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Carmina Teresa Fuss Division of Endocrinology and Diabetology, Department of Medicine I, University Hospital Würzburg, Würzburg, Germany

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Stephanie Burger-Stritt Division of Endocrinology and Diabetology, Department of Medicine I, University Hospital Würzburg, Würzburg, Germany

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Silke Horn Division of Endocrinology and Diabetology, Department of Medicine I, University Hospital Würzburg, Würzburg, Germany

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Ann-Cathrin Koschker Division of Endocrinology and Diabetology, Department of Medicine I, University Hospital Würzburg, Würzburg, Germany

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Kathrin Frey Division of Endocrinology and Diabetology, Department of Medicine I, University Hospital Würzburg, Würzburg, Germany

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Almuth Meyer Division of Endocrinology and Diabetology, Department of Internal Medicine, Helios Klinikum Erfurt, Erfurt, Germany

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Stefanie Hahner Division of Endocrinology and Diabetology, Department of Medicine I, University Hospital Würzburg, Würzburg, Germany

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Summary

Standard treatment of hypoparathyroidism consists of supplementation of calcium and vitamin D analogues, which does not fully restore calcium homeostasis. In some patients, hypoparathyroidism is refractory to standard treatment with persistent low serum calcium levels and associated clinical complications. Here, we report on three patients (58-year-old male, 52-year-old female, and 48-year-old female) suffering from severe treatment-refractory postsurgical hypoparathyroidism. Two patients had persistent hypocalcemia despite oral treatment with up to 4 µg calcitriol and up to 4 g calcium per day necessitating additional i.v. administration of calcium gluconate 2–3 times per week, whereas the third patient presented with high frequencies of hypocalcemic and treatment-associated hypercalcemic episodes. S.c. administration of rhPTH (1–34) twice daily (40 µg/day) or rhPTH (1–84) (100 µg/day) only temporarily increased serum calcium levels but did not lead to long-term stabilization. In all three cases, treatment with rhPTH (1–34) as continuous s.c. infusion via insulin pump was initiated. Normalization of serum calcium and serum phosphate levels was observed within 1 week at daily 1–34 parathyroid hormone doses of 15 µg to 29.4 µg. Oral vitamin D and calcium treatment could be stopped or reduced and regular i.v. calcium administration was no more necessary. Ongoing efficacy of this treatment has been documented for up to 7 years so far. Therefore, we conclude that hypoparathyroidism that is refractory to both conventional treatment and s.c. parathyroid hormone (single or twice daily) may be successfully treated with continuous parathyroid hormone administration via insulin pump.

Learning points:

  • Standard treatment of hypoparathyroidism still consists of administration of calcium and active vitamin D.

  • Very few patients with hypoparathyroidism also do not respond sufficiently to standard treatment or administration of s.c. parathyroid hormone once or twice daily.

  • In those cases, continuous s.c. administration of parathyroid hormone via insulin pump may represent a successful treatment alternative.

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Anna Popławska-Kita Departments of Endocrinology, Diabetology and Internal Medicine

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Marta Wielogórska Departments of Endocrinology, Diabetology and Internal Medicine

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Łukasz Poplawski Radiology, Medical University of Bialystok, Bialystok, Poland

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Katarzyna Siewko Departments of Endocrinology, Diabetology and Internal Medicine

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Agnieszka Adamska Departments of Endocrinology, Diabetology and Internal Medicine

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Piotr Szumowski Departments of Nuclear Medicine, Medical University of Bialystok, Bialystok, Poland

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Piotr Myśliwiec 1st Clinic Department of General and Endocrine Surgery, Medical University of Bialystok, Bialystok, Poland

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Janusz Myśliwiec Departments of Nuclear Medicine, Medical University of Bialystok, Bialystok, Poland

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Joanna Reszeć Departments of Medical Pathomorphology, Medical University of Bialystok, Bialystok, Poland

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Grzegorz Kamiński Department of Endocrinology and Radioisotopy Therapy, Military Institute of Medicine, Warsaw, Poland

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Janusz Dzięcioł Departments of Human Anatomy, Medical University of Bialystok, Bialystok, Poland

