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Mike Lin Department of Endocrinology and Diabetes, Royal North Shore Hospital, New South Wales, Australia

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Venessa Tsang Department of Endocrinology and Diabetes, Royal North Shore Hospital, New South Wales, Australia
Cancer Genetics Unit, Kolling Institute of Medical Research, New South Wales, Australia
Faculty of Health and Medicine, University of Sydney, New South Wales, Australia

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Janice Brewer Department of Anatomical Pathology, Royal North Shore Hospital, New South Wales, Australia

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Roderick Clifton-Bligh Department of Endocrinology and Diabetes, Royal North Shore Hospital, New South Wales, Australia
Cancer Genetics Unit, Kolling Institute of Medical Research, New South Wales, Australia
Faculty of Health and Medicine, University of Sydney, New South Wales, Australia

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Matti L Gild Department of Endocrinology and Diabetes, Royal North Shore Hospital, New South Wales, Australia
Cancer Genetics Unit, Kolling Institute of Medical Research, New South Wales, Australia
Faculty of Health and Medicine, University of Sydney, New South Wales, Australia

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Summary

Lymphocytic hypophysitis is a rare neuroendocrine disease characterised by an autoimmune inflammatory disorder of the pituitary gland. We report a 50-year-old woman who presented with headaches and bilateral sixth cranial nerve palsies. MRI of the pituitary revealed extensive fibrosis involving the sellar and extending into both cavernous sinuses causing bilateral occlusion of the internal carotid arteries (ICA). Transphenoidal biopsy confirmed the diagnosis of infiltrative fibrotic lymphocytic hypophysitis. Symptoms resolved with high dose of oral steroids but relapsed on tapering, requiring several treatments of i.v. pulse steroids over 8 months. Rituximab combined with mycophenolate mofetil was required to achieve long-term symptom relief. Serial MRI pituitary imaging showed stabilisation of her disease without reduction in sellar mass or regression of ICA occlusion. The patient’s brain remained perfused solely by her posterior circulation. This case demonstrates an unusual presentation of a rare disease and highlights a successful steroid-sparing regimen in a refractory setting.

Learning points:

  • Lymphocytic hypophysitis is a rare inflammatory disorder of the pituitary gland. In exceptional cases, there is infiltration of the cavernous sinus with subsequent occlusion of the internal carotid arteries.

  • First-line treatment of lymphocytic hypophysitis is high-dose glucocorticoids. Relapse after tapering or discontinuation is common and its use is limited by long-term adverse effects.

  • There is a paucity of data for treatment of refractory lymphocytic hypophysitis. Goals of treatment should include improvement in symptoms, correction of hormonal insufficiencies, reduction in lesion size and prevention of recurrence.

  • Steroid-sparing immunosuppressive drugs such as rituximab and mycophenolate mofetil have been successful in case reports. This therapeutic combination represents a viable alternative treatment for refractory disease.

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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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L I Astaf’eva N.N. Burdenko National Medical Research Centre of Neurosurgery, Moscow, Russian Federation

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Y G Sidneva N.N. Burdenko National Medical Research Centre of Neurosurgery, Moscow, Russian Federation

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B A Kadashev N.N. Burdenko National Medical Research Centre of Neurosurgery, Moscow, Russian Federation

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P L Kalinin N.N. Burdenko National Medical Research Centre of Neurosurgery, Moscow, Russian Federation

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G A Melnichenko National Medical Research Centre of Endocrinology, Moscow, Russian Federation

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S A Agadzhanian Department of English Language for Natural Faculties, Lomonosov Moscow State University, Moscow, Russian Federation

