Related Disciplines > Surgery

You are looking at 81 - 90 of 92 items

P Hanson Endocrinology, University Hospital of Coventry and Warwickshire, Coventry, UK

Search for other papers by P Hanson in
Google Scholar
PubMed
Close
,
M Pandit Endocrinology, University Hospital of Coventry and Warwickshire, Coventry, UK

Search for other papers by M Pandit in
Google Scholar
PubMed
Close
,
V Menon Endocrinology, University Hospital of Coventry and Warwickshire, Coventry, UK

Search for other papers by V Menon in
Google Scholar
PubMed
Close
,
S Roberts Endocrinology, University Hospital of Coventry and Warwickshire, Coventry, UK

Search for other papers by S Roberts in
Google Scholar
PubMed
Close
, and
T M Barber Endocrinology, University Hospital of Coventry and Warwickshire, Coventry, UK

Search for other papers by T M Barber in
Google Scholar
PubMed
Close

Summary

The case is a 34-year-old woman with long-standing type 1 diabetes mellitus with existing follow-up in the outpatient clinic at the Warwickshire Institute for the Study of Diabetes, Endocrinology and Metabolism, UHCW. She had maintained good glycaemic control and glycaemic stability with basal bolus regimen for many years. She had not developed any diabetes-related complications and had no other co-morbidities. Six months ago, she presented to A&E with sudden-onset, well-localised and severe pain in the right iliac fossa, just lateral to the para-umbilical area. Her biochemistry was normal. Ultrasound scan, however, revealed a right-sided ovarian cyst, which was thought to have caused pain to her. She was discharged from A&E with simple analgesia. On subsequent gynaecological follow-up 4 weeks later, her pain remained severe and examination revealed an exquisitely tender subcutaneous nodule at the same location measuring 2 cm in diameter. Magnetic resonance imaging (MRI) scan at the time revealed a 1 cm mass in the subcutaneous adipose tissue, which co-localised to her pain. The mass demonstrated a central fat signal surrounded by a peripheral ring: observations consistent with fat necrosis. There were other smaller subcutaneous nodules also observed in the left para-umbilical area. Subsequent surgical resection of the main area of fat necrosis was performed. The patient made an excellent recovery and her pain resolved post-operatively. Histology confirmed the presence of fat necrosis. Fat necrosis is a rare complication of s.c. insulin injection. This case illustrates the importance of considering this diagnosis in patients who inject insulin and develop localised injection-site pain.

Learning points

  • Fat necrosis is a rare complication of insulin injections that can manifest with severe, persistent and well-localised pain.

  • Fat necrosis can masquerade as other pathologies causing diagnostic confusion.

  • The imaging modality of choice for accurate diagnosis of fat necrosis is MRI.

  • Histological confirmation of fat necrosis is important.

  • Appropriate management of localised fat necrosis is surgical excision, with avoidance of further insulin injections into the affected area.

Open access
Maryam Rahman Department of Neurosurgery

Search for other papers by Maryam Rahman in
Google Scholar
PubMed
Close
,
Ignacio Jusué-Torres Department of Neurosurgery

Search for other papers by Ignacio Jusué-Torres in
Google Scholar
PubMed
Close
,
Abdulrahman Alkabbani Division of Endocrinology, Department of Medicine

Search for other papers by Abdulrahman Alkabbani in
Google Scholar
PubMed
Close
,
Roberto Salvatori Division of Endocrinology, Department of Medicine

Search for other papers by Roberto Salvatori in
Google Scholar
PubMed
Close
,
Fausto J Rodríguez Department of Pathology, Johns Hopkins University, 600 North Wolfe Street, Phipps 1-111, Baltimore, Maryland 21287, USA

Search for other papers by Fausto J Rodríguez in
Google Scholar
PubMed
Close
, and
Alfredo Quinones-Hinojosa Department of Neurosurgery

Search for other papers by Alfredo Quinones-Hinojosa in
Google Scholar
PubMed
Close

Summary

Pituitary adenomas are usually solitary lesions. Rarely, patients may present with two distinct pituitary tumors. We report a case of synchronous secretory pituitary adenomas in a woman who initially presented with elevated prolactin levels. She was initially treated with cabergoline, but, after many years, she began developing symptoms consistent with acromegaly. Imaging revealed two distinct tumors within the pituitary gland. Endocrinological investigation confirmed acromegaly. At the time of surgery, two separate tumors were identified and resected. Pathological analysis demonstrated one tumor as a prolactinoma, and the other tumor as a GH-secreting adenoma. Postoperatively, her GH and IGF1 levels normalized, while the prolactin level remained slightly above normal. This case highlights that GH and prolactin level elevation is not always from co-secretion by the same adenoma.

