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Tzy Harn Chua Department of Endocrinology, Changi General Hospital, Singapore

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Wann Jia Loh Department of Endocrinology, Changi General Hospital, Singapore

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Summary

Severe hyponatremia and osmotic demyelination syndrome (ODS) are opposite ends of a spectrum of emergency disorders related to sodium concentrations. Management of severe hyponatremia is challenging because of the difficulty in balancing the risk of overcorrection leading to ODS as well as under-correction causing cerebral oedema, particularly in a patient with chronic hypocortisolism and hypothyroidism. We report a case of a patient with Noonan syndrome and untreated anterior hypopituitarism who presented with symptomatic hyponatremia and developed transient ODS.

Learning points:

  • Patients with severe anterior hypopituitarism with severe hyponatremia are susceptible to the rapid rise of sodium level with a small amount of fluid and hydrocortisone.

  • These patients with chronic anterior hypopituitarism are at high risk of developing ODS and therefore, care should be taken to avoid a rise of more than 4–6 mmol/L per day.

  • Early recognition and rescue desmopressin and i.v. dextrose 5% fluids to reduce serum sodium concentration may be helpful in treating acute ODS.

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Mariana Barbosa Department of Endocrinology, Hospital de Braga, Braga, Portugal

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Sílvia Paredes Department of Endocrinology, Hospital de Braga, Braga, Portugal

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Maria João Machado Department of Neurosurgery, Hospital de Braga, Braga, Portugal

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Rui Almeida Department of Neurosurgery, Hospital de Braga, Braga, Portugal
Pituitary Consult, Hospital de Braga, Braga, Portugal

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Olinda Marques Department of Endocrinology, Hospital de Braga, Braga, Portugal
Pituitary Consult, Hospital de Braga, Braga, Portugal

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Summary

Gonadotropin-releasing hormone (GnRH) agonists, currently used in the treatment of advanced prostate cancer, have been described as a rare cause of pituitary apoplexy, a potentially life-threatening clinical condition. We report the case of a 69-year-old man with a known pituitary macroadenoma who was diagnosed with prostate cancer and started treatment with GnRH agonist leuprorelin (other hormones were not tested before treatment). Few minutes after drug administration, the patient presented with acute-onset severe headache, followed by left eye ptosis, diplopia and vomiting. Pituitary MRI revealed tumor enlargement and T1-hyperintense signal, compatible with recent bleeding sellar content. Laboratory endocrine workup was significant for low total testosterone. The patient was managed conservatively with high-dose steroids, and symptoms significantly improved. This case describes a rare phenomenon, pituitary apoplexy induced by GnRH agonist. We review the literature regarding this condition: the pathophysiological mechanism involved is not clearly established and several hypotheses have been proposed. Although uncommon, healthcare professionals and patients should be aware of this complication and recognize the signs, preventing a delay in diagnosis and treatment.

Learning points:

  • Pituitary apoplexy (PA) is a potentially life-threatening complication that can be caused by gonadotropin-releasing hormone agonist (GnRHa) administration for the treatment of advanced prostate cancer.

  • This complication is rare but should be taken into account when using GnRHa, particularly in the setting of a known pre-existing pituitary adenoma.

  • PA presents with classic clinical signs and symptoms that should be promptly recognized.

  • Patients should be instructed to seek medical care if suspicious symptoms occur.

  • Healthcare professionals should be aware of this complication, enabling its early recognition, adequate treatment and favorable outcome.

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Diana Catarino Endocrinology, Diabetes and Metabolism Department, Centro Hospitalar e Universitário de Coimbra EPE, Coimbra, Portugal

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Cristina Ribeiro Endocrinology, Diabetes and Metabolism Department, Centro Hospitalar e Universitário de Coimbra EPE, Coimbra, Portugal

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Leonor Gomes Endocrinology, Diabetes and Metabolism Department, Centro Hospitalar e Universitário de Coimbra EPE, Coimbra, Portugal

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Isabel Paiva Endocrinology, Diabetes and Metabolism Department, Centro Hospitalar e Universitário de Coimbra EPE, Coimbra, Portugal

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Summary

Pituitary infections, particularly with fungus, are rare disorders that usually occur in immunocompromised patients. Cushing’s syndrome predisposes patients to infectious diseases due to their immunosuppression status. We report the case of a 55-year-old woman, working as a poultry farmer, who developed intense headache, palpebral ptosis, anisocoria, prostration and psychomotor agitation 9 months after initial diabetes mellitus diagnosis. Cranioencephalic CT scan showed a pituitary lesion with bleeding, suggesting pituitary apoplexy. Patient underwent transsphenoidal surgery and the neuropathologic study indicated a corticotroph adenoma with apoplexy and fungal infection. Patient had no preoperative Cushing’s syndrome diagnosis. She was evaluated by a multidisciplinary team who decided not to administer anti-fungal treatment. The reported case shows a rare association between a corticotroph adenoma and a pituitary fungal infection. The possible contributing factors were hypercortisolism, uncontrolled diabetes and professional activity. Transsphenoidal surgery is advocated in these infections; however, anti-fungal therapy is still controversial.

