Resolution Of Hypercortisolism Research Articles

Irreversible Alterations of Susceptibility in Cushing's Disease: A Longitudinal QSM Study.

Chronic exposure to hypercortisolism is associated with accelerated aging and neurodegenerative diseases, while Cushing's disease (CD) is the most common form of endogenous hypercortisolism exposure. This study aimed to assess longitudinal susceptibility changes in CD using quantitative susceptibility mapping (QSM) before and after resolution of hypercortisolism. In this study, 24 CD patients and 24 healthy controls underwent magnetic resonance imaging (MRI) with QSM. All CD patients underwent MRI scans before and after the curative operation. After resolution of hypercortisolism, irreversibly altered susceptibility values were found in the anterior cingulate cortex, frontal lobe, caudate, and red nucleus. These alterations were significantly correlated with the changes in gray matter/white matter volumes and clinical features. Additionally, decreased susceptibility was found in several regions in CD patients. Chronic exposure to hypercortisolism may be related to susceptibility alterations and significantly correlated with altered brain volumes and clinical features. in CD patients. The decrease of susceptibility might suggest the involvement of the calcium deregulation in these alterations.

ODP303 Cushing's Syndrome Due to Ectopic Adrenocorticotropic Hormone-Secreting Metastatic Neuroendocrine Tumor

Abstract Introduction Cushing's syndrome (CS) due to ectopic ACTH secretion (EAS), is an infrequent form of ACTH-dependent CS. EAS results from unregulated ACTH secretion by neuroendocrine tumors (NETs) of various locations resulting in severe hypercortisolism. The progression of CS is usually precipitous. To improve outcomes, it is imperative to recognize early so that treatments of cortisol-induced comorbidities and targeted treatments can be initiated. Case Presentation A 74-year-old female presented to an outside hospital with fatigue, bilateral lower extremity edema and intractable hypokalemia. She was later transferred to our facility, where workup of CS was initiated. She underwent a 1mgdexamethasone suppression test (DST)which resulted in an unsuppressed 8amcortisol of 34.7 ug/dL (<2 ug/dL). This was followed by a 4mgDST, again resulting in an unsuppressed 8amcortisol of25.4 ug/dL. A 24-hour urine cortisol level was collected and resulted at 2,108.9 ug/d (<45 ug/d). ACTH level was elevated at 136 pg/ml (7.2-63.3 pg/ml). CT abdomen/pelvis identified liver lesions concerning for metastatic disease. PET DOTATATE revealed multiple liver lesions and a few pulmonary nodules. Biopsy of liver lesions demonstrated strongly positive staining of synaptophysin and chromogranin confirming NET, with a weak TTF-1 positivity suggestive of pulmonary origin. Lanreotide injections were initiated for tumor control and ketoconazole for treatment of persistent hypercortisolism. Despite titration of ketoconazole, symptoms and labs showed minimal improvement. Osilodrostat was then added as a third line and examethasone was initiated in a block and replace approach. The regimen of lanreotide, osilodrostat and ketoconazole improved her24-hoururine cortisol to 15 from 2,109 ug/24hr. Subsequently patient underwent trans-arterial chemoembolization (TACE) of the liver lesions with resolution of hypercortisolism. She is currently only on Lanreotide for tumor control. Discussion NET with ectopic ACTH production is rare. While primary tumor location was not definitively identified, multiple lung lesions and histopathology suggested primary lung tumor with hepatic metastases. 20-40% of all NETS originate in the chest. Patient management necessitates a multidisciplinary approach not only for the diagnosis and treatment of CS but also for the specific management of neuroendocrine tumors (NET). This case highlights a rare presentation of EAS, and aggressive treatment modalities needed to treat symptoms of excess cortisol production and tumor progression. Presentation: No date and time listed

Trends in regional morphological changes in the brain after the resolution of hypercortisolism in Cushing's disease: a complex phenomenon, not mere partial reversibility.