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Dorota Tobiaszewska Departments of Endocrinology, Diabetology and Internal Medicine

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Małgorzata Szelachowska Departments of Endocrinology, Diabetology and Internal Medicine

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Adam Jacek Krętowski Departments of Endocrinology, Diabetology and Internal Medicine

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Summary

Papillary thyroid gland carcinoma is the most common type of malignancy of the endocrine system. Metastases to the pituitary gland have been described as a complication of papillary thyroid cancer in few reported cases since 1965. We report the case of a 68-year-old female patient with a well-differentiated form of thyroid gland cancer. Despite it being the most common malignant cancer of the endocrine system, with its papillary form being one of the two most frequently diagnosed thyroid cancers, the case we present is extremely rare. Sudden cardiac arrest during ventricular fibrillation occurred during hospitalization. Autopsy of the patient revealed papillary carcinoma of the thyroid, follicular variant, with metastasis to the sella turcica, and concomitant sarcoidosis of heart, lung, and mediastinal and hilar lymph nodes. Not only does atypical metastasis make our patient’s case most remarkable, but also the postmortem diagnosis of sarcoidosis makes her case particularly unusual.

Learning points:

  • The goal of presenting this case is to raise awareness of the clinical heterogeneity of papillary cancer and promote early diagnosis of unexpected metastasis and coexisting diseases to improve clinical outcomes.

  • Clinicians must be skeptical. They should not fall into the trap of diagnostic momentum or accept diagnostic labels at face value. Regardless of the potential mechanisms, clinicians should be aware of the possibility of the coexistence of thyroid cancer and sarcoidosis as a differential diagnosis of lymphadenopathy.

  • This case highlights the importance of the diagnostic and therapeutic planning process and raises awareness of the fact that one uncommon disease could be masked by another extremely rare disorder.

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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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Rayna Patel Department of Stroke Medicine, Addenbrooke’s Hospital, Cambridge, UK

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Waheed Mustafa Department of Orthopaedic Surgery, Basildon University Hospital, Nethermayne, Basildon, Essex, UK

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Michael T Sheaff Department of Pathology, St Bartholemew’s Hospital, London, UK

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Sami Khan Department of Radiology, Basildon University Hospital, Nethermayne, Basildon, Essex, UK

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Summary

IgG4-related disease (IgG4-RD) is a rare but increasingly recognised condition, emerging as a clinical entity following the observation of the associations of autoimmune pancreatitis. IgG4-RD is characterised by extensive infiltration of IgG4-positive plasma cells into multiple organs and raised serum IgG4 levels. Clinical manifestations of IgG4 disease classically include autoimmune pancreatitis, lacrimal or salivary gland infiltration (formerly known as Mikulicz disease) and retroperitoneal fibrosis. More rarely, IgG4 disease can cause pituitary hypophysitis. Although most frequently described in middle-aged males, the epidemiology and pathogenesis of the disease remain largely undefined. Nevertheless, an understanding of the wide variety of clinical manifestations of this multi-system condition is undeniably important given the often excellent outcomes following treatment. We describe an unusual presentation of IgG4 disease with isolated diabetes insipidus secondary to pituitary hypophysitis. The patient in question subsequently developed chest pain secondary to mediastinal lymphadenopathy and tubulo-interstitial nephritis leading to renal dysfunction. He was successfully treated with oral steroids and had regular follow-up, and remains well at follow-up 2 years later.

Learning points

  • IgG4 disease, although rare, is increasing in prevalence largely due to increased recognition of its clinical manifestations, including autoimmune pancreatitis, lacrimal or salivary gland infiltration, retroperitoneal fibrosis and, more rarely, lymphocytic hypophysitis presenting as diabetes insipidus.

  • IgG4 disease is highly treatable, and symptoms may show complete resolution with administration of steroids, highlighting the importance of correct and timely diagnosis.

  • Causes of lymphocytic hypophysitis are varied and not distinguishable radiologically. Given the difficulty in biopsying the pituitary, careful attention must be paid to the systemic clinical presentation to provide clues as to the underlying disorder.

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