Learning points

  • Synchronous pituitary adenomas represent <0.5% of pituitary tumors requiring surgery.

  • In the setting of elevated GH and prolactin levels, one cannot assume that they are co-secreted by the same adenoma.

  • A careful study of hormonal workup and pre-operative imaging is necessary for synchronous pituitary adenomas to assure resection of both tumors.

Open access
Siew Hui Foo
Search for other papers by Siew Hui Foo in
Google Scholar
PubMed
Close
and
Shahada A H Sobah Department of Haematology, Ampang Hospital, Selangor, Malaysia

Search for other papers by Shahada A H Sobah in
Google Scholar
PubMed
Close

Summary

Hypopituitarism is a rare presentation of Burkitt's lymphoma (BL). The purpose of this report is to present a case of BL presenting with panhypopituitarism and to review other case reports of lymphoma presenting with pituitary dysfunction to highlight the distinguishing features of these cases from other benign aetiologies of pituitary dysfunction such as non-functioning pituitary adenomas. We reviewed a total of 11 cases of lymphoma presenting with pituitary dysfunction published from 1998 to 2013 including the present case. The demographics, clinical presentations, laboratory features, radiological findings, histological diagnosis, treatment administered and outcomes were described. Of the total number of patients, 45.5% of the cases had diffuse large B-cell lymphoma while 27.3% had BL. Anterior pituitary dysfunction was more common than posterior pituitary dysfunction at presentation. The other common associated presenting symptoms were painful ophthalmoplegia, cranial nerve palsies and constitutional symptoms. Hypothalamic–pituitary abnormalities were often demonstrated radiologically to be associated with cavernous sinus and/or stalk involvement. All patients who completed immunochemotherapy responded haematologically. Pituitary dysfunction also improved in most cases although the recovery tended to be partial. In conclusion, a high index of suspicion of underlying malignancy, such as lymphoma, should be present in patients presenting with acute pituitary dysfunction associated with painful ophthalmoplegia, rapidly evolving neurological features, radiological features atypical of a pituitary adenoma and constitutional symptoms. An early diagnosis is essential as prompt initiation of definitive therapy will induce disease remission and recovery of pituitary dysfunction.

Learning points

  • Hypopituitarism may be the presenting symptom of lymphoma in the absence of associated overt symptoms or signs of a haematological malignancy resulting in delay in diagnosis and institution of treatment.

  • Pituitary dysfunction due to tumour infiltration has a greater tendency to involve the posterior pituitary and infundibulum resulting in diabetes insipidus and hyperprolactinaemia compared with a non-functioning pituitary adenoma.

  • The common associated symptoms of hypopituitarism due to lymphoma infiltration of the hypothalamic–pituitary system include painful ophthalmoplegia, cranial nerve palsies and constitutional symptoms.

  • Radiological abnormalities of the hypothalamic–pituitary region are usually present and often associated with cavernous sinus or stalk involvement.

  • With early institution of definitive treatment, both haematological response and improvement of pituitary dysfunction are expected although the reversal of hypopituitarism tends to be partial and delayed.

  • A high index of suspicion of underlying malignancy such as lymphoma should be present in patients presenting with acute pituitary dysfunction associated with painful ophthalmoplegia, radiological features atypical of pituitary adenomas and constitutional symptoms to enable early diagnosis and prompt initiation of definitive therapy.

Open access
Lauren J Baker Endocrinology Department, Concord Hospital, Sydney, New South Wales, Australia

Search for other papers by Lauren J Baker in
Google Scholar
PubMed
Close
,
Anthony J Gill Pathology Department, Royal North Shore Hospital, Sydney, New South Wales, Australia
Sydney Medical School, University of Sydney, Sydney, New South Wales, Australia

Search for other papers by Anthony J Gill in
Google Scholar
PubMed
Close
,
Charles Chan Sydney Medical School, University of Sydney, Sydney, New South Wales, Australia
Anatomical Pathology Department, Concord Hospital, Sydney, New South Wales, Australia

Search for other papers by Charles Chan in
Google Scholar
PubMed
Close
,
Betty P C Lin Sydney Medical School, University of Sydney, Sydney, New South Wales, Australia
Anatomical Pathology Department, Concord Hospital, Sydney, New South Wales, Australia

Search for other papers by Betty P C Lin in
Google Scholar
PubMed
Close
, and
Bronwyn A Crawford Endocrinology Department, Concord Hospital, Sydney, New South Wales, Australia
Sydney Medical School, University of Sydney, Sydney, New South Wales, Australia