The adverse effects of hypercortisolism on the human brain have been highlighted in previous studies of Cushing’s disease (CD). However, the relative alterations in regional hypercortisolism in the brain remain unclear. Thus, we investigated regional volumetric alterations in CD patients. We also analyzed the associations between these volumetric changes and clinical characteristics. The study participants comprised of active CD (n = 60), short-term-remitted CD (n = 28), and long-term-remitted CD (n = 32) patients as well as healthy control subjects (n = 66). Gray matter volumes (GMVs) were measured via voxel-based morphometry. The GMVs of substructures were defined using the automated anatomical labeling (AAL) atlas. Trends toward normalization in GMV were found in most brain substructures of CD patients. Different trends, including enlarged, irreversible, and unaffected, were observed in the other subregions, such as the amygdala, thalamus, and caudate. Morphological changes in GMVs after the resolution of hypercortisolism are a complex phenomenon; the characteristics of these changes significantly differ within the brain substructures.

Thymic Hyperplasia Developing After Treatment of Cushing’s Syndrome, Management and Literature Review

Thymic Hyperplasia Developing After Treatment of Cushing’s Syndrome, Management and Literature Review Introduction/ Background: Rebound thymic hyperplasia (RTH) has been described after the resolution of hypercortisolism from multiple conditions. It can potentially lead to a variety of diagnostic dilemmas. We describe a case where thymic enlargement was seen after unilateral adrenalectomy for adrenal Cushing’s syndrome. Thymic hyperplasia developing after treatment of adrenal Cushing’s syndrome has been very rarely reported. Clinical Case: 43 year old female with type 2 diabetes, hypertension, morbid obesity, depression underwent left adrenalectomy for management of Cushing’s syndrome. Two years after adenoma resection, the patient underwent a CT angiogram of the chest as part of a work up for chest pain which revealed a new anterior mediastinal soft tissue attenuation mass (30–40 Hounsfield units) measuring 6.5cm x2.5cm x 3.7cm. It was found to be abutting the inferior margin of the left brachiocephalic vein and anterior margin of the aortic arch. MRI chest demonstarated mild homogeneous postcontrast enhancement and loss of signal on the opposed phase T1 sequence indicating microscopic fat, deemed to be consistent with thymic hyperplasia. Patient had a detailed discussion of risks versus benefits of biopsy/surgery/conservative management with thoracic surgeon and opted to be managed conservatively. MRI Chest done at 3 month follow up showed a decrease in the size of the mass to 5.8 x 2.4 x 5.cm. She has been on chronic hydrocortisone therapy since adrenalectomy and multiple attempts to wean her off of steroids have been unsuccessful. She continues to be followed closely in the outpatient setting for presumed RTH. Clinical Lesson/ Conclusion: This case highlights the importance of recognizing RTH in patients treated for Cushing’s syndrome. Rapid enlargement of the thymus post adrenalectomy can often lead to unnecessary biopsies and surgeries. The underlying mechanism though unclear has been attributed to depletion of thymic lymphocytes caused by high cortisol concentrations of the plasma leading to RTH when the cortisol level declines. Keywords: Thymic Hypertrophy, Cushing’s syndrome, Thymic hyperplasia

Cushing's Syndrome Effects on the Thyroid.

The most known effects of endogenous Cushing’s syndrome are the phenotypic changes and metabolic consequences. However, hypercortisolism can exert important effects on other endocrine axes. The hypothalamus–pituitary–thyroid axis activity can be impaired by the inappropriate cortisol secretion, which determinates the clinical and biochemical features of the “central hypothyroidism”. These findings have been confirmed by several clinical studies, which also showed that the cure of hypercortisolism can determine the recovery of normal hypothalamus–pituitary–thyroid axis activity. During active Cushing’s syndrome, the “immunological tolerance” guaranteed by the hypercortisolism can mask, in predisposed patients, the development of autoimmune thyroid diseases, which increases in prevalence after the resolution of hypercortisolism. However, the immunological mechanism is not the only factor that contributes to this phenomenon, which probably includes also deiodinase-impaired activity. Cushing’s syndrome can also have an indirect impact on thyroid function, considering that some drugs used for the medical control of hypercortisolism are associated with alterations in the thyroid function test. These considerations suggest the utility to check the thyroid function in Cushing’s syndrome patients, both during the active disease and after its remission.

Aldosterone- and cortisol-cosecreting adrenal adenoma, ovarian hyperthecosis and breast cancer.

