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G Leksic Division of Endocrinology and Diabetes, Department of Internal Medicine

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A M Alduk Department of Radiology, University Hospital Centre Zagreb, Zagreb, Croatia

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V Molnar Division of Endocrinology and Diabetes, Department of Internal Medicine

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A Haxhiu Division of Endocrinology and Diabetes, Department of Internal Medicine

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A Balasko Division of Endocrinology and Diabetes, Department of Internal Medicine

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N Knezevic Department of Urology, Department of Internal Medicine, University Hospital Centre Zagreb, School of Medicine, University of Zagreb, Zagreb, Croatia

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T Dusek Division of Endocrinology and Diabetes, Department of Internal Medicine, University Hospital Centre Zagreb, School of Medicine, University of Zagreb, Zagreb, Croatia

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D Kastelan Division of Endocrinology and Diabetes, Department of Internal Medicine, University Hospital Centre Zagreb, School of Medicine, University of Zagreb, Zagreb, Croatia

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Summary

Primary aldosteronism (PA) is characterised by aldosterone hypersecretion and represents a common cause of secondary hypertension. During diagnostic evaluation, it is essential to determine the aetiology of PA since the treatment of unilateral and bilateral disease differs significantly. Adrenal vein sampling (AVS) has been implemented as a gold standard test for the diagnosis of PA subtype. However, due to the AVS complexity, costs and limited availability, many patients with PA are being treated based on the computed tomography (CT) findings. In this article, we present two patients with discrepant CT and AVS results, demonstrating that AVS is the only reliable method for localising the source of aldosterone excess.

Learning points:

  • CT is an unreliable method for distinguishing aldosterone-producing adenoma (APA) from bilateral adrenal hyperplasia (BAH).

  • CT can be misleading in defining lateralisation of the aldosterone excess in case of unilateral disease (APA).

  • AVS is the gold standard test for defining the PA subtype.

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Maria Mercedes Pineyro Clínica de Endocrinología y Metabolismo

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Daiana Arrestia Clínica de Endocrinología y Metabolismo

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Mariana Elhordoy Clínica de Endocrinología y Metabolismo

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Ramiro Lima Neurocirugía, Hospital de Clínicas, Facultad de Medicina, Universidad de la República, Montevideo, Uruguay

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Saul Wajskopf Neurocirugía, Hospital de Clínicas, Facultad de Medicina, Universidad de la República, Montevideo, Uruguay

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Raul Pisabarro Clínica de Endocrinología y Metabolismo

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Maria Pilar Serra Clínica de Endocrinología y Metabolismo

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Summary

Spontaneous reossification of the sellar floor after transsphenoidal surgery has been rarely reported. Strontium ranelate, a divalent strontium salt, has been shown to increase bone formation, increasing osteoblast activity. We describe an unusual case of a young patient with Cushing’s disease who was treated with strontium ranelate for low bone mass who experienced spontaneous sellar reossification after transsphenoidal surgery. A 21-year-old male presented with Cushing’s features. His past medical history included delayed puberty diagnosed at 16 years, treated with testosterone for 3 years without further work-up. He was diagnosed with Cushing’s disease initially treated with transsphenoidal surgery, which was not curative. The patient did not come to follow-up visits for more than 1 year. He was prescribed strontium ranelate 2 g orally once daily for low bone mass by an outside endocrinologist, which he received for more than 1 year. Two years after first surgery he was reevaluated and persisted with active Cushing’s disease. Magnetic resonance image revealed a left 4 mm hypointense mass, with sphenoid sinus occupation by a hyperintense material. At repeated transsphenoidal surgery, sellar bone had a very hard consistency; surgery was complicated and the patient died. Sellar reossification negatively impacted surgery outcomes in this patient. While this entity is possible after transsphenoidal surgery, it remains unclear whether strontium ranelate could have affected sellar ossification.

Learning points:

  • Delayed puberty can be a manifestation of Cushing’s syndrome. A complete history, physical examination and appropriate work-up should be performed before initiating any treatment.

  • Sellar reossification should always be taken into account when considering repeated transsphenoidal surgery. Detailed preoperative evaluation of bony structures by computed tomography ought to be performed in all cases of reoperation.

  • We speculate if strontium ranelate may have affected bone mineralization at the sellar floor. We strongly recommend that indications for prescribing this drug should be carefully followed.

