The importance of the follow-up after bilateral adrenal adenomectomy for Cushing's syndrome

2018 ◽  
Author(s):  
Nicoleta Daniela Calinescu ◽  
Amalia Ioana Arhire ◽  
Carmen Gabriela Barbu
Keyword(s):  
Cushing’S Syndrome ◽  
Cushing's Syndrome

scholarly journals Median 10 years follow-up of patients with covert Cushing’s syndrome: a case series

2021 ◽  
Vol 15 (1) ◽  
Author(s):  
Farzad Najafipour ◽  
Amir Bahrami ◽  
Mitra Niafar ◽  
Jalil Houshyar ◽  
Monireh Halimi ◽  
...  
Keyword(s):  
Cushing’S Syndrome ◽  
Adrenocorticotropic Hormone ◽  
Hormone Secretion ◽  
Cushing's Syndrome ◽  
Whole Body ◽  
Whole Body Scan ◽  
Body Scan ◽  
Posterior Aspect ◽  
Ectopic Adrenocorticotropic Hormone

Abstract Background Ectopic adrenocorticotropic hormone secretion syndrome occurs in 10% of all patients with adrenocorticotropic-hormone-dependent hypercortisolism. It is usually associated with overt malignancies or with occult and indolent tumors. This study aims to confirm the source of ectopic adrenocorticotropic hormone in four patients with ectopic Cushing’s syndrome over time. Case presentation A 38-year-old Iranian man with Cushing’s syndrome underwent bilateral adrenalectomy since the source of ectopic adrenocorticotropic hormone secretion was not localized and pituitary imaging was normal. A whole-body scan revealed a right-lung tumoral mass with mediastinal lymph node metastasis. The mass was assumed a lung carcinoid tumor with mediastinal adenopathy. Right-lung mid-zone lobectomy and mediastinal lymphadenectomy were done. In a 47-year-old Iranian man with Cushing’s syndrome, whole-body computed tomography scan revealed a pulmonary nodule in the posterior segment of the left lower lobe of the lung. The third case was a 25-year-old Iranian man who presented with symptoms and signs of Cushing’s syndrome. Pituitary magnetic resonance imaging revealed a microadenoma 5 × 9 mm. Whole-body scan showed abnormal focal somatostatin receptors analog avid lesion in the posterior aspect of inferior third of right lung, highly suggestive of ectopic adrenocorticotropic-hormone-producing tumor. The last case was a 43-year-old Iranian woman with Marfan syndrome with a history of mitral and aortic valve replacement and chronic dissection of the aorta, who presented with symptoms and signs of Cushing’s syndrome. She underwent bilateral adrenalectomy 1 year later owing to failure to locate ectopic adrenocorticotropic hormone syndrome. Whole-body scan showed abnormally increased radiotracer uptake in the midline of the skull base and posterior aspect of the middle zone of left hemithorax and bed of left lobe of thyroid. Conclusion The clinical spectrum of ectopic adrenocorticotropic hormone secretion syndrome is wide, and distinguishing Cushing’s disease from ectopic adrenocorticotropic hormone secretion syndrome is difficult. Initial failure to identify a tumor is common. Pulmonary carcinoid or occult source of ectopic adrenocorticotropic hormone secretion syndrome is usually the cause. In occult cases of ectopic adrenocorticotropic hormone in which the tumor cannot be localized, serial follow-up with serial computed tomography, magnetic resonance imaging, or scintigraphy is recommended for several years until the tumor can be localized and treated.

scholarly journals Dopamine agonist-responsive Cushing’s disease

2019 ◽  
Vol 12 (2) ◽  
pp. bcr-2018-228045
Author(s):  
Gurpreet Anand ◽  
Andrea Bink ◽  
Felix Beuschlein ◽  
Christoph Schmid
Keyword(s):  
Growth Hormone ◽  
Dopamine Agonist ◽  
Cushing’S Syndrome ◽  
Tumour Size ◽  
Hypogonadotropic Hypogonadism ◽  
Immunohistochemical Analysis ◽  
Cushing's Syndrome ◽  
Central Hypothyroidism ◽  
Mri Scans