SummaryWe describe a 56-year-old postmenopausal woman with hypertension, hypokalemia and severe alopecia who was found to have a 4.5-cm lipid-poor left adrenal mass on CT scan performed to evaluate her chronic right-sided abdominal pain. Hormonal studies revealed unequivocal evidence of primary aldosteronism and subclinical hypercortisolemia of adrenal origin. Although a laparoscopic left adrenalectomy rendered her normotensive, normokalemic and adrenal insufficient for 2.5 years, her alopecia did not improve and she later presented with facial hyperpigmentation acne, worsening hirsutism, clitoromegaly, and an estrogen receptor-positive breast cancer. Further testing demonstrated markedly elevated serum androstenedione and total and free testosterone and persistently undetectable DHEAS levels. As biochemical and radiologic studies ruled out primary adrenal malignancy and obvious ovarian neoplasms, a bilateral salpingo-oophorectomy was undertaken, which revealed bilateral ovarian hyperthecosis. This case highlights how the clinical manifestations associated with hyperaldosteronism and hypercortisolemia masqueraded the hyperandrogenic findings. It was only when her severe alopecia failed to improve after the resolution of hypercortisolism, hyperandrogenic manifestations worsened despite adrenal insufficiency and an estrogen receptor-positive breast cancer was found, did it becomes apparent that her symptoms were due to ovarian hyperthecosis.Learning points:As cortisol cosecretion appears to be highly prevalent in patients with primary aldosteronism, the term ‘Connshing’ syndrome has been suggested.The associated subclinical hypercortisolemia could be the driver for the increased metabolic alterations seen in patients with Conn syndrome.The identification of these dual secretors before adrenal venous sampling could alert the clinician about possible equivocal test results.The identification of these dual secretors before unilateral adrenalectomy could avoid unexpected postoperative adrenal crises.Hyperfunctioning adrenal and ovarian lesions can coexist, and the clinical manifestations associated with hypercortisolemia can masquerade the hyperandrogenic findings.

Neuropsychological and Emotional Functioning in Patients with Cushing's Syndrome.

Patients with Cushing's syndrome (CS) frequently report impairments in cognitive and emotional functioning. Given neuroimaging research that implicates alterations in structure and function in the brain in this population, goals of this study were to investigate neuropsychological and emotional functioning, with particular emphasis on complex attention and memory. In a clinical sample of 18 adults with CS referred for neuropsychological evaluation (age 41.6 ± 10.6, 72% Caucasian), patients' most common subjective complaints were in attention and increased irritability. On objective testing, patients exhibited significant declines in the consistency of their sustained attention and visual-spatial functioning compared to normative peers. Patients exhibited on average significantly reduced initial learning following first exposure to visual and verbal stimuli but intact retention of information learned. Patients with CS endorsed highly elevated levels of somatization, depression, and anxiety, and 59% of them scored in the clinically elevated range for somatization and depressive symptomatology. Exploratory analyses suggested that the 11 patients with active Cushing's exhibited lower processing speed, poorer sustained attention, naming, and cognitive flexibility compared to the 7 patients who achieved biochemical remission. Patients with active Cushing's also reported higher levels of somatization and anxiety compared to patients in biochemical remission. Overall, this study provides new insights into complex attention and memory deficits in patients with CS and concern regarding cognitive and emotional issues despite resolution of hypercortisolism. Our study opens several avenues for further exploration.

SAT-LB76 Impact of Glucocorticoid Cosecretion in Primary Aldosteronism on Thyroid Autoantibody Titers During the Course of Disease

Context: Excess aldosterone is associated with the increased risk of cardio- and cerebrovascular events as well as metabolic comorbidities not only due to its hypertensive effect but also due to its proinflammatory action. Autonomous cortisol secretion (ACS) in the setting of primary aldosteronism (PA) is known to worsen cardiovascular outcome and potentially exhibit immunosuppressive effects.The aim of this study was to determine the impact of ACS status in patients with PA on kinetics of thyroid autoantibodies (anti-TPO, anti-TG) pre and post therapy initiation.Patients and Methods: 97 PA patients (43 with unilateral, 54 with bilateral PA) from the database of the German Conn’s Registry were included. Anti-TPO and anti-TG levels were measured pre and 6 to 12 months post therapeutic intervention. Patients were assessed for ACS according to their 24h urinary cortisol excretion, late night salivary cortisol and low-dose dexamethason suppression test.Results: Abnormal test results in line with ACS were identified in 74.2% of patients. Significant increases in anti-TPO levels were observed in adrenalectomized patients with at least one abnormal test (p = 0.049), adrenalectomized patients with at least two pathological ACS tests (p = 0.015) and adrenalectomized patients with pathologic dexamethasone suppression tests (p = 0.018). No antibody increases were observed in unilateral PA patients without ACS and in patients with bilateral PA receiving mineralocorticoid antagonist therapy.Conclusion: ACS appears to be a relevant factor in PA affecting thyroid autoimmune disease. The biochemical and clinical course maybe be exacerbated after resolution of hypercortisolism by adrenalectomy in PA.

