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  • Plurihormonal pituitary adenoma x
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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.

Taxonomy:
Clinical Overview, Gland/Organ, Pituitary, Topic, Pituitary, Hormone, GH, IGF1, Prolactin, Thyroxine (T4), Triiodothyronine (T3), TSH, Condition/ Syndrome, Acromegaly, Goitre (multinodular), Hyperthyroidism, Hypogonadism, Pituitary adenoma, Plurihormonal pituitary adenoma, Thyrotrophic adenoma, Patient Demographics, Age, Adult, Gender, Male, Ethnicity, White, Country of Treatment, Australia, Diagnosis and Treatment, Signs and Symptoms, Anxiety, Dizziness, Goitre, Headache, Heat intolerance, Hypertension, Hyperthyroidism, Hypogonadism, Insomnia, Osteopenia, Palpitations, Tachycardia, Ventricular hypertrophy, Investigation, Angiography, Blood pressure, BMI, Bone mineral density, DEXA scan, Echocardiogram, FT3, FT4, GH, GH suppression, Haematoxylin and eosin staining, Heart rate, Histopathology, IGF1, Immunostaining, MRI, Pituitary function, Sex hormone binding globulin, SHBG, Thyroid function, Thyroid ultrasonography, TRH stimulation, Triiodothyronine (T3) suppression, TSH, Intervention, Resection of tumour, Transsphenoidal surgery, Medication, Aspirin, Beta-blockers, Carbimazole, Metoprolol, Statins, Related Disciplines, Surgery, Publication Details, Case Report Type, Unique/unexpected symptoms or presentations of a disease
DOI:
https://doi.org/10.1530/EDM-19-0068
Volume/Issue:
Volume 2019: Issue 1
Open access
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.

Taxonomy:
Clinical Overview, Gland/Organ, Pituitary, Topic, Pituitary, Hormone, Cortisol, GH, IGF1, Prolactin, Somatostatin, Thyroxine (T4), Triiodothyronine (T3), TSH, Condition/ Syndrome, Cerebrospinal fluid leakage, Hyperthyroidism, Meningitis, Pituitary adenoma, Plurihormonal pituitary adenoma, Patient Demographics, Age, Adult, Gender, Female, Ethnicity, White, Country of Treatment, United Kingdom, Diagnosis and Treatment, Signs and Symptoms, Back pain, Cerebrospinal fluid rhinorrhoea, Headache, Hyperthyroidism, Neck pain/discomfort, Investigation, ACTH stimulation, Cortisol, CT scan, FT3, FT4, GH, GH day curve, Haematoxylin and eosin staining, Histopathology, IGF1, Immunohistochemistry, MRI, Prolactin, TSH, Intervention, Resection of tumour, Transsphenoidal surgery, Medication, Antibiotics, Lanreotide, Somatostatin analogues, Related Disciplines, Oncology, Publication Details, Case Report Type, Unique/unexpected symptoms or presentations of a disease
DOI:
https://doi.org/10.1530/EDM-18-0130
Volume/Issue:
Volume 2019: Issue 1
Open access
Laura Hamilton Adams Department of Endocrinology, Diabetes, and Metabolism, University of Kentucky, Lexington, Kentucky, USA

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Derick Adams Department of Endocrinology, Diabetes, and Metabolism, University of Kentucky, Lexington, Kentucky, USA

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Summary

Co-secreting TSH and growth hormone pituitary adenomas are rare. We present a case of a 55-year-old woman who presented with symptoms of neck fullness. Ultrasound revealed multiple thyroid nodules and examination revealed several clinical features of acromegaly. She was found to have a co-secreting TSH and growth hormone pituitary macroadenoma. She underwent surgical resection followed by gamma knife radiation, which resulted in complete remission of her TSH and GH-secreting adenoma.

Learning points:

  • TSH-secreting pituitary adenomas are rare and about one-third co-secrete other hormones.

  • Thyroid nodules are common in acromegaly and can be the presenting sign of a growth hormone-secreting pituitary adenoma.

  • In the workup of acromegaly, assessment of other pituitary hormones is essential, even in the absence of symptoms of other pituitary hormone dysfunction.

  • Complete remission of co-secreting GH and TSH pituitary macroadenomas is possible with surgery and radiation alone.