Learning points:

  • Pituitary infections are rare disorders caused by bacterial, viral, fungal and parasitic infections.

  • Pituitary fungal infections usually occur in immunocompromised patients.

  • Cushing’s syndrome, as immunosuppression factor, predisposes patients to infectious diseases, including fungal infections.

  • Diagnosis of pituitary fungal infection is often achieved during histopathological investigation.

  • Treatment with systemic anti-fungal drugs is controversial.

  • Endocrine evaluation is recommended at the time of initial presentation of pituitary manifestations.

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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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Shinichiro Teramoto Department of Neurosurgery, Juntendo University School of Medicine, Tokyo, Japan

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Yuichi Tange Department of Neurosurgery, Juntendo University School of Medicine, Tokyo, Japan

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Hisato Ishii Department of Neurosurgery, Juntendo University Urayasu Hospital, Chiba, Japan

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Hiromasa Goto Department of Metabolism and Endocrinology, Juntendo University School of Medicine, Tokyo, Japan

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Ikuko Ogino Department of Neurosurgery, Juntendo University School of Medicine, Tokyo, Japan

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Hajime Arai Department of Neurosurgery, Juntendo University School of Medicine, Tokyo, Japan

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Summary

A 67-year-old woman with a past history of type 2 diabetes mellitus presented with worsening glycemic control. She had some acromegaly symptoms and magnetic resonance imaging demonstrated a pituitary tumor. Endocrinological examination found the resting growth hormone (GH) level within the normal range, but elevated insulin-like growth factor 1 level. A 75 g oral glucose tolerance test showed inadequate suppression of nadir GH levels. Acromegaly due to GH-secreting pituitary tumor was diagnosed. The patient underwent endoscopic transsphenoidal surgery resulting in gross total removal of the tumor and recovered well postoperatively. Histological examination of the tumor showed coexistence of relatively large gangliocytoma cells and pituitary adenoma cells, suggesting mixed gangliocytoma-pituitary adenoma. In addition, colocalization of GH and GH-releasing hormone (GHRH) in pituitary adenoma cells was revealed, so the adenomatous components were more likely to produce GHRH in our mixed gangliocytoma-pituitary adenoma case. Mixed gangliocytoma-pituitary adenoma is very rare, and the present unique case demonstrated only the adenomatous components associated with GHRH production.

Learning points:

  • Sellar gangliocytoma coexisting with pituitary adenoma is recognized as a mixed gangliocytoma-pituitary adenoma and is very rare.

  • A proposed developmental mechanism of growth hormone (GH)-secreting mixed gangliocytoma-pituitary adenoma involves GH-releasing hormone (GHRH) produced by the gangliocytic components promoting the growth of tumor including GH-secreting adenomatous components.

  • Since our present case indicated that the adenomatous components of mixed gangliocytoma-pituitary adenoma could secrete both GH and GHRH simultaneously, progression of GH-secreting mixed gangliocytoma and pituitary adenoma may involve exposure to spontaneously produced GHRH due to the adenomatous components.

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Hui Yi Ng Department of Clinical Medicine, Level 4, Macquarie University, 2 Technology Place, Macquarie University, New South Wales, Australia

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Divya Namboodiri Department of Clinical Medicine, Level 4, Macquarie University, 2 Technology Place, Macquarie University, New South Wales, Australia

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Diana Learoyd University of Sydney, Faculty of Medicine and Health, Northern Clinical School, Reserve Road St Leonards, New South Wales, Australia

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Andrew Davidson Department of Neurosurgery, Level 2, Macquarie University, 2 Technology Place Macquarie University, New South Wales, Australia

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Bernard Champion Department of Clinical Medicine, Level 4, Macquarie University, 2 Technology Place, Macquarie University, New South Wales, Australia

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Veronica Preda Department of Clinical Medicine, Level 4, Macquarie University, 2 Technology Place, Macquarie University, New South Wales, Australia

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Summary

Co-secreting thyrotropin/growth hormone (GH) pituitary adenomas are rare; their clinical presentation and long-term management are challenging. There is also a paucity of long-term data. Due to the cell of origin, these can behave as aggressive tumours. We report a case of a pituitary plurihormonal pit-1-derived macroadenoma, with overt clinical hyperthyroidism and minimal GH excess symptoms. The diagnosis was confirmed by pathology showing elevated thyroid and GH axes with failure of physiological GH suppression, elevated pituitary glycoprotein hormone alpha subunit (αGSU) and macroadenoma on imaging. Pre-operatively the patient was rendered euthyroid with carbimazole and underwent successful transphenoidal adenomectomy (TSA) with surgical cure. Histopathology displayed an elevated Ki-67 of 5.2%, necessitating long-term follow-up.