A 47-year-old Caucasian man was referred to our clinic with a severe clinical and biochemical phenotype of endogenous hypercortisolism for further evaluation and treatment. In addition to confirming adrenocorticotropic hormone (ACTH)-dependent Cushing’s syndrome, we found left temporal hemianopsia, massively increased prolactin, increased growth hormone/insulin-like growth factor 1 values, hypogonadotropic hypogonadism and central hypothyroidism. As the cause of these abnormalities we revealed an invasive macroadenoma of the pituitary secreting ACTH, prolactin and growth hormone, resulting not only in a clinically predominant picture of Cushing’s syndrome but also causing hypogonadotropic hypogonadism and central hypothyroidism. The patient responded surprisingly well to dopamine agonist treatment leading not only to normalisation of prolactin levels but also to clinical and biochemical remission of Cushing’s syndrome. Tumour size decreased successively in follow-up MRI scans. Despite lacking immunohistochemical analysis of tumour tissue, we assume plurihormonal secretion of ACTH, prolactin and growth hormone from pituitary macroadenoma, which fortunately responded well to dopamine agonist treatment.

Long-term morphological, hormonal, and clinical follow-up in a single unit on 118 patients with adrenal incidentalomas

2010 ◽  
Vol 162 (4) ◽  
pp. 779-785 ◽  
Author(s):  
R Giordano ◽  
E Marinazzo ◽  
R Berardelli ◽  
A Picu ◽  
M Maccario ◽  
...  
Keyword(s):  
Cushing’S Syndrome ◽  
Cumulative Risk ◽  
Cushing's Syndrome ◽  
Endocrine Function ◽  
Adrenal Incidentalomas ◽  
Term Outcome ◽  
Long Term Outcome ◽  
Significant Clinical Improvement

ObjectiveTo evaluate long-term morphological, functional, and clinical outcome in adrenal incidentalomas.Design and methodsA total of 118 patients (77 F and 47 M; age 62.3±1.0 years) with adrenal incidentalomas were evaluated at baseline and followed-up for median 3 years (range 1–10 years) by clinical, biochemical, hormonal, and morphological evaluation. Among them, six patients with diagnosis of subclinical Cushing's syndrome (SCS) underwent surgery.ResultsAt entry, 86% (n=102) of tumors were nonfunctioning (NF) and 14% (n=16) showed SCS. Comparing NF with SCS patients, a significantly higher percentage of dyslipidemia was found in the group of SCS patients (50 vs 23%,P=0.033). During follow-up, adrenal function remained normal in all NF patients, none of them developed subclinical or overt endocrine disease. The cumulative risk of mass enlargement was globally low (25%), but progressive up to 8 years. SCS was confirmed in all patients, and none of them shifted to overt Cushing's syndrome. The cumulative risk of developing metabolic–cardiovascular abnormalities was globally low (22%), but progressive up to 8 years and new diseases were recorded in the group of NF patients only (three patients with dyslipidemia, four with impaired fasting glucose/impaired glucose tolerance, and three with diabetes mellitus). SCS patients who underwent surgery did not show any significant clinical improvement.ConclusionsThe risk of mass enlargement, hormonal, and metabolic impairment over time is globally low. Conservative management seems to be appropriate, but further prospective studies are needed to establish the long-term outcome of such patients, especially for metabolic status, cardiovascular risk profile and their relationship with endocrine function.

scholarly journals Thymus hyperplasia after resolution of hypercortisolism in ACTH-dependent Cushing’s syndrome: the importance of thymic vein catheterization

2006 ◽  
Vol 154 (6) ◽  
pp. 807-811 ◽  
Author(s):  
Malebranche Berardo C Cunha Neto ◽  
Márcio Carlos Machado ◽  
Flávia Mesquita ◽  
Nina Rosa de Castro Musolino ◽  
Andrea Cecília Toscanini ◽  
...  
Keyword(s):  
Differential Diagnosis ◽  
Cushing’S Syndrome ◽  
Transsphenoidal Surgery ◽  
Cushing's Syndrome ◽  
Chest Ct ◽  
Spontaneous Resolution ◽  
Tumor Visualization ◽  
Histopathological Studies ◽  
Thymic Lobe