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Summary

A 32-year-old woman presented with primary amenorrhoea, prolactin (PRL) level of 154 150 mIU/L and was diagnosed with a giant pituitary adenoma measuring maximum 6.2 cm. Cabergoline (CAB) treatment at a dose of 0.5 mg/week was prescribed to the patient. The treatment decreased the tumour size after 3 months (MRI scans of the brain) and brought back to normal the level of the PRL (345 mIU/L) after 6 months of CAB treatment. After 7 months of CAB treatment, menarche was achieved, and after 12 months, the patient became pregnant. She discontinued taking CAB at 4-week gestation. The pregnancy resulted in a missed miscarriage at 6–7 weeks; an abortion was conducted by the vacuum aspiration method. The MRI scans of the brain did not show any tumour enlargement. After 18 months from the start of the treatment the patient got pregnant for the second time. At 25-week gestation an MRI scan of the brain was conducted which did not show any increase in the tumour size. At 38 weeks the patient delivered a healthy full-term girl via C-section. The patient chose not to breastfeed and resumed CAB therapy after the delivery. During the treatment, the PRL level returned to the normal range and the menstrual cycle was restored. After 3 years the patient got pregnant for the third time. The patient did not receive CAB during the pregnancies; the examination did not show any tumour enlargement. Further MRI scans did not show any tumour growth. CAB therapy was effective in normalization of the PRL level, tumour shrinkage, menarche and pregnancy-induction which led to the birth of healthy children in a woman with primary amenorrhoea and a giant prolactinoma invading the skull base bones.

Learning points:

  • Giant prolactinomas are very rarely found in women.

  • Cabergoline therapy can be effective in the normalization of the PRL level, tumour shrinkage, menarche induction in a woman with primary amenorrhoea, and giant prolactinoma.

  • Cabergoline therapy can be effective in pregnancy induction which leads to the birth of children in a woman with giant prolactinoma.

  • Cabergoline discontinuation did not trigger tumour enlargement during pregnancy.

Open access
Ellena Cotton Faculty of Biology, Medicine and Health, University of Manchester, Manchester Academic Health Sciences Centre, Manchester, UK
Specialist Medicine, Manchester University Foundation Trust, Manchester, UK

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David Ray Oxford Centre for Diabetes, Endocrinology and Metabolism, University of Oxford, Oxford, UK

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Summary

A young woman carrying germline DICER1 mutation was discovered to have a pituitary microprolactinoma when she became amenorrhoic. The mutation was identified as a result of family screening following the early death of the patient’s daughter with ovarian cancer. The patient was in follow-up screening for thyroid disease, and investigations were initiated when she became amenorrhoic. MR scan revealed a 6 mm diameter pituitary microadenoma and raised prolactin. The prolactin was efficiently suppressed with low-dose cabergoline, and her menstrual cycles resumed. Dicer is an RNase enzyme, which is essential for processing small non-coding RNAs. These molecules play pleiotropic roles in regulating gene expression, by targeting mRNA sequences for degradation. DICER1 plays different roles depending on cell context, but is thought to be a functional tumour suppressor gene. Accordingly, germline mutation in one DICER1 allele is insufficient for oncogenesis, and a second hit on the other allele is required, as a result of postnatal somatic mutation. Loss of DICER1 is linked to multiple tumours, with prominent endocrine representation. Multinodular goitre is frequent, with increased risk of differentiated thyroid cancer. Rare, developmental pituitary tumours are reported, including pituitary blastoma, but not reports of functional pituitary adenomas. As DICER1 mutations are rare, case reports are the only means to identify new manifestations and to inform appropriate screening protocols.

Learning points:

  • DICER1 mutations lead to endocrine tumours.

  • DICER1 is required for small non-coding RNA expression.

  • DICER1 carriage and microprolactinoma are both rare, but here are reported in the same individual, suggesting association.

  • Endocrine follow-up of patients carrying DICER1 mutations should consider pituitary disease.

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Benedetta Zampetti Endocrinology Unit, Niguarda Hospital, Milan, Italy

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Giorgia Simonetti Neurooncology Unit, Fondazione IRCCS Neurological Institute Carlo Besta, Milan, Italy

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Roberto Attanasio Endocrinology Service, Galeazzi Institute IRCCS, Milan, Italy

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Antonio Silvani Neurooncology Unit, Fondazione IRCCS Neurological Institute Carlo Besta, Milan, Italy

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Renato Cozzi Endocrinology Unit, Niguarda Hospital, Milan, Italy

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Summary

We describe the 20-year course of a 63-year-old male with a macroprolactinoma that acquired resistance to treatment and aggressive behavior after a 4-year successful treatment with cabergoline. He was submitted to multiple surgical resections by a skilled surgeon, fractionated radiotherapy and was eventually treated with temozolomide. After a first 6-month standard cycle, a relapse occurred and he was treated again successfully.

