Patient Demographics > Country of Treatment > Czech Republic
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Summary
Severe Cushing’s syndrome from an ectopic adrenocorticotropic hormone-producing tumour is rare but often demands rapid diagnostics and treatment of hypercortisolism with its comorbidities. Pharmacotherapy of hypercortisolism by ketoconazole, metyrapone and osilodrostat is currently available. If unsuccessful or insufficient a bilateral adrenalectomy is an option. We present a 28-year-old female with severe Cushing’s syndrome caused by a bronchial metastatic neuroendocrine tumour (NET). Hypercortisolism was efficiently treated by osilodrostat with block–replace and then titration regimen. A once-daily dose was finally used with normalised cortisol levels. Androgen levels measured by liquid chromatography–mass spectrometry were slightly elevated during the treatment but without any symptoms. A simple once-daily use of osilodrostat with titration regimen led to normalised cortisol levels in a severe Cushing’s syndrome patient with an uncurable bronchial NET. Transient hypocortisolism during treatment appeared but was easily treated by hydrocortisone.
Learning points
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Cushing’s syndrome from an ectopic adrenocorticotropic hormone-producing tumour is rare.
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Cortisol upregulation is often severe and rapid, though clinical signs are not always fully pronounced.
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Rapid treatment is a key for preventing and reducing complications such as fractures, thromboembolism, bleeding, hyperglycaemia, and arterial hypertension.
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The novel potent steroidogenesis inhibitor osilodrostat can be used as first-line treatment for reducing hypercortisolism.
Academic Department of Internal Medicine, Charles University in Prague, Faculty of Medicine in Hradec Kralove, Hradec Kralove, Czech Republic
Search for other papers by Eva Krčálová in
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Academic Department of Internal Medicine, Charles University in Prague, Faculty of Medicine in Hradec Kralove, Hradec Kralove, Czech Republic
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Academic Department of Internal Medicine, Charles University in Prague, Faculty of Medicine in Hradec Kralove, Hradec Kralove, Czech Republic
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Academic Department of Internal Medicine, Charles University in Prague, Faculty of Medicine in Hradec Kralove, Hradec Kralove, Czech Republic
Search for other papers by Pavel Žák in
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Summary
Radioiodine (RAI) has played a crucial role in differentiated thyroid cancer treatment for more than 60years. However, the use of RAI administration in patients with papillary thyroid microcarcinoma (even multifocal) is now being widely discussed and often not recommended. In accordance with European consensus, and contrary to the American Thyroid Association (ATA) guidelines, we recently performed RAI thyroid remnant ablation in a patient with differentiated papillary multifocal microcarcinoma. The post-therapeutic whole-body scan and SPECT/CT revealed the real and unexpected extent of disease, with metastases to upper mediastinal lymph nodes. This finding led to the patient’s upstaging from stage I to stage IVa according to the American Joint Committee on Cancer/International Union Against Cancer criteria.
Learning points
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131I is a combined beta–gamma emitter, thus allowing not only residual thyroid tissue ablation but also metastatic tissue imaging.
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RAI remnant ablation omission also means post-treatment whole-body scan omission, which may lead to disease underestimation, due to incorrect nodal and metastatic staging.
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RAI should be considered also in “low-risk” patients, especially when the lymph node involvement is not reliably documented.
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Lower administered RAI activity (30mCi, 1.1GBq) may be a workable compromise in low-risk patients, not indicated for RAI remnant ablation according to ATA guidelines.