Illness Index ; Thoracic Neoplasms/complications/diagnosis/*pathology

BACKGROUND: It has been widely accepted that the epitope(s) and/or functional characteristics of thyrotropin receptor antibodies (TSHRAb) from Graves' patients are heterogenous among patients. However, the clinical significance of such heterogeneity has not been systematically evaluated yet. We were to elucidate and find the clinical significance of heterogeneity for TSH receptor antibodies in Graves' disease. METHODS: We measured stimulating TSHRAb (TSAb) activities using CHO-hTSHR cells, FRTL-5 cells and chimeric receptor expressing cells (Mc1 + 2 and Mc2), specific blocking TSHRAb (TSBAb) activities using Mc2 cells and TBII activities using porcine thyroid membrane in 136 patients with untreated hyperthyroid Graves' disease. RESULTS: Based on various TSHRAb activities from each patient, the patients could be categorized into 7 subgroups by cluster analysis; 1) Group 1 (n = 41) was characterized by moderate TSAb activities both in CHO-hTSHR cells and in FRTL-5 cells, typical TSAb epitope, rare blocking antibodies and high TBII activities. 2) Group 2 (n = 16) was characterized by the presence of blocking TSHRAb in most patients, albeit the other characteristics were the same as those in Group 1. 3) Group 3 (n = 19) patients had low TSAb activities both in CHO-hTSHR cells and in FRTL-5 cells, seldom had blocking TSHRAb, but they had high TBII activities. 4) Group 4 (n = 30) could be categorized as 'mild disease' group, as they had low activities in all kinds of TSHRAb assay and had low antimicrosomal antibody activities. 5) Group 5 (n = 14) was characterized by moderate TSAb activities with atypical epitope(s), rare blocking TSHRAb and moderate TBII activities. 6) Group 6 (n = 10) patients had very high TSAb activities with typical epitopes, seldom blocking TSHRAb and low TBII activities. 7) Group 7 (n = 6) was characterized by very high TSAb activities with atypical epitopes and high TBII activities. Pretreatment serum thyroid hormone level was low only in group 4 patients compared to the other 6 groups (p < 0.05). The size of goiter was significantly larger in those in group 1 and group 3 (p < 0.05) compared to the other 5 groups. The prevalence of clinically significant ophthalmopathy was higher in group 2 patients than the other 6 groups (50% vs. 27.5%, p = 0.06). Among 6 kinds of TSHRAb activities, only the blocking TSHRAb activity was significantly associated with the presence of ophthalmopathy in multivariate analysis. CONCLUSION: These results suggest that the differences in epitopes for TSAb or the presence of blocking TSHRAb is not a major factor in determining the degree of thyrotoxicosis in Graves' disease. Although the pathogenic mechanism is not clear yet, we suggest that patients with ophthalmopathy have different TSHRAb repertoire from those without ophthalmopathy in Graves' disease.
Adolescent ; Adult ; Aged ; Cluster Analysis ; Comparative Study ; Female ; Graves' Disease/*classification/*immunology ; Human ; Immunoglobulins, Thyroid-Stimulating/*analysis/blood ; Logistic Models ; Male ; Middle Age ; Multivariate Analysis ; Receptors, Thyrotropin/analysis/*immunology ; Sensitivity and Specificity ; Severity of Illness Index

BACKGROUND: BCG, a potent inducer of Th1 immune response, has been suggested to suppress Th2 response which is known to mediate IgE-mediated allergic disorders, in particular allergic asthma. Schultz-Dale reaction is known to be a model of IgE-mediated hypersensitivity. This study was done to investigate whether BCG infection suppresses the Schultz-Dale reaction by inhibiting Th2 response and allergen-specific IgE production. METHODS: Twenty-four Sprague-Dawley rats were sensitized and provoked with ovalbumin (OVA). A pretreatment of 6 x 10(4) colony forming units of BCG or saline was done 7 days before sensitization. The Schultz-Dale reaction was represented as tracheal smooth muscle contractions to 50 micrograms/mL OVA challenge in vitro. Serum OVA-specific IgE levels and IFN-gamma and IL-4 concentrations in bronchoalveolar lavage fluid (BALF) were measured. RESULTS: The Schultz-Dale reaction and serum OVA-specific IgE levels were significantly decreased in BCG infected and OVA sensitized rats compared with only sensitized rats (p < 0.01 and p < 0.05, respectively). As compared with only sensitized rats, IL-4 concentration and a ratio of IFN-gamma:IL-4 in BCG infected and OVA sensitized rats were significantly decreased (p < 0.001) and increased (p < 0.05), respectively. The Schultz-Dale reaction was correlated with OVA-specific IgE levels (r = 0.50, p < 0.05), IL-4 concentration (r = 0.69, p < 0.001), and ratio of IFN-:IL-4 (r = -0.44, p < 0.05). OVA-specific IgE levels were correlated with IL-4 concentration (r = 0.61, p < 0.01) and ratio of IFN-gamma:IL-4 (r = -0.48, p < 0.05). CONCLUSION: These findings suggest that BCG infection prior to allergen sensitization may inhibit Schultz-Dale reaction developed in the sensitized rat tracheal smooth muscle via the suppressive effects of Th2 immune response and allergen-specific IgE production.
Animal ; Bronchial Provocation Tests ; Bronchoalveolar Lavage Fluid/*chemistry ; Comparative Study ; Cytokines/analysis ; Disease Models, Animal ; Hypersensitivity, Immediate/*immunology ; Immunization ; Immunoglobulin E/*analysis ; Male ; Mycobacterium bovis/*immunology ; Probability ; Rats ; Rats, Sprague-Dawley ; Reference Values ; Sensitivity and Specificity ; T-Lymphocytes/*immunology ; Tuberculosis/*immunology/veterinary

