Benign metastasizing leiomyomas (BMLs) occur predominantly in women during reproductive years. The condition is characterized by uterine leiomyomas associated with the development, typically years later, of slow-growing metastatic lesions. The most commonly affected organs are the lungs, but BMLs have been reported in lymph nodes, deep soft tissues, mesentery, bones, the central nervous system, and the heart. In many cases, these lesions have an indolent course and are discovered rather incidentally. However, occasionally they can present with debilitating symptoms or even life-threatening complications. The presence of estrogen and progester- U TERINE LEIOMYOMAS HAVE been reported in as-sociation with extrauterine benign-appearing smooth muscle tumors. This condition has been described mainly in women of reproductive age but can occur at any age. It has been referred to in the literature as benign metastasizing leiomyoma (BML). This term was introduced in 1939 by Steiner (1) in his report of a case of a 36-yr-old woman who died after massive pulmonary and hilar lymphatic metastases of a uterine leiomyoma. The interest of this condition to endocrinologists is its susceptibility to the hormonal milieu. Knowledge of this susceptibility may lead to effective clinical control of this metastasizing tumor by hormonal manipulation. We report two cases of BML in which hormonal manipulation with LHRH agonists, antiestrogens, and aromatase inhibitors (AIs) caused alleviation of symptoms and regression of the metastatic lesions. Case 1A 47-yr-old Caucasian woman consulted because of recurrent left subscapular pain that presented every few weeks with spontaneous resolution within a few days. Her complaint was initially attributed to cervical arthrosis and treated with analgesics. Three years later she developed numbness over her left hand, and a firm mass of about 5 cm was noticed in the left supraclavicular area (Fig. 1). A magnetic resonance image (MRI) revealed a large paraspinal tumor involving the brachial plexus and extending into the vertebral canal (Fig. 2). C5-T1 laminectomies, debulking of the extraspinal, and near complete removal of the intraspinal component was carried out. Pathological evaluation revealed a well-differentiated leiomyosarcoma [1-3 mitoses per 10 high-power fields (HPFs) and no necrosis] with bony involvement (Fig. 3).Radiotherapy was planned but the patient refused it. Shortly afterward she began experiencing cervical pain unrelieved by opioid analgesia, with a similar cyclical pattern to her previous pain; this prompted consultation to endocrinology. Her past medical history was remarkable for a total hysterectomy at age 36 yr for a 6-cm uterine leiomyoma. The histological blocks were retrieved and reexamined; the new report described a tumor with ill-defined infiltrative borders, areas of coagulative necrosis, mitotic rate of 4/10 HPFs, and few pathological mitotic figures but no significant cytological atypia. The conclusion was smooth muscle tumor of low malignant potential (2). The histol...
ACTH-independent macronodular adrenal hyperplasia (AIMAH) is a very rare cause of endogenous Cushing’s syndrome (CS). In this review, the clinical characteristics, the pathophysiology, and the management of AIMAH are described. AIMAH typically presents with overt CS, but subclinical oversecretion of cortisol has been increasingly described. The diagnosis is suspected by adrenal nodular enlargement on conventional imaging following the demonstration of ACTH-independent hypercortisolism. Final diagnosis is established by histological examination of the adrenal tissue. Bilateral adrenalectomy is the treatment of choice but unilateral adrenalectomy has been proposed in selected cases. In patients with subclinical CS, the decision to treat should be individualized. The pathophysiology of this condition has begun to be elucidated in recent years. Diverse aberrant membrane-bound receptors expressed in a non-mutated form in the adrenal gland have been found to be implicated in the regulation of steroidogenesis in AIMAH. When systematically screened, most patients with AIMAH and CS or subclinical CS exhibit an in vivo aberrant cortisol response to one or various ligands suggesting the presence of aberrant adrenal receptors. A protocol designed to screen patients for the presence of these aberrant receptors should be undertaken in all patients with AIMAH. The identification of these receptors provides the potential for novel pharmacological therapies by suppressing the endogenous ligands or blocking the receptor with specific antagonists.
BackgroundModest reductions in weight and small increases in step- related activity (e.g., walking) can improve glycemic and blood pressure control in type 2 diabetes mellitus (DM2). We examined changes in these parameters following training in time- efficient preparation of balanced, low- energy meals combined with pedometer- based step count monitoring.MethodsSeventy- two adults with DM2 were enrolled in a 24- week program (i.e., 15 three- hour group sessions). They prepared meals under a chef’s supervision, and discussed eating behaviours/nutrition with a registered dietitian. They maintained a record of pedometer- assessed step counts. We evaluated changes from baseline to 24 weeks in terms of weight, step counts, hemoglobin A1c (HbA1c, glycemic control), blood pressure, and eating control ability (Weight Efficacy Lifestyle WEL Questionnaire). 53 participants (73.6%) completed assessments.ResultsThere were improvements in eating control (11.2 point WEL score change, 95% CI 4.7 to 17.8), step counts (mean change 869 steps/day, 95% CI 198 to 1,540), weight (mean change −2.2%; 95% CI −3.6 to −0.8), and HbA1c (mean change −0.3% HbA1c, 95% CI −0.6 to −0.1), as well as suggestion of systolic blood pressure reduction (mean change −3.5 mm Hg, 95% CI −7.8 to 0.9). Findings were not attributable to medication changes. In linear regression models (adjusted for age, sex, ethnicity, insulin use, season), a −2.5% weight change was associated with a −0.3% HbA1c change (95% CI −0.4 to −0.2) and a −3.5% systolic blood pressure change (95% CI −5.5 to −1.4).ConclusionsIn this ‘proof of concept’ study, persistence with the program led to improvements in eating and physical activity habits, glycemia reductions, and suggestion of blood pressure lowering effects. The strategy thus merits further study and development to expand the range of options for vascular risk reduction in DM2.
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