Learning points:

  • Prolactinomas are the most frequent type of pituitary adenoma.

  • They usually have a benign course.

  • In most cases dopamine-agonist drugs, mainly cabergoline, are first-line (and usually only) treatment.

  • Occasionally prolactinomas can have or acquire resistance to treatment and/or aggressive behavior.

  • Temozolomide (TMZ), an oral alkylating drug, can be effective in such aggressive tumors.

  • Multimodal treatment (surgery, radiation, cabergoline and TMZ) is warranted in aggressive pituitary tumors.

  • We describe here successful rechallenge with TMZ after relapse occurring 18 months after a first TMZ cycle.

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Yew Wen Yap Leighton Hospital, Crewe, Crewe, UK

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Steve Ball Manchester University NHS Foundation Trust, Manchester, UK

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Zubair Qureshi Leighton Hospital, Crewe, Crewe, UK

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Summary

The coexistence of primary hypothyroidism and thyroid-stimulating hormone (TSH)-stimulating pituitary macroadenoma can be a rare occurrence and can make diagnosis very challenging. We describe a case of a 44-year-old female with a history of fatigue, poor concentration, weight gain and amenorrhoea together with biochemical evidence of primary autoimmune hypothyroidism. Her initial TSH levels were elevated with low normal free thyroxine (T4) levels. Levothyroxine treatment was initiated and the dose was gradually titrated to supraphysiologic doses. This led to the normalisation of her TSH levels but her free T4 and triiodothyronine (T3) levels remained persistently elevated. This prompted a serum prolactin check which returned elevated at 2495 µ/L, leading onto pituitary imaging. A MRI of the pituitary gland revealed a pituitary macroadenoma measuring 2.4 × 2 × 1.6 cm. Despite starting her on cabergoline therapy with a reduction in her prolactin levels, her TSH levels began to rise even further. Additional thyroid assays revealed that she had an abnormally elevated alpha subunit at 3.95 (age-related reference range <3.00). This corresponded to a thyroid-secreting hormone pituitary macroadenoma. She went on to have a transphenoidal hypophysectomy. Histology revealed tissues staining for TSH, confirming this to be a TSH-secreting pituitary macroadenoma. This case highlighted the importance of further investigations with thyroid assay interferences, heterophile antibodies, alpha subunit testing and anterior pituitary profile in cases of resistant and non-resolving primary hypothyroidism.

Learning points:

  • Levothyroxine treatment in primary hypothyroidism can potentially unmask the presence of a latent TSH-secreting pituitary macroadenoma, which can make diagnosis very challenging.

  • A high index of suspicion should prompt clinicians to further investigate cases of primary hypothyroidism which despite increasing doses of levothyroxine treatment with normalisation of TSH, the free T4 and T3 levels remain persistently elevated.

  • Clinicians should consider investigating for adherence to levothyroxine, thyroid assay interference, heterophile antibodies, TSH dilution studies, alpha subunit and anterior pituitary profile testing to further clarity the diagnosis in these patients.

  • Although coexistent cases of TSHoma with primary hypothyroidism are rare, it should always be in the list of differential diagnoses in cases of unresolving primary hypothyroidism.

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Michelle Maher Endocrinology, Imperial College Healthcare NHS Trust, London, UK
National University of Ireland, Galway, Ireland

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Federico Roncaroli University of Manchester, Manchester, UK

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Nigel Mendoza Endocrinology, Imperial College Healthcare NHS Trust, London, UK

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Karim Meeran Endocrinology, Imperial College Healthcare NHS Trust, London, UK

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Natalie Canham Liverpool Womens NHS Foundation Trust, Liverpool, UK

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Monika Kosicka-Slawinska London North West Healthcare NHS Trust, London, UK

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Birgitta Bernhard London North West Healthcare NHS Trust, London, UK

