Patient Demographics > Country of Treatment > Belgium

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Karen Decaestecker Department of Diabetology-Endocrinology, AZ Nikolaas, Sint-Niklaas, Belgium

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Veerle Wijtvliet Department of Diabetology-Endocrinology, AZ Nikolaas, Sint-Niklaas, Belgium

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Peter Coremans Department of Diabetology-Endocrinology, AZ Nikolaas, Sint-Niklaas, Belgium

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Nike Van Doninck Department of Diabetology-Endocrinology, AZ Nikolaas, Sint-Niklaas, Belgium

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Summary

ACTH-dependent hypercortisolism is caused by an ectopic ACTH syndrome (EAS) in 20% of cases. We report a rare cause of EAS in a 41-year-old woman, presenting with clinical features of Cushing’s syndrome which developed over several months. Biochemical tests revealed hypokalemic metabolic alkalosis and high morning cortisol and ACTH levels. Further testing, including 24-hour urine analysis, late-night saliva and low-dose dexamethasone suppression test, confirmed hypercortisolism. An MRI of the pituitary gland was normal. Inferior petrosal sinus sampling (IPSS) revealed inconsistent results, with a raised basal gradient but no rise after CRH stimulation. Additional PET-CT showed intense metabolic activity in the left nasal vault. Biopsy of this lesion revealed an unsuspected cause of Cushing’s syndrome: an olfactory neuroblastoma (ONB) with positive immunostaining for ACTH. Our patient underwent transnasal resection of the tumour mass, followed by adjuvant radiotherapy. Normalisation of cortisol and ACTH levels was seen immediately after surgery. Hydrocortisone substitution was started to prevent withdrawal symptoms. As the hypothalamic–pituitary–axis slowly recovered, daily hydrocortisone doses were tapered and stopped 4 months after surgery. Clinical Cushing’s stigmata improved gradually.

Learning points:

  • Ectopic ACTH syndrome can originate from tumours outside the thoracoabdominal region, like the sinonasal cavity.

  • The diagnostic accuracy of IPSS is not 100%: both false positives and false negatives may occur and might be due to a sinonasal tumour with ectopic ACTH secretion.

  • Olfactory neuroblastoma (syn. esthesioneuroblastoma), named because of its sensory (olfactory) and neuroectodermal origin in the upper nasal cavity, is a rare malignant neoplasm. It should not be confused with neuroblastoma, a tumour of the sympathetic nervous system typically occurring in children.

  • If one criticises MRI of the pituitary gland because of ACTH-dependent hypercortisolism, one should take a close look at the sinonasal field as well.

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Elke Thijs Internal Medicine, University Hospital, Ghent, Belgium

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Katrien Wierckx Endocrinology, OLV Hospital, Aalst, Belgium

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Stefaan Vandecasteele Nephrology – Infectious Diseases, A.Z.-Sint Jan, Bruges, Belgium

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Annick Van den Bruel Endocrinology, A.Z.-Sint Jan, Bruges, Belgium

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Summary

A 42-year-old man with complaints of muscle soreness and an increased pigmentation of the skin was referred because of a suspicion of adrenal insufficiency. His adrenocorticotropic hormone and cortisol levels indicated a primary adrenal insufficiency (PAI) and treatment with hydrocortisone and fludrocortisone was initiated. An etiological workup, including an assessment for anti-adrenal antibodies, very long-chain fatty acids, 17-OH progesterone levels and catecholamine secretion, showed no abnormalities. 18Fluorodeoxyglucose positron emission tomography/CT showed bilateral enlargement of the adrenal glands and bilateral presence of an adrenal nodule, with 18fluorodeoxyglucose accumulation. A positive tuberculin test and positive family history of tuberculosis were found, and tuberculostatic drugs were initiated. During the treatment with the tuberculostatic drugs the patient again developed complaints of adrenal insufficiency, due to insufficient dosage of hydrocortisone because of increased metabolism of hydrocortisone.

Learning points:

  • Shrinkage of the adrenal nodules following tuberculostatic treatment supports adrenal tuberculosis being the common aetiology.

  • The tuberculostatic drug rifampicin is a CYP3A4 inducer, increasing the metabolism of hydrocortisone. Increase the hydrocortisone dosage upon initiation of rifampicin in case of (adrenal) tuberculosis.

  • A notification on the Addison’s emergency pass could be considered to heighten physician’s and patients awareness of hydrocortisone drug interactions.