Granulomatous hypophysitis is a rare pituitary condition that commonly presents with enlargement of the pituitary gland. A 31-year-old woman was admitted to the hospital with a severe headache and bitemporal hemianopsia. Magnetic resonance imaging (MRI) showed an 18 x 10-mm sellar mass with suprasellar extension and compression of the optic chiasm. Interestingly, brain MRI had shown no abnormal finding 4 months previously. On hormonal examination, hypopituitarism with mild hyperprolactinemia was noted. The biopsy revealed granulomatous changes with multinucleated giant cells. We herein report this rare case and discuss the relevant literature.
Adult ; Biopsy ; Female ; Giant Cells/pathology ; Granuloma/complications/*diagnosis/therapy ; Headache/etiology ; Hemianopsia/etiology ; Humans ; Hyperprolactinemia/etiology ; Hypopituitarism/etiology ; Inflammation/complications/*diagnosis/therapy ; Magnetic Resonance Imaging ; Optic Chiasm/pathology ; Pituitary Diseases/complications/*diagnosis/therapy ; Pituitary Function Tests ; Pituitary Gland/*pathology/surgery ; Predictive Value of Tests ; Severity of Illness Index ; Treatment Outcome

Cardiotoxicity associated with 5-fluorouracil (FU) is an uncommon, but potentially lethal, condition. The case of an 83-year-old man with colon cancer who developed chest pain during 5-FU infusion is presented. The electrocardiogram (ECG) showed pronounced ST elevation in the lateral leads, and the chest pain was resolved after infusion of nitroglycerin. A coronary angiogram (CAG) revealed that the patient had significant atherosclerosis in the proximal left circumflex artery. Coronary artery spasm with fixed stenosis was considered, and a drug-eluting stent was implanted. After 8 hours, the patient complained of recurring chest pain, paralleled by ST elevation on the ECG. The chest pain subsided after administration of intravenous nitroglycerin followed by sublingual nifedipine. Repeated CAG showed patency of the previous stent. This case supports the vasospastic hypothesis of 5-FU cardiac toxicity, indicating that a calcium channel blocker may be effective in the prevention or treatment of 5-FU cardiotoxicity.
Aged, 80 and over ; Angina Pectoris/chemically induced ; Antineoplastic Combined Chemotherapy Protocols/administration & dosage/*adverse effects ; Calcium Channel Blockers/administration & dosage ; Colonic Neoplasms/*drug therapy ; Coronary Angiography ; Coronary Vasospasm/*chemically induced/diagnosis/therapy ; Drug-Eluting Stents ; Electrocardiography ; Fluorouracil/administration & dosage/*adverse effects ; Humans ; Leucovorin/administration & dosage/adverse effects ; Male ; Nifedipine/administration & dosage ; Nitroglycerin/administration & dosage ; Organoplatinum Compounds/administration & dosage/adverse effects ; Percutaneous Coronary Intervention/instrumentation ; Recurrence ; Severity of Illness Index ; Treatment Outcome ; Vasodilator Agents/administration & dosage

There have been reports of the coexistence of abdominal aortic aneurysm (AAA) with intra-abdominal malignancy including gastric, colonic, pancreatic, and renal. We herein report a case of a previously undiagnosed AAA and a presenting complaint consistent with acute cholecystitis. Following cholecystectomy, this was noted to be a rare form of chronic cholecystitis: xanthogranulomatous cholecystitis. There is a known possible association of this uncommon condition with gallbladder cancer. The management of concomitant pathologies can present a real challenge to the multidisciplinary team, especially with large aneurysms.
Adenocarcinoma/*complications/radiography/secondary/surgery ; Aged ; Aortic Aneurysm, Abdominal/*complications/radiography/surgery ; Biopsy ; Blood Vessel Prosthesis Implantation ; Cholecystectomy ; Cholecystitis/*complications/pathology/radiography/surgery ; Endovascular Procedures ; Female ; Gallbladder Neoplasms/*complications/pathology/radiography/surgery ; Granuloma/*complications/pathology/radiography/surgery ; Humans ; Tomography, X-Ray Computed ; Treatment Outcome ; Xanthomatosis/*complications/pathology/radiography/surgery