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Ayanthi A Wijewardene Departments of Medicine

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Sarah J Glastras Departments of Endocrinology, Diabetes & Metabolism, Royal North Shore Hospital, Sydney, Australia
Kolling Institute of Medical Research
Sydney Medical School, University of Sydney, Sydney, Australia

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Diana L Learoyd Departments of Endocrinology, Diabetes & Metabolism, Royal North Shore Hospital, Sydney, Australia
Sydney Medical School, University of Sydney, Sydney, Australia

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Bruce G Robinson Departments of Endocrinology, Diabetes & Metabolism, Royal North Shore Hospital, Sydney, Australia
Sydney Medical School, University of Sydney, Sydney, Australia

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Venessa H M Tsang Departments of Endocrinology, Diabetes & Metabolism, Royal North Shore Hospital, Sydney, Australia
Sydney Medical School, University of Sydney, Sydney, Australia

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Summary

Medullary thyroid cancer (MTC) is a rare neuroendocrine tumour that originates from the parafollicular cells of the thyroid gland. The most common presentation of MTC is with a single nodule; however, by the time of diagnosis, most have spread to the surrounding cervical lymph nodes. Cushing’s syndrome is a rare complication of MTC and is due to ectopic adrenocorticotrophic hormone (ACTH) secretion by tumour cells. Cushing’s syndrome presents a challenging diagnostic and management issue in patients with MTC. Tyrosine kinase inhibitors (TKI) previously used for the management of metastatic MTC have become an important therapeutic option for the management of ectopic ACTH in metastatic MTC. The article describes three cases of ectopic ACTH secretion in MTC and addresses the significant diagnostic and management challenges related to Cushing’s syndrome in metastatic MTC.

Learning points:

  • Medullary thyroid cancer (MTC) is a rare neuroendocrine tumour.

  • Cushing’s syndrome is a rare complication of MTC that has a significant impact on patients’ morbidity and mortality.

  • Tyrosine kinase inhibitors (TKI) provide an important therapeutic option for the management of ectopic ACTH in metastatic MTC.

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Omayma Elshafie Department of Medicine, Sultan Qaboos University Hospital, Muscat, Sultanate of Oman

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Yahya Al Badaai Department of Surgery, Sultan Qaboos University Hospital, Muscat, Sultanate of Oman

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Khalifa Alwahaibi Department of Surgery, Sultan Qaboos University Hospital, Muscat, Sultanate of Oman

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Asim Qureshi Department of Pathology, Sultan Qaboos University Hospital, Muscat, Sultanate of Oman

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Samir Hussein Department of Radiology, Sultan Qaboos University Hospital, Muscat, Sultanate of Oman

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Faisal Al Azzri Department of Radiology, Sultan Qaboos University Hospital, Muscat, Sultanate of Oman

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Ali Almamari Department of Medicine, Sultan Qaboos University Hospital, Muscat, Sultanate of Oman

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Nicholas Woodhouse Department of Medicine, Sultan Qaboos University Hospital, Muscat, Sultanate of Oman

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Summary

A 48-year-old hypertensive and diabetic patient presented with a 10-year history of progressive right facial pain, tinnitus, hearing loss, sweating, and palpitations. Investigations revealed a 5.6 cm vascular tumor at the carotid bifurcation. Her blood pressure (BP) was 170/110, on lisinopril 20 mg od and amlodipine 10 mg od and 100 U of insulin daily. A catecholamine-secreting carotid body paraganglioma (CSCBP) was suspected; the diagnosis was confirmed biochemically by determining plasma norepinephrine (NE) level, 89 000 pmol/l, and chromogranin A (CgA) level, 279 μg/l. Meta-iodobenzylguanidine and octreotide scanning confirmed a single tumor in the neck. A week after giving the patient a trial of octreotide 100 μg 8 h, the NE level dropped progressively from 50 000 to 25 000 pmol/l and CgA from 279 to 25 μg/l. Treatment was therefore continued with labetalol 200 mg twice daily (bid) and long-acting octreotide-LA initially using 40 mg/month and later increasing to 80 mg/month. On this dose and with a reduced labetalol intake of 100 mg bid, BP was maintained at 130/70 and her symptoms resolved completely. CgA levels returned to normal in the first week and these were maintained throughout the 3 month treatment period. During tumor resection, there were minimal BP fluctuations during the 10 h procedure. We conclude that short-term high-dose octreotide-LA might prove valuable in the preoperative management of catecholamine-secreting tumors. To the best of our knowledge, this is the first report on the successful use of octreotide in a CSCBP.

Learning points

  • The value of octreotide scanning in the localization of extra-adrenal pheochromocytoma.

  • Control of catecholamine secretion using high-dose octreotide.

  • This is a report of a rare cause of secondary diabetes and hypertension.

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