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David Collier The William Harvey Research Institute, Barts and The London School of Medicine and Dentistry, Queen Mary University of London, London, UK

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Juliana Drummond The William Harvey Research Institute, Barts and The London School of Medicine and Dentistry, Queen Mary University of London, London, UK

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Kassiani Skordilis University Hospitals Birmingham NHS Foundation Trust, Mindelsohn Way, Edgbaston, Birmingham, UK

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Nicola Tufton The Royal London Hospital, Barts Health NHS Trust, London UK

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Anastasia Gontsarova Endocrinology, Imperial College Healthcare NHS Trust, London, UK

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Niamh Martin Endocrinology, Imperial College Healthcare NHS Trust, London, UK

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Márta Korbonits The William Harvey Research Institute, Barts and The London School of Medicine and Dentistry, Queen Mary University of London, London, UK

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Florian Wernig Endocrinology, Imperial College Healthcare NHS Trust, London, UK

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Summary

Symptomatic pituitary adenomas occur with a prevalence of approximately 0.1% in the general population. It is estimated that 5% of pituitary adenomas occur in a familial setting, either in isolated or syndromic form. Recently, loss-of-function mutations in genes encoding succinate dehydrogenase subunits (SDHx) or MYC-associated factor X (MAX) have been found to predispose to pituitary adenomas in co-existence with paragangliomas or phaeochromocytomas. It is rare, however, for a familial SDHx mutation to manifest as an isolated pituitary adenoma. We present the case of a pituitary lactotroph adenoma in a patient with a heterozygous germline SDHB mutation, in the absence of concomitant neoplasms. Initially, the adenoma showed biochemical response but poor tumour shrinkage in response to cabergoline; therefore, transsphenoidal surgery was performed. Following initial clinical improvement, tumour recurrence was identified 15 months later. Interestingly, re-initiation of cabergoline proved successful and the lesion demonstrated both biochemical response and tumour shrinkage. Our patient’s SDHB mutation was identified when we realised that her father had a metastatic paraganglioma, prompting genetic testing. Re-inspection of the histopathological report of the prolactinoma confirmed cells with vacuolated cytoplasm. This histological feature is suggestive of an SDHx mutation and should prompt further screening for mutations by immunohistochemistry and/or genetic testing. Surprisingly, immunohistochemistry of this pituitary adenoma demonstrated normal SDHB expression, despite loss of SDHB expression in the patient’s father’s paraganglioma.

Learning points:

  • Pituitary adenomas may be the presenting and/or sole feature of SDHB mutation-related disease.

  • SDHx mutated pituitary adenomas may display clinically aggressive behaviour and demonstrate variable response to medical treatment.

  • Histological evidence of intracytoplasmic vacuoles in a pituitary adenoma might suggest an SDH-deficient tumour and should prompt further screening for SDHx mutations.

  • Immunohistochemistry may not always predict the presence of SDHx mutations.

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Ana G Ferreira Endocrinology and Diabetes Department, Garcia de Orta Hospital, Almada, Portugal

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Tiago N Silva Endocrinology and Diabetes Department, Garcia de Orta Hospital, Almada, Portugal

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Henrique V Luiz Endocrinology and Diabetes Department, Garcia de Orta Hospital, Almada, Portugal

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Filipa D Campos Hemato-Oncology Department, Garcia de Orta Hospital, Almada, Portugal

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Maria C Cordeiro Endocrinology and Diabetes Department, Garcia de Orta Hospital, Almada, Portugal

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Jorge R Portugal Endocrinology and Diabetes Department, Garcia de Orta Hospital, Almada, Portugal