An atypical case of ectopic ACTH syndrome in an adolescent boy.

Ectopic adrenocorticotropic hormone (ACTH) syndrome (EAS) is exceedingly rare in children and scarcely reported. Pancreatic neuroendocrine tumours (NETs) can rarely lead to secretion of ectopic ACTH. A 14-year-old boy presented with hyperpigmentation, hypertension and intermittent abdominal pain, and was diagnosed with endogenous hypercortisolism. An incidental pancreatic mass discovered on routine ultrasonogram (USG) revealed the source of ACTH. He underwent successful excision of the mass with resolution of hypercortisolism. The histopathology revealed a Pancreatic NET and immunohistochemistry was positive for ACTH stain.

Thigh Muscle Fat Infiltration Is Associated With Impaired Physical Performance Despite Remission in Cushing's Syndrome.

Muscle weakness is common in patients with Cushing's syndrome (CS) and may persist after the resolution of hypercortisolism. Intramuscular fatty infiltration has been associated with the deterioration of muscle performance in several conditions. To quantify the degree of fatty infiltration in the thigh muscles of "cured" CS patients and evaluate the relationship between intramuscular fatty infiltration and physical performance. This was a cross-sectional study. Tertiary referral center. Thirty-six women with CS in remission, and 36 controls matched for age, BMI, menopausal status, and level of physical activity. We analyzed the percentage fat fraction (FF) of the thigh muscles in the anterior, posterior, and combined anterior and posterior compartments using MRI and 2-point Dixon sequence. We assessed muscle function and strength using the following tests: gait speed (GS), timed up and go (TUG), 30-second chair stand, and hand grip strength. Fat fraction in all the compartments analyzed was increased in patients as compared with controls. The performance on TUG, 30-second chair stand, and GS was more impaired in CS patients versus controls. In patients, greater FF was negatively associated with performance on functional tests. Fat fraction in the combined anterior and posterior compartments predicted performance on TUG (ß 0.626, P < 0.000) and GS (ß -0.461, P = 0.007), after adjusting for age, BMI, menopausal status, and muscle mass. Thigh muscle fatty infiltration is increased in "cured" CS patients and is associated with poorer muscle performance. Future studies are needed to establish therapeutic strategies to improve muscle weakness in these patients.

Reversible and the irreversible structural alterations on brain after resolution of hypercortisolism in Cushing’s disease

The adverse effects of hypercortisolism on the human brain have been highlighted in previous studies of Cushing’s disease (CD). However, the reversibility of brain damage after the resolution of hypercortisolism remains unclear. Thus, we studied the potential volumetric reversibility in biochemically remitted CD patients. Cross-sectional analysis demonstrated the active CD patients (n = 61) had the smallest gray matter (GM) volumes (553.33 ± 45.90 CM3) among four groups. While the GM volumes of short-term remitted CD patients (586.62 ± 46.89 CM3, n = 28) and long-term remitted CD patients (596.58 ± 45.95 CM3, n = 35) were between those of the active CD patients and healthy control subjects (628.14 ± 46.88 CM3, n = 74). Moreover, significant positive correlations between remitted time and GM volumes were only found in short-term remitted CD patients. On the contrary, the alterations of white matter (WM) in CD patients seem to be independent of concomitant hypercortisolism, persisting after remission. A preliminary longitude analysis also demonstrated similar results. Volumetric reversibility of GM, but not WM is highly prevalent in short-term after resolution of hypercortisolism in Cushing disease. Our study enhances our understanding of the reversible and the irreversible structural alterations in the human brain due to hypercortisolism.

