Primary leiomyosarcoma of the nipple-areola complex: report of a case and review of literature.

Primary leiomyosarcoma of the nipple-areola complex is extremely rare. Less than ten such cases have been reported in English literature so far. Herein we describe a 52-year-old female presenting with a 1.5 cmx1.1 cmx0.7 cm nodular lesion over her left nipple, and leiomyosarcoma was proved by pathological examination of the excised specimen. Positron emitted tomogram (PET) revealed no abnormal signal other than the primary site. Microscopically, this poorly circumscribed tumor was composed of interlacing bundles of smooth muscle cells with bizarre and pleomorphic nuclei, as well as prominent nucleoli. Its mitotic count was up to 7 mitoses per 10 high power fields (HPF). Immunohistochemical study of tumor cells revealed positive stain for alpha-smooth muscle actin and vimentin; and negative for cytokeratin, CD34 and S-100. Left simple mastectomy was undertaken and no residual mass lesion was noted on the resected specimen. Related literatures about the diagnosis and treatment for breast leiomyosarcoma will be presented here.

Castleman's disease mimicking intrapulmonary malignancy.

Castleman's disease is a rare disorder characterized by noncancerous growth that may develop in lymph node tissues throughout the body. Most often this occurs in the chest and abdomen, and rarely in the peripherals (10% to 15%). In this article we describe a case of asymptomatic intrapulmonary Castleman's disease mimicking pulmonary malignancy (hyaline-vascular type) in a 42-year-old man who underwent curative surgery.

Serum adiponectin is reduced in acromegaly and normalized after correction of growth hormone excess.

Adiponectin, an adipocyte-derived hormone, possesses insulin-sensitizing, antiinflammatory, and antiatherogenic properties. We hypothesized that hypoadiponectinemia was present in acromegaly, as in other conditions with increased insulin resistance and cardiovascular risk. Using an in-house RIA, serum adiponectin was determined in 35 patients with active acromegaly and 35 age-, sex-, and body mass index-matched healthy controls. Twenty-five patients were restudied after GH-lowering therapies. Serum adiponectin was significantly reduced in the acromegalic patients (4.3 +/- 1.8 vs. 6.7 +/- 1.8 microg/ml in controls; P < 0.001), but was increased after treatment with Sandostatin LAR, a long-acting somatostatin analog (5.8 +/- 2.6 vs. 3.8 +/- 1.6 microg/ml pretreatment; P < 0.001; n = 15) or transsphenoidal surgery (6.5 +/- 2.7 vs. 3.9 +/- 1.5 microg/ml preoperation; P < 0.01; n = 10). Fasting insulin was an independent determinant of serum adiponectin levels (P < 0.01) in control subjects, contributing to 11.7% of the variance in circulating adiponectin. In cultured 3T3-L1 adipocytes, adiponectin mRNA levels were decreased by insulin (1.5 microm; P < 0.005) or IGF-I (1 microg/ml; P < 0.05), but not by GH (1 microm) or somatostatin (1 microm). In conclusion, hypoadiponectinemia is present in active acromegaly, probably secondary to the inhibitory effect of high circulating insulin levels. Hypoadiponectinemia, reversible with GH-lowering therapies, may contribute to the increased insulin resistance and cardiovascular risk in patients with acromegaly.

Chronic treatment with growth hormone stimulates adiponectin gene expression in 3T3-L1 adipocytes.

Growth hormone (GH) is an important regulator of adiposity and systemic energy metabolism. Here, we have investigated the effects of GH on production of adiponectin, an anti-diabetic and anti-atherogenic hormone secreted exclusively from adipocytes. Analysis using real time quantitative PCR revealed that GH significantly increased adiponectin gene expression in a dose-dependent manner. Time course study showed that the expression of adiponectin gene started to increase only after 30 h of GH treatment (10(-8) M), suggesting it to be a chronic effect. GH-mediated induction of adiponectin gene expression was completely blocked by treatment with the Janus kinase2 (JAK2) inhibitor AG490 and the P38 mitogen activated protein (MAP) kinase inhibitor SB203580, while the specific inhibitors of phosphatidylinositol-3-kinase (LY294002) and p70S6 kinase (rapamycin) moderately enhanced GHs effect. Co-incubation of adipocytes with GH and the PPARgamma agonist rosiglitazone produced additive effects on induction of adiponectin gene expression. These results collectively suggest that GH increases adiponectin gene expression through the JAK2-P38 MAP kinase pathway, and that elevation of adiponectin production might represent a novel mechanism by which GH regulates systemic energy metabolism and insulin sensitivity.

Cytomegalovirus ischemic colitis of a diabetic renal transplant recipient.

We report a diabetic renal transplant recipient with cytomegalovirus (CMV) disease who presented with tarry stool diarrhea because of multiple colonic ulcerations. Histopathology revealed diffuse colonic ulcers following a process of ischemic vasculitis. The colonic ulcers disappeared dramatically after 2 wk of intravenous ganciclovir therapy. Hyper-immunosuppression was initially suspected but acute rejection (AR) developed after immunosuppressive reduction during the ganciclovir therapy. The AR was successfully reversed and the dosage of cyclosporine was returned to the same level prior to the onset of CMV disease. Our experience suggests that ganciclovir is quite effective for healing colonic ulcers caused by CMV and acute allograft rejection may occur during therapy.

Pulmonary granulomatosis associated with insoluble fillers in a heroin addict.

We report a case of acute respiratory distress syndrome (ARDS) and pulmonary granulomatosis after intravenous injection of heroin. This 46-year-old male had a 2-year history of heroin addiction. The recent admission was due to loss of consciousness after intravenous injection of diluted heroin with unknown filler. Initial chest X-ray revealed a picture of ARDS. Blood routine and biochemistry were normal except for leukocytosis. Urine morphine test was positive. The blood and sputum culture yielded no pathogens. After supportive treatment, his condition improved, and the follow-up chest X-ray showed diffuse micronodules in both lung fields. Subsequently, open-lung biopsy of the right upper and lower lobes on the 26th hospital day showed large amounts of foreignbody granulomas distributed over perivascular areas, bronchiolar areas and interstitium. Staining for bacteria, fungi, and mycobacteria was all negative. The morphology of foreign-bodies could be divided into 2 types: 1) periodic acid-Schiff (PAS)-positive, aggregated fine crystals in round form; and 2) PAS-negative, yellow long crystals. The pathological diagnosis of pulmonary granulomatosis was made based on the finding of filler foreign bodies. The nature of these foreign bodies remained undetermined. The case suggests that pulmonary granulomatosis with the radiographic appearance of chronic interstitial pneumonia can occur in patients with a history of heroin use.