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Sellar plasmacytomas are rare and the differential diagnosis with non-functioning pituitary adenomas might be difficult because of clinical and radiological resemblance. They usually present with neurological signs and intact anterior pituitary function. Some may already have or eventually progress to multiple myeloma. We describe a case associated with extensive anterior pituitary involvement, which is a rare form of presentation. A 68-year-old man was referred to our Endocrinology outpatient clinic due to gynecomastia, reduced libido and sexual impotence. Physical examination, breast ultrasound and mammography confirmed bilateral gynecomastia. Blood tests revealed slight hyperprolactinemia, low testosterone levels, low cortisol levels and central hypothyroidism. Sellar MRI showed a heterogeneous sellar mass (56 × 60 × 61 mm), initially suspected as an invasive macroadenoma. After correcting the pituitary deficits with hydrocortisone and levothyroxine, the patient underwent transsphenoidal surgery. Histological examination revealed a plasmacytoma and multiple myeloma was ruled out. The patient was unsuccessfully treated with radiation therapy (no tumor shrinkage). Myeloma ultimately developed, with several other similar lesions in different locations. The patient was started on chemotherapy, had a bone marrow transplant and is now stable (progression free) on lenalidomide and dexamethasone. The presenting symptoms and panhypopituitarism persisted, requiring chronic replacement treatment with levothyroxine, hydrocortisone and testosterone.

Learning points:

  • Plasmacytomas, although rare, are a possible type of sellar masses, which have a completely different treatment approach, so it is important to make the correct diagnosis.

  • Usually, they present with neurological signs and symptoms and a well-preserved pituitary function, but our case shows that anterior pituitary function can be severely compromised.

  • Making a more extensive evaluation (clinical and biochemical) might provide some clues to this diagnosis.

Open access
Oscar D Bruno Division of Endocrinology
Fundacion de Endocrinologia

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Ricardo Fernández Pisani Division of Neurosurgery, Hospital de Clínicas, Universidad de Buenos Aires, Buenos Aires, Argentina

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Gabriel Isaac Division of Endocrinology

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Armando Basso Division of Neurosurgery, Hospital de Clínicas, Universidad de Buenos Aires, Buenos Aires, Argentina

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Summary

The role of mechanical forces influencing the growth of a pituitary adenoma is poorly understood. In this paper we report the case of a young man with hyperprolactinaemia and an empty sella secondary to hydrocephalia, who developed a macroprolactinoma following the relief of high intraventricular pressure.

Learning points:

  • The volume of a pituitary tumour may be influenced not only by molecular but also by local mechanical factors.

  • Intratumoural pressure, resistance of the sellar diaphragm and intracranial liquid pressure may play a role in the final size of a pituitary adenoma.

  • The presence of hydrocephalus may hide a pituitary macroadenoma.

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Ekaterina Manuylova Department of Endocrinology, Diabetes and Metabolism

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Laura M Calvi Department of Endocrinology, Diabetes and Metabolism

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Catherine Hastings Department of Neurosurgery

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G Edward Vates Department of Neurosurgery

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Mahlon D Johnson Department of Pathology, University of Rochester, Rochester, New York, USA

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William T Cave Jr Department of Endocrinology, Diabetes and Metabolism

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Ismat Shafiq Department of Endocrinology, Diabetes and Metabolism

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Summary

Co-secretion of growth hormone (GH) and prolactin (PRL) from a single pituitary adenoma is common. In fact, up to 25% of patients with acromegaly may have PRL co-secretion. The prevalence of acromegaly among patients with a newly diagnosed prolactinoma is unknown. Given the possibility of mixed GH and PRL co-secretion, the current recommendation is to obtain an insulin-like growth factor-1 (IGF-1) in patients with prolactinoma at the initial diagnosis. Long-term follow-up of IGF-1 is not routinely done. Here, we report two cases of well-controlled prolactinoma on dopamine agonists with the development of acromegaly 10–20 years after the initial diagnoses. In both patients, a mixed PRL/GH-cosecreting adenoma was confirmed on the pathology examination after transsphenoidal surgery (TSS). Therefore, periodic routine measurements of IGF-1 should be considered regardless of the duration and biochemical control of prolactinoma.

Learning points:

  • Acromegaly can develop in patients with well-controlled prolactinoma on dopamine agonists.

  • The interval between prolactinoma and acromegaly diagnoses can be several decades.

  • Periodic screening of patients with prolactinoma for growth hormone excess should be considered and can 
lead to an early diagnosis of acromegaly before the development of complications.

Open access