Autoimmune Diseases in Patients with Cushing’s Syndrome after Resolution of Hypercortisolism: Case Reports and Literature Review

Introduction Cushing's syndrome (CS) is a clinical condition characterized by excessive cortisol production, associated with metabolic complications, such as diabetes mellitus, dyslipidemia, metabolic syndrome, hypertension, and cardiovascular diseases. Nowadays, the occurrence of autoimmune diseases in CS have not been completely evaluated in the previous studies. Objective The aim of this study was to evaluate the occurrence of autoimmune diseases in CS patients after successfully treated. Materials and Methods From January 2001 to December 2017, in our Secondary Hypertension Unit, we evaluated 147 CS patients (91 with ACTH-independent disease, 54 with ACTH-dependent disease, and 2 patients with ectopic ACTH production. Results 109 CS patients (74.1%) were surgically treated (67 ACTH-independent CS patients (61.5%) undergone adrenalectomy and 42 ACTH-dependent CS (38.5%) undergone transsphenoidal surgery) and evaluated after 6, 12, and 24 months after clinical and biochemical remission of disease. In 9 (8.3%) of overall treated CS patients (8.3%), during follow-up, we observed the onset of some manifestations of autoimmune diseases. In particular, one patient had a systemic lupus erythematosus, one patient had rheumatoid arthritis, 4 patients reported autoimmune thyroiditis (Basedow-Graves' disease and Hashimoto's thyroiditis), one patient had clinical features of psoriasis, one patient showed myasthenia gravis, and one patient had giant cell arteritis. Conclusions Our results demonstrate that patients successfully treated for CS could develop autoimmune diseases. Therefore, after treatment, CS patients need to be strictly monitored in order to evaluate the possible onset of autoimmune diseases.

Volumetric magnetic resonance imaging analysis in patients with short-term remission of Cushing's disease.

The data on patients with short-term remission of Cushing's disease (CD) might provide information that is not available from previous long-term remission studies. We aimed to investigate structural changes in the brain in these patients and to examine whether these changes were associated with clinical characteristics. A cross-sectional study was performed. Thirty-four patients with CD (14 with CD in short-term remission and 20 with active CD) and 34 controls matched for age, sex and education underwent clinical evaluation and magnetic resonance imaging brain scans. Biometric measurements, disease duration and remission duration data were collected. Grey matter volumes in the whole brain were examined using voxel-based morphometry (VBM). No differences were observed in the grey matter volumes of the medial frontal gyrus (MFG) and cerebellum between the patients with remitted CD and healthy controls, whereas patients with active CD had smaller grey matter volumes in these two regions compared with controls and patients with remitted CD. Furthermore, significant correlations were found between remission time and grey matter values in these regions in short-term remission patients with CD. Additionally, greater grey matter volumes in the bilateral caudate of short-term remission patients with CD were observed. Trends for structural restoration were found in CD patients with short-term remission. This finding was associated with the number of days elapsed since curative surgery and the current age of the patients. This study enhances our understanding of potential reversibility after the resolution of hypercortisolism in CD patients.

Skeletal Fragility in Endogenous Hypercortisolism.

Skeletal fragility is a frequent complication of endogenous hypercortisolism, and fragility fractures may be the first clinical manifestation of the disease. Fractures involve more frequently the vertebrae and may occur in 30-50% of the patients exposed to glucocorticoid excess, in close relationship with severity and duration of hypercortisolism. Although improvement of bone mineral density was reported after resolution of hypercortisolism, there are patients with persistently high fracture risk after the cure of hypercortisolism, and other patients in whom the resolution of hypercortisolism may take a long time, implying a multistep therapeutic approach. Since vertebral fractures tend to occur early during the natural history of disease, a skeletal-specific approach should be undertaken in these patients; however, the cost-effectiveness of this approach is still largely unknown since data on effectiveness and safety of bone-active drugs in endogenous hypercortisolism are scarce.