Taxonomy:
Clinical Overview, Gland/Organ, Pituitary, Topic, Pituitary, Hormone, Cortisol, FSH, GH, IGF1, IGF2, LH, Prolactin, Thyroxine (T4), TSH, Condition/ Syndrome, Acromegaly, Diabetes insipidus - neurogenic/central, Hyperthyroidism, Hypogonadism, Plurihormonal pituitary adenoma, Thyroid nodule, Patient Demographics, Age, Adult, Gender, Female, Ethnicity, White, Country of Treatment, United States, Diagnosis and Treatment, Signs and Symptoms, Amenorrhoea, Diabetes insipidus, Face - coarse features, Feet - increased size, Hands - enlargement, Headache, Hyperthyroidism, Hypogonadism, Neck mass, Neck pain/discomfort, Polyuria, Soft tissue swelling, Thyroid nodule, Investigation, ACTH stimulation, Cortisol (9am), FSH, FT4, GH, Histopathology, IGF1, IGF2, LH, MRI, Plasma osmolality, Prolactin, Sodium, Thyroid function, Thyroid ultrasonography, TSH, Urine osmolality, Intervention, Gamma knife radiosurgery, Resection of tumour, Transsphenoidal surgery, Medication, Desmopressin, Glucocorticoids, Hydrocortisone, Publication Details, Case Report Type, Unique/unexpected symptoms or presentations of a disease
DOI:
https://doi.org/10.1530/EDM-18-0067
Volume/Issue:
Volume 2018: Issue 1
Open access
Philip C Johnston Department of Endocrinology, Diabetes and Metabolism, Cleveland Clinic Foundation, 9500 Euclid Avenue Desk F20, Cleveland, Ohio 44195, USA

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Amir H Hamrahian Department of Endocrinology, Diabetes and Metabolism, Cleveland Clinic Foundation, 9500 Euclid Avenue Desk F20, Cleveland, Ohio 44195, USA

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Richard A Prayson Patholgy and Laboratory Medicine Institute, Rose Ella Burkhardt Brain Tumor and Neuro-Oncology Center, Cleveland Clinic, Cleveland, Ohio 44195, USA

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Laurence Kennedy Department of Endocrinology, Diabetes and Metabolism, Cleveland Clinic Foundation, 9500 Euclid Avenue Desk F20, Cleveland, Ohio 44195, USA

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Robert J Weil Department of Neurosurgery and the Neurological Institute, Rose Ella Burkhardt Brain Tumor and Neuro-Oncology Center, Cleveland Clinic, Cleveland, Ohio 44195, USA

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Summary

A 54-year-old woman presented with bi-temporal hemianopia, palpitations, and diaphoresis. An invasive pituitary macroadenoma was discovered. The patient had biochemical evidence of secondary hyperthyroidism and GH excess; however, she did not appear to be acromegalic. Surgical removal of the pituitary mass revealed a plurihormonal TSH/GH co-secreting pituitary adenoma. TSH-secreting adenomas can co-secrete other hormones including GH, prolactin, and gonadotropins; conversely, co-secretion of TSH from a pituitary adenoma in acromegaly is infrequent.

Learning points

  • This case highlights an unusual patient with a rare TSH/GH co-secreting pituitary adenoma with absence of the clinical features of acromegaly.

  • Plurihormonality does not always translate into the clinical features of hormonal excess.

  • There appears to be a clinical and immunohistochemical spectrum present in plurihormonal tumors.

Taxonomy:
Clinical Overview, Gland/Organ, Pituitary, Topic, Pituitary, Hormone, GH, TSH, Condition/ Syndrome, Pituitary adenoma, Plurihormonal pituitary adenoma, Patient Demographics, Age, Adult, Gender, Female, Ethnicity, White, Country of Treatment, United States, Diagnosis and Treatment, Signs and Symptoms, Hemianopia, Hirsutism, Hot flushes, Obesity, Palpitations, Sweating, Investigation, ACTH stimulation, Cortisol (serum), FT4, GH, IGF1, Immunohistochemistry, MRI, Prolactin, Thyroid function, Total T3, Total T4, TSH, Intervention, Transsphenoidal surgery, Medication, Methimazole, Octreotide, Related Disciplines, Surgery, Publication Details, Case Report Type, Unique/unexpected symptoms or presentations of a disease
DOI:
https://doi.org/10.1530/EDM-14-0070
Volume/Issue:
Volume 2015: Issue 1
Open access