Learning points:

  • Thyrotropinomas are rare and likely under-diagnosed due to under-recognition of secondary hyperthyroidism.

  • Thyrotropinomas and other plurihormonal pit-1-derived adenomas are more aggressive adenomas according to WHO guidelines.

  • Co-secretion occurs in 30% of thyrotropinomas, requiring diligent investigation and long-term follow-up of complications.

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Stephanie Wei Ping Wong Department of Diabetes and Endocrinology, St Helens and Knowsley Teaching Hospitals NHS Trust, Prescot, UK

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Yew Wen Yap Department of Diabetes and Endocrinology, St Helens and Knowsley Teaching Hospitals NHS Trust, Prescot, UK

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Ram Prakash Narayanan Department of Diabetes and Endocrinology, St Helens and Knowsley Teaching Hospitals NHS Trust, Prescot, UK

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Mohammad Al-Jubouri Department of Biochemistry, St Helens and Knowsley Teaching Hospitals NHS Trust, Prescot, UK

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Ashley Grossman Centre for Endocrinology, William Harvey Research Institute, Barts and London School of Medicine and Dentistry, Queen Mary University of London, London, UK

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Christina Daousi Department of Diabetes and Endocrinology, Aintree University Hospital NHS Foundation Trust, Liverpool, UK

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Yahya Mahgoub Department of Diabetes and Endocrinology, St Helens and Knowsley Teaching Hospitals NHS Trust, Prescot, UK

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Summary

We report our experience on managing a case of florid Cushing’s disease with Methicillin-resistant Staphylococcus aureus (MRSA) sepsis using intravenous etomidate in the intensive care unit of a UK district general hospital.

Learning points:

  • Severe Cushing’s syndrome is associated with high morbidity and mortality.

  • Etomidate is a safe and effective medical therapy to rapidly lower cortisol levels even in the context of severe sepsis and immunosuppression.

  • Etomidate should ideally be administered in an intensive care unit but is still feasible in a district general hospital.

  • During treatment with etomidate, accumulation of serum 11β-deoxycortisol (11DOC) levels can cross-react with laboratory cortisol measurement leading to falsely elevated serum cortisol levels. For this reason, serum cortisol measurement using a mass spectrometry assay should ideally be used to guide etomidate prescription.

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E Sanz-Sapera Endocrinology, Vall d’Hebron Hospital, Barcelona, Spain

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S Sarria-Estrada Radiology, Vall d’Hebron Hospital, Barcelona, Spain

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F Arikan Neurosurgery, Vall d’Hebron Hospital, Barcelona, Spain

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B Biagetti Endocrinology, Vall d’Hebron Hospital, Barcelona, Spain

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Summary

Pituitary apoplexy is a rare but potentially life-threatening clinical syndrome characterised by ischaemic infarction or haemorrhage into a pituitary tumour that can lead to spontaneous remission of hormonal hypersecretion. We report the case of a 50-year-old man who attended the emergency department for sudden onset of headache. A computed tomography (CT) scan at admission revealed pituitary haemorrhage and the blood test confirmed the clinical suspicion of acromegaly and an associated hypopituitarism. The T1-weighted magnetic resonance imaging (MRI) showed the classic pituitary ring sign on the right side of the pituitary. Following admission, he developed acute-onset hyponatraemia that required hypertonic saline administration, improving progressively. Surprisingly, during the follow-up, IGF1 levels became normal and he progressively recovered pituitary function.

Learning points:

  • Patients with pituitary apoplexy may have spontaneous remission of hormonal hypersecretion. If it is not an emergency, we should delay a decision to undertake surgery following apoplexy and re-evaluate hormone secretion.

  • Hyponatraemia is an acute sign of hypocortisolism in pituitary apoplexy. However, SIADH although uncommon, could appear later as a consequence of direct hypothalamic insult and requires active and individualised treatment. For this reason, closely monitoring sodium at the beginning of the episode and throughout the first week is advisable to guard against SIADH.

  • Despite being less frequent, if pituitary apoplexy is limited to the tumour, the patient can recover pituitary function previously damaged by the undiagnosed macroadenoma.