Editorial: Neurological and Psychiatric Disorders in Endocrine Diseases

In the last few decades, a number of reciprocal associations between endocrine system dysfunctions and neurological or psychiatric manifestations have appeared, and only in the minority of the cases this link has been fully elucidated (1). Unfortunately, the complexity, and sometimes also the severity, of the clinical issues and the limited availability of reliable experimental models for the study of the endocrine system–nervous system cross-interactions do not help the fast achievement of scientific progresses. Isolated neurological or psychiatric symptoms as well as more complex disorders can occur in the setting of most endocrine diseases. Such manifestations can be the direct result of hyper- or hyposecretion of hormones from the endocrine glands or may occur as events secondary to the pathogenic mechanisms of the endocrinopathy, like in the case of autoimmunity affecting both endocrine glands and the brain (2). Moreover, the medical or surgical treatment of endocrine diseases can sometimes determine the occurrence of neurological or psychiatric syndromes. Some genetic aberrations can also lead to conditions affecting both the endocrine and the nervous system with a variety of possible manifestations. On the other side, many psychiatric or neurological diseases, including but not limiting to conditions, which affect the hypothalamus or the pituitary gland, may impair the physiological endocrine functions and demonstrate once again the intricate complexity of the psychoneuroendocrine connection. Covering all basic, translational, and clinical aspects in a single Research Topic on the “Neurological and psychiatric disorders in endocrine diseases” cannot be realistically feasible. The scope is simply raising a number of interesting points from experts in the field to bring a contribution to the knowledge and maybe direct future research in this very intriguing area. Thus, we have collected reviews and original research articles, which may speak to a wide audience, including neurologists, psychiatrists, endocrinologists, and basic scientists in neuroscience, endocrinology, and metabolism. In the clinical area, an interesting study assesses the plasma levels of the muscle-derived hormone irisin in anorexic women, without finding any significant correlation between irisin level and physical exercise (3). Another fascinating study analyzes the complexity of psychiatric and personality disorders in women with polycystic ovary syndrome, highlighting the issue of psychological distress in this patient group (4). A comprehensive picture of gender dysphoria, a psychiatric condition, which requires endocrine management, in Ireland is presented (5). In a short review, the most relevant reproductive, neurodevelopmental, and genetic aspects of hypogonadotropic hypogonadal syndromes are outlined (6). A German study indicates that psychopathology significantly predicts quality of life in patients with acromegaly and suggests that depressive symptoms and anxiety, being modifiable factors, may represent relevant targets for a broad treatment intervention in acromegalic patients (7). In a concise review, the psychiatric alterations, the neurocognitive impairment, and the altered quality of life affecting at some extent the majority of the patients with Cushing’s syndrome are summarized, and the authors highlight that resolution of hypercortisolism, a challenging and non-granted achievement, does not always lead to the complete remission of the neuropsychiatric changes or restore the quality of life (8). With an incursion in the surgical field, the complications potentially affecting the laryngeal nerves during thyroid surgery are extensively reviewed, suggesting that still the incidence of laryngeal nerves damage secondary to thyroid surgery cannot be suppressed but may be maintained in a low range with thorough surgical techniques and the use of intraoperative neuromonitoring (9). Three articles address the complex interactions between metabolism and neuropsychiatric symptoms. The first one focuses on biological differences between restrictive anorexia nervosa and constitutional thinness, a controversial concept to describe young girls who follow a normal diet and differ from restrictive anorexia nervosa on a number of endocrine parameters (10). At the opposite of the spectrum, the second one reviews the role of inflammatory processes in the neuropsychiatric comorbidity associated with obesity (11). Finally, the last one summarizes the fascinating link through ghrelin peptides between appetite/reward/growth hormone axis and psychiatric disorders (12).

Neuropsychiatric disorders in Cushing's syndrome.

Endogenous Cushing's syndrome (CS), a rare endocrine disorder characterized by cortisol hypersecretion, is associated with psychiatric and neurocognitive disorders. Major depression, mania, anxiety, and neurocognitive impairment are the most important clinical abnormalities. Moreover, patients most often complain of impairment in quality of life, interference with family life, social, and work performance. Surprisingly, after hypercortisolism resolution, despite the improvement of the overall prevalence of psychiatric and neurocognitive disorders, the brain volume loss at least partially persists and it should be noted that some patients may still display depression, anxiety, panic disorders, and neurocognitive impairment. This brief review aimed at describing the prevalence of psychiatric and neurocognitive disorders and their characterization both during the active and remission phases of CS. The last section of this review is dedicated to quality of life, impaired during active CS and only partially resolved after resolution of hypercortisolism.