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Charlotte Delcourt Departments of Endocrinology and Nutrition, Université catholique de Louvain, Cliniques Universitaires Saint-Luc, Brussels, Belgium

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Halil Yildiz Internal Medicine, Université catholique de Louvain, Cliniques Universitaires Saint-Luc, Brussels, Belgium

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Alessandra Camboni Pathology, Université catholique de Louvain, Cliniques Universitaires Saint-Luc, Brussels, Belgium

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Eric Van den Neste Hematology, Université catholique de Louvain, Cliniques Universitaires Saint-Luc, Brussels, Belgium

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Véronique Roelants Nuclear Medicine, Université catholique de Louvain, Cliniques Universitaires Saint-Luc, Brussels, Belgium

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Alexandra Kozyreff Ophthalmology, Université catholique de Louvain, Cliniques Universitaires Saint-Luc, Brussels, Belgium

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Jean Paul Thissen Departments of Endocrinology and Nutrition, Université catholique de Louvain, Cliniques Universitaires Saint-Luc, Brussels, Belgium

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Dominique Maiter Departments of Endocrinology and Nutrition, Université catholique de Louvain, Cliniques Universitaires Saint-Luc, Brussels, Belgium

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Raluca Maria Furnica Departments of Endocrinology and Nutrition, Université catholique de Louvain, Cliniques Universitaires Saint-Luc, Brussels, Belgium

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Summary

A 26-year-old woman presented with persistent headache and tiredness. Biological investigations disclosed a moderate inflammatory syndrome, low PTH-hypercalcemia and complete anterior hypopituitarism. A magnetic resonance imaging (MRI) of the pituitary gland was performed and revealed a symmetric enlargement with a heterogeneous signal. Ophthalmological examination showed an asymptomatic bilateral anterior and posterior uveitis, and a diagnosis of pituitary sarcoidosis was suspected. As the localization of lymphadenopathies on the fused whole-body FDG-PET/computerized tomography (CT) was not evoking a sarcoidosis in first instance, an excisional biopsy of a left supraclavicular adenopathy was performed showing classic nodular sclerosis Hodgkin’s lymphoma (HL). A diagnostic transsphenoidal biopsy of the pituitary gland was proposed for accurate staging of the HL and surprisingly revealed typical granulomatous inflammation secondary to sarcoidosis, leading to the diagnosis of a sarcoidosis–lymphoma syndrome. The co-existence of these diseases constitutes a diagnostic challenge and we emphasize the necessity of exact staging of disease in order to prescribe adequate treatment.

Learning points:

  • The possibility of a sarcoidosis–lymphoma syndrome, although rare, should be kept in mind during evaluation for lymphadenopathies.

  • In the case of such association, lymphoma usually occurs after sarcoidosis. However, sarcoidosis and lymphoma can be detected simultaneously and development of sarcoidosis in a patient with previous lymphoma has also been reported.

  • An accurate diagnosis of the disease and the respective organ involvements, including biopsy, is necessary in order to prescribe adequate treatment.

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Raluca Maria Furnica Departments of Endocrinology, Pathology, and Neuroradiology, Université catholique de Louvain, Cliniques Universitaires Saint-Luc, Brussels, Belgium

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Julie Lelotte Departments of Pathology, and Neuroradiology, Université catholique de Louvain, Cliniques Universitaires Saint-Luc, Brussels, Belgium

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Thierry Duprez Departments of Neuroradiology, Université catholique de Louvain, Cliniques Universitaires Saint-Luc, Brussels, Belgium

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Dominique Maiter Departments of Endocrinology, Pathology, and Neuroradiology, Université catholique de Louvain, Cliniques Universitaires Saint-Luc, Brussels, Belgium

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Orsalia Alexopoulou Departments of Endocrinology, Pathology, and Neuroradiology, Université catholique de Louvain, Cliniques Universitaires Saint-Luc, Brussels, Belgium

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Summary

A 26-year-old woman presented with severe postpartum headaches. Magnetic resonance imaging (MRI) revealed a symmetric, heterogeneous enlargement of the pituitary gland. Three months later, she developed central diabetes insipidus. A diagnosis of postpartum hypophysitis was suspected and corticosteroids were prescribed. Six months later, the pituitary mass showed further enlargement and characteristics of a necrotic abscess with a peripheral shell and infiltration of the hypothalamus. Transsphenoidal surgery was performed, disclosing a pus-filled cavity which was drained. No bacterial growth was observed, except a single positive blood culture for Staphylococcus aureus, considered at that time as a potential contaminant. A short antibiotic course was, however, administered together with hormonal substitution for panhypopituitarism. Four months after her discharge, severe headaches recurred. Pituitary MRI was suggestive of a persistent inflammatory mass of the sellar region. She underwent a new transsphenoidal resection of a residual abscess. At that time, the sellar aspiration fluid was positive for Staphylococcus aureus and she was treated with antibiotics for 6 weeks, after which she had complete resolution of her infection. The possibility of a pituitary abscess, although rare, should be kept in mind during evaluation for a necrotic inflammatory pituitary mass with severe headaches and hormonal deficiencies.

Learning points:

  • The possibility of a pituitary abscess, although rare, should be kept in mind during evaluation for a necrotic inflammatory pituitary mass with severe headaches and hormonal deficiencies.

  • In a significant proportion of cases no pathogenic organism can be isolated.

  • A close follow-up is necessary given the risk of recurrence and the high rate of postoperative pituitary deficiencies.

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