Thymic hyperplasia has been described after the resolution of hypercortisolism from several etiologies, causing great diagnostic dilemmas. We describe a case where the catheterization of the thymic vein was essential for the differential diagnosis of a thymic enlargement in an adrenalectomized patient with ACTH-dependent Cushing’s syndrome. The patient was a 48-year-old female with clinical and laboratorial data suggesting Cushing’s disease. She underwent a transsphenoidal surgery with no tumor visualization and no remission of the syndrome. Histopathological studies disclosed a normal pituitary. She underwent a bilateral adrenalectomy and 8 months later a chest CT showed an increase of left thymic lobe, which was previously non-existent. After a negative 111In-pentetreotide scintigraphy, the patient underwent simultaneous and bilateral catheterism of the petrosus sinuses and catheterization of the thymic and inominate veins and no ACTH gradient was shown among the sites of collection. She did not undergo thoracotomy and a follow-up was established. During the evolution, there was a spontaneous regression of the thymic lesion 38 months after the diagnosis. The ACTH gradient during the catheterization of thymic vein was essential for the differential diagnosis of the thymic enlargement tumor after hypercortisolism resolution in ACTH-dependent Cushing’s syndrome, especially in this case, where the ACTH source was occult, thus avoiding an invasive surgical procedure for a benign entity with spontaneous resolution.

scholarly journals Arterial thrombosis as primary presentation of endogenous Cushing’s syndrome

2019 ◽  
Vol 12 (2) ◽  
pp. bcr-2018-227491
Author(s):  
Vijay Alexander ◽  
Maria Koshy ◽  
Riddhi Dasgupta ◽  
Ronald Albert Carey
Keyword(s):  
Cushing’S Syndrome ◽  
Arterial Thrombosis ◽  
Clinical Manifestations ◽  
Dynamic Mri ◽  
Cushing's Syndrome ◽  
Positron Emission ◽  
History Of ◽  
Laboratory Investigations ◽  
Primary Presentation

Cushing’s syndrome is known to present with a characteristic set of clinical manifestations and complications, well described in literature. However, hypercoagulability remains an under recognised entity in Cushing’s syndrome. A 31-year-old woman from Southern India presented with history of fever, left upper quadrant pain and progressive breathing difficulty for 3 weeks. Clinical examination revealed discriminatory features of Cushing’s syndrome. Laboratory investigations showed biochemical features of endogenous ACTH-dependent Cushing’s syndrome. Imaging of the abdomen revealed splenic collection, left-sided empyema and extensive arterial thrombosis. Gadolinium enhanced dynamic MRI of the pituitary gland revealed no evidence of an adenoma while a Ga-68 DOTATATE positron emission tomography CT scan ruled out an ectopic Cushing’s. A diagnosis of endogenous Cushing’s syndrome causing a prothrombotic state with extensive arterial thrombosis was made. She was initiated on oral anticoagulation and oral ketoconazole for medical adrenal suppression. She subsequently underwent bilateral adrenalectomy and was well at follow-up.

scholarly journals Cushing's syndrome associated with a nested stromal epithelial tumor of the liver: hormonal, immunohistochemical, and molecular studies

2009 ◽  
Vol 161 (5) ◽  
pp. 805-810 ◽  
Author(s):  
Anne Rod ◽  
Manuela Voicu ◽  
Laurence Chiche ◽  
Céline Bazille ◽  
Hervé Mittre ◽  
...  
Keyword(s):  
Cushing’S Syndrome ◽  
Malignant Tumors ◽  
Cushing's Syndrome ◽  
Good Prognosis ◽  
University Hospital ◽  
Ectopic Acth Syndrome ◽  
Epithelial Tumor ◽  
Ectopic Acth ◽  
Normal Human