Persistent Comorbidities in Cushing’s Syndrome after Endocrine Cure

It was assumed that resolution of hypercortisolism in Cushing syndrome (CS) was followed by normalization of morbidity; however, in the last decade evidence is accumulating that patients with cured CS still have increased morbidity and mortality after the biochemical control of hypercortisolism. Patients with CS have an increased cardiovascular and metabolic risk and persistent accumulation of central fat, with an unfavorable adipokine profile, not only during the active phase of the disease but also long after biochemical remission. Clinical management should be particularly careful in identifying global cardiovascular risk, as a primary goal during the followup of these patients, aimed at improving global vascular morbidity. Moreover bone mass is reduced not only due to the endogenous hypercortisolism but also due to duration and dose of exogenous glucocorticoid (GC) replacement therapy after surgery. Thus, therapy in operated patients with inhibition of the hypothalamic-pituitary-adrenal axis should be reduced to the lowest dose and duration possible. Specific treatments should be considered in patients with decreased bone mass, aimed at reducing the increased fracture incidence. Finally, cognitive and health related quality of life impairments, described in active disease, are still abnormal after endocrine cure. Thus, residual morbidity persists in cured CS, suggesting irreversibility of GC-induced phenomena, typical of chronic hypercortisolism.

Suspected metastatic Adrenocortical Carcinoma revealing as Pulmonary Kaposi Sarcoma in Adrenal Cushing's syndrome

Background: Kaposi sarcoma (KS) is a malignant disease most commonly diagnosed in the setting of a human immunodeficiency virus (HIV) infection and in patients receiving immunosuppressive treatment. Pulmonary KS has never been reported in association with endogenous Cushing’s syndrome (CS). Case presentation: A 60-year-old woman presented with symptoms and signs of CS. Adrenal CS was confirmed by standard biochemical evaluation. Imaging revealed a right adrenal lesion (diameter 3.5 cm) and multiple pulmonary nodules, suggesting a cortisol-secreting adrenal carcinoma with pulmonary metastases. The patient underwent right adrenalectomy with a pathohistological diagnosis of an adrenal adenoma. Subsequent thoracoscopic wedge resection of one lung lesion revealed pulmonary KS with positive immunostaining for human herpes virus 8 (HHV-8). HIV-serology was negative. Hydrocortisone replacement was initiated for secondary adrenal insufficiency after surgery. Post-operative follow up imaging showed complete remission of all KS-related pulmonary nodules solely after resolution of hypercortisolism. Conclusion: KS may occur in the setting of endogenous CS and may go into remission after cure of hypercortisolism without further specific treatment.

Occurrence of De Quervain's thyroiditis after resolution of hypercortisolism following SOM230 treatment for Cushing's disease and surgery for an adrenocortical adenoma: report of two cases

Searchable abstracts of presentations at key conferences in endocrinology ISSN 1470-3947 (print) | ISSN 1479-6848 (online)

Cushing syndrome, metabolic syndrome and inflammation: a suggested way out

Endogenous hypercortisolism is associated with an increased cardiovascular risk. Cushing Syndrome (CS) shares many clinical features with metabolic syndrome, including abdominal obesity, systemic arterial hypertension, insulin resistance, dyslipidemia, and thrombotic diatesis. Moreover, CS represents an interesting pattern of an endocrine disorder associated with chronic lowgrade inflammation which is not blunted by the resolution of hypercortisolism. The proinflammatory state that accompanies the metabolic syndrome may provide a connection between CS, inflammation and metabolic processes, which is highly deleterious for vascular functions. There is evidence that dietary patterns similar to those of the Mediterranean-style diet exert ositive effects on almost all components of the metabolic syndrome and other conditions associated with, including inflammation, insulin resistance, and endothelial dysfunction. Therefore, an intervention strategy based on lifestyle changes may play a role in patients with resolution of hypercortisolism in which the anti-inflammatory effects of cortisol are lost and cytokines levels are increased. In this setting, the Mediterranean healthy dietary pattern may represent an innovative approach in order to improve the disease course and to reduce in the long term the cardiovascular risk of people affected by CS