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Anne Marie Hannon Departments of Endocrinology and Diabetes, Cork University Hospital, Cork, Ireland

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Isolda Frizelle Departments of Endocrinology and Diabetes, Cork University Hospital, Cork, Ireland

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George Kaar Departments of Neurosurgery, Cork University Hospital, Cork, Ireland

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Steven J Hunter Department of Endocrinology and Diabetes, Royal Victoria Hospital, Belfast, UK

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Mark Sherlock Department of Endocrinology and Diabetes, Beaumont Hospital, Dublin, Ireland

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Christopher J Thompson Department of Endocrinology and Diabetes, Beaumont Hospital, Dublin, Ireland

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Domhnall J O’Halloran Departments of Endocrinology and Diabetes, Cork University Hospital, Cork, Ireland

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the Irish Pituitary Database Group
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Summary

Pregnancy in acromegaly is rare and generally safe, but tumour expansion may occur. Managing tumour expansion during pregnancy is complex, due to the potential complications of surgery and side effects of anti-tumoural medication. A 32-year-old woman was diagnosed with acromegaly at 11-week gestation. She had a large macroadenoma invading the suprasellar cistern. She developed bitemporal hemianopia at 20-week gestation. She declined surgery and was commenced on 100 µg subcutaneous octreotide tds, with normalisation of her visual fields after 2 weeks of therapy. She had a further deterioration in her visual fields at 24-week gestation, which responded to an increase in subcutaneous octreotide to 150 µg tds. Her vision remained stable for the remainder of the pregnancy. She was diagnosed with gestational diabetes at 14/40 and was commenced on basal bolus insulin regimen at 22/40 gestation. She otherwise had no obstetric complications. Foetal growth continued along the 50th centile throughout pregnancy. She underwent an elective caesarean section at 34/40, foetal weight was 3.2 kg at birth with an APGAR score of 9. The neonate was examined by an experienced neonatologist and there were no congenital abnormalities identified. She opted not to breastfeed and she is menstruating regularly post-partum. She was commenced on octreotide LAR 40 mg and referred for surgery. At last follow-up, 2 years post-partum, the infant has been developing normally. In conclusion, our case describes a first presentation of acromegaly in pregnancy and rescue of visual field loss with somatostatin analogue therapy.

Learning points:

  • Tumour expansion may occur in acromegaly during pregnancy.

  • Treatment options for tumour expansion in pregnancy include both medical and surgical options.

  • Somatostatin analogues may be a viable medical alternative to surgery in patients with tumour expansion during pregnancy.

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Yoko Olmedilla Endocrinology and Nutrition Service, Gregorio Marañón General Universitary Hospital, Madrid, Spain

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Shoaib Khan Oxford Centre for Endocrinology, Diabetes and Metabolism, Churchill Hospital, Oxford, UK

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Victoria Young Departments of Neuroradiology, John Radcliffe Hospital, Oxford, UK

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Robin Joseph Departments of Neuroradiology, John Radcliffe Hospital, Oxford, UK

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Simon Cudlip Departments of Neurosurgery, John Radcliffe Hospital, Oxford, UK

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Olaf Ansgorge Departments of Neuropathology, John Radcliffe Hospital, Oxford, UK

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Ashley Grossman Oxford Centre for Endocrinology, Diabetes and Metabolism, Churchill Hospital, Oxford, UK

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Aparna Pal Oxford Centre for Endocrinology, Diabetes and Metabolism, Churchill Hospital, Oxford, UK

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Summary

A 21 year-old woman was found to have a pituitary macroadenoma following an episode of haemophilus meningitis. Biochemical TSH and GH excess was noted, although with no clear clinical correlates. She was treated with a somatostatin analogue (SSA), which restored the euthyroid state and controlled GH hypersecretion, but she re-presented with a further episode of cerebrospinal fluid (CSF) leak and recurrent meningitis. Histology following transsphenoidal adenomectomy revealed a Pit-1 lineage plurihormonal adenoma expressing GH, TSH and PRL. Such plurihormonal pituitary tumours are uncommon and even more unusual to present with spontaneous bacterial meningitis. The second episode of CSF leak and meningitis appears to have been due to SSA therapy-induced tumour shrinkage, which is not a well-described phenomenon in the literature for this type of tumour.

Learning points:

  • Pit-1 lineage GH/TSH/PRL-expressing plurihormonal pituitary adenomas are uncommon. Moreover, this case is unique as the patient first presented with bacterial meningitis.

  • Inmunohistochemical plurihormonality of pituitary adenomas does not necessarily correlate with biochemical and clinical features of hormonal hypersecretion.

  • Given that plurihormonal Pit-1 lineage adenomas may behave more aggressively than classical pituitary adenomas, accurate pathological characterization of these tumours has an increasing prognostic relevance.

  • Although unusual, a CSF leak and meningitis may be precipitated by SSA therapy of a pituitary macroadenoma via tumour shrinkage.