ContextEctopic ACTH syndrome (EAS) is principally associated with aggressive malignant tumors but also with neuroendocrine tumors of good prognosis. Recently, rare nonhepatocytic nested stromal and epithelial tumors (NSET) were characterized by their possible association with Cushing's syndrome of which biochemical and physiopathological features were still incompletely studied.ObjectiveTo describe the clinical and hormonal characteristics of an EAS originating from a liver NSET and further understand the mechanism of cortisol overproduction.Design and settingThis is a clinical case report from the Endocrinology Department of Caen University Hospital, France.Patient and interventionA 17-year-old female patient was found to have a large liver NSET with mild Cushingoid clinical features and intense biological hypercortisolism but moderate ACTH secretion. Resection of the tumor was curative with a 30-month follow-up.ResultsThe epithelial component of the tumor coexpressed ACTH mildly, corticotropin-releasing hormone (CRH) strongly, and 11β-hydroxysteroid dehydrogenase at a level comparable with normal human hepatocytes.ConclusionsLiver NSET is a new cause of EAS, which may evoke hypercortisolism by multiple biochemical pathways.

Comparative assessment of ACTH and lipotropin plasma levels in the diagnosis and follow-up of patients with Cushing's syndrome: A study of 210 cases

1989 ◽  
Vol 86 (6) ◽  
pp. 678-684 ◽  
Author(s):  
Jean Marc Kuhn ◽  
Marie François Proeschel ◽  
Danielle J. Seurin ◽  
Xavier Y. Bertagna ◽  
Jean Pierre Luton ◽  
...  
Keyword(s):  
Cushing’S Syndrome ◽  
Plasma Levels ◽  
Cushing's Syndrome ◽  
Comparative Assessment

Adrenal Incidentaloma and Subclinical Cushing’s Syndrome: A Longitudinal Follow-Up Study by Endoscopic Ultrasound

2015 ◽  
Vol 38 (04) ◽  
pp. 411-419 ◽  
Author(s):  
Maike Collienne ◽  
Nina Timmesfeld ◽  
Simona Bergmann ◽  
Joachim Goebel ◽  
Peter Kann
Keyword(s):  
Growth Rate ◽  
Cushing’S Syndrome ◽  
Endoscopic Ultrasound ◽  
Adrenal Incidentaloma ◽  
Cushing's Syndrome ◽  
Endocrine Function ◽  
Maximum Diameter ◽  
Subclinical Cushing’S Syndrome ◽  
The Mean

Abstract Purpose Adrenal incidentaloma (AI) and adrenal masses in cases of subclinical Cushing’s syndrome (SCS) initially require follow-up imaging. In this study we used endoscopic ultrasound (EUS) as a method for high-resolution imaging. The aim was to evaluate the growth rate of AI and SCS by EUS. Materials and Methods This retrospective analysis included 93 out of 229 patients with AI or SCS who were investigated longitudinally by EUS in our university hospital between 1997 and 2013. The longitudinal follow-up required at least two investigations by EUS and evaluation of endocrine function. Plasma renin, serum aldosterone, 24 h urinary catecholamines and 2 mg dexamethasone suppression test were performed. EUS was performed at baseline and during follow-up. Each time, the maximum diameter was measured. Three groups were defined: non-functioning adenomas (NFA), non-functioning nodular hyperplasias (NFH) and SCS. Results 86 patients had non-functioning masses [NFM] (59 NFA, 48 NFH) and 7 patients had SCS (10 masses). At baseline the mean diameter was 19.4 (± 9.3) mm (NFM) and 19.6 (± 9.2) mm (SCS). The mean follow-up period was 31.6 ± 28.7 months. The estimated mean growth rates per year were low: They were 0.35 mm/yr [NFA], 0.02 mm/yr [NFH] and 0.53 mm/yr [SCS]. Furthermore, there was no malignant progression of any mass. Conclusion The growth rate as determined by EUS was low for all tumor entities observed in this study. There was no difference in tumor growth between the groups.

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