Search for other papers by Bronwyn A Crawford in
Google Scholar
PubMed
Close

Summary

In 2006, a 58-year-old woman presented with thyrotoxicosis. She had undergone left hemithyroidectomy 14 years before for a benign follicular adenoma. Ultrasound imaging demonstrated bilateral cervical lymphadenopathy with enhanced tracer uptake in the left lateral neck on a Technetium-99m uptake scan. Fine-needle aspiration biopsy of a left lateral neck node was insufficient for a cytological diagnosis; however, thyroglobulin (Tg) washings were strongly positive. The clinical suspicion was of functionally active metastatic thyroid cancer in cervical lymph nodes. A completion thyroidectomy and bilateral cervical lymph node dissection were performed. Histology demonstrated benign multinodularity in the right hemithyroid, with bilateral reactive lymphadenopathy and 24 benign hyperplastic thyroid nodules in the left lateral neck that were classified as parasitic thyroid nodules. As there had been a clinical suspicion of thyroid cancer, and the hyperplastic/parasitic thyroid tissue in the neck was extensive, the patient was given ablative radioactive iodine (3.7 GBq). After 2 years, a diagnostic radioactive iodine scan was clear and the serum Tg was undetectable. The patient has now been followed for 7 years with no evidence of recurrence. Archived tissue from a left lateral neck thyroid nodule has recently been analysed for BRAF V600E mutation, which was negative.

Learning points

  • Thyrotoxicosis due to functional thyroid tissue in the lateral neck is very rare and may be due to metastatic thyroid cancer or benign parasitic thyroid tissue.

  • Parasitic thyroid nodules should be considered as a differential diagnosis of lateral neck thyroid deposits, particularly where there is a history of prior thyroid surgery.

  • Parasitic thyroid nodules may occur as a result of traumatic rupture or implantation from a follicular adenoma at the time of surgery.

  • The use of ablative radioactive iodine may be appropriate, as resection of all parasitic thyroid tissue can prove difficult.

  • BRAF mutational analysis of parasitic thyroid tissue may provide extra reassurance in the exclusion of papillary thyroid carcinoma.

Open access
Ramez Ibrahim Royal Hallamshire Hospital, Sheffield, UK

Search for other papers by Ramez Ibrahim in
Google Scholar
PubMed
Close
,
Atul Kalhan University Hospital of Wales, Cardiff, UK

Search for other papers by Atul Kalhan in
Google Scholar
PubMed
Close
,
Alistair Lammie Cardiff University, Cardiff, UK

Search for other papers by Alistair Lammie in
Google Scholar
PubMed
Close
,
Christine Kotonya Bronglais Hospital, Aberystwyth, UK

Search for other papers by Christine Kotonya in
Google Scholar
PubMed
Close
,
Ravindra Nannapanenni University Hospital of Wales, Cardiff, UK

Search for other papers by Ravindra Nannapanenni in
Google Scholar
PubMed
Close
, and
Aled Rees Institute of Molecular and Experimental Medicine, Cardiff University, Cardiff, UK

Search for other papers by Aled Rees in
Google Scholar
PubMed
Close

Summary

A 30-year-old female presented with a history of secondary amenorrhoea, acromegalic features and progressive visual deterioration. She had elevated serum IGF1 levels and unsuppressed GH levels after an oral glucose tolerance test. Magnetic resonance imaging revealed a heterogeneously enhancing space-occupying lesion with atypical extensive calcification within the sellar and suprasellar areas. Owing to the extent of calcification, the tumour was a surgical challenge. Postoperatively, there was clinical, radiological and biochemical evidence of residual disease, which required treatment with a somatostatin analogue and radiotherapy. Mutational analysis of the aryl hydrocarbon receptor-interacting protein (AIP) gene was negative. This case confirms the relatively rare occurrence of calcification within a pituitary macroadenoma and its associated management problems. The presentation, biochemical, radiological and pathological findings are discussed in the context of the relevant literature.

Learning points

  • Calcification of pituitary tumours is relatively rare.

  • Recognising calcification in pituitary adenomas on preoperative imaging is important in surgical decision-making.

  • Gross total resection can be difficult to achieve in the presence of extensive calcification and dictates further management and follow-up to achieve disease control.

Open access
Caterina Policola Department of Endocrinology and Metabolic Diseases, Università Cattolica del Sacro Cuore, Rome, Italy

Search for other papers by Caterina Policola in
Google Scholar
PubMed
Close
,
Victoria Stokes Oxford Centre for Diabetes, Endocrinology and Metabolism, Oxford University Hospitals NHS Trust, Old Road, Oxford OX3 7LE, UK

Search for other papers by Victoria Stokes in
Google Scholar
PubMed
Close
,
Niki Karavitaki Oxford Centre for Diabetes, Endocrinology and Metabolism, Oxford University Hospitals NHS Trust, Old Road, Oxford OX3 7LE, UK

Search for other papers by Niki Karavitaki in
Google Scholar
PubMed
Close
, and
Ashley Grossman Oxford Centre for Diabetes, Endocrinology and Metabolism, Oxford University Hospitals NHS Trust, Old Road, Oxford OX3 7LE, UK

Search for other papers by Ashley Grossman in
Google Scholar
PubMed
Close

Summary

Opiate drugs such as morphine are in extensive use for pain relief and palliation. It is well established that these drugs can cause changes in endocrine function, but such effects are not always sufficiently appreciated in clinical practice, especially in relation to the hypothalamic–pituitary–adrenal (HPA) axis. Herein, we report on an 18-year-old man who was diagnosed with a slipped left femoral epiphysis following a long history of pain in his leg. On examination, he was thought to look relatively young for his age and therefore the orthopaedic surgeons arranged an endocrine assessment, which showed an undetectable concentration of serum cortisol and a suppressed concentration of testosterone; therefore, he was referred urgently with a diagnosis of hypopituitarism. We elicited a history that he had been treated with opiate analgesics for 3 days at the time of his original blood tests. Full endocrine assessment including a short Synacthen test revealed that he now had normal adrenal and pituitary function. We conclude that his morphine therapy had caused profound suppression of his HPA and pituitary–gonadal axes and suggest that clinicians should be aware of these significant changes in patients on even short-term opiate therapy.

Learning points

  • Therapy with opiates is the standard therapy for severe acute and chronic pain.

  • Such drugs cause profound changes in endocrine function.

  • Importantly, opiates suppress the HPA axis at a central level.

  • Short-term therapy with morphine could be the cause of biochemical adrenocortical insufficiency.

  • Morphine and related drugs also suppress the pituitary–gonadal axis.

  • After discontinuation of therapy with such drugs, adrenal function improves.

Open access
Gemma Xifra Departments of Diabetes, Endocrinology and Nutrition, Hospital Dr Josep Trueta, Girona, Spain

Search for other papers by Gemma Xifra in
Google Scholar
PubMed
Close
,
Silvia Mauri Departments of Diabetes, Endocrinology and Nutrition, Hospital Dr Josep Trueta, Girona, Spain

Search for other papers by Silvia Mauri in
Google Scholar
PubMed
Close
,
Jordi Gironès Surgery, Hospital Dr Josep Trueta, Girona, Spain

Search for other papers by Jordi Gironès in
Google Scholar
PubMed
Close
,
José Ignacio Rodríguez Hermosa Surgery, Hospital Dr Josep Trueta, Girona, Spain

Search for other papers by José Ignacio Rodríguez Hermosa in
Google Scholar
PubMed
Close
,
Josep Oriola Biochemistry and Molecular Genetics Department, CDB Hospital Clinic, Barcelona, Spain

Search for other papers by Josep Oriola in
Google Scholar
PubMed
Close
,
Wifredo Ricart Departments of Diabetes, Endocrinology and Nutrition, Hospital Dr Josep Trueta, Girona, Spain

Search for other papers by Wifredo Ricart in
Google Scholar
PubMed
Close
, and
José Manuel Fernández-Real Departments of Diabetes, Endocrinology and Nutrition, Hospital Dr Josep Trueta, Girona, Spain
CIBERobn, pathophysiology of obesity and nutrition, Spain

Search for other papers by José Manuel Fernández-Real in
Google Scholar
PubMed
Close

Summary

Background: Thyroid hormone resistance (RTH) is a rare cause of thyroid dysfunction. High TSH levels, as described in RTH syndrome, are known to be associated with an increased risk of developing thyroid nodules with subsequent growth and malignancy.

Patient findings: In 2006, a 29-year-old Caucasian man presented with a palpable mass in the neck. Increased free thyroxine and triiodothyronine levels were found in the context of unsuppressed TSH levels, despite no signs or symptoms of hyperthyroidism. Ultrasonography revealed a multinodular and enlarged goitre, and fine-needle aspiration cytology revealed suspicious features of malignancy. After excluding pituitary tumour and levothyroxine (l-T4) treatment, the patient was diagnosed with generalized RTH. Screening for all the known mutations in thyroid hormone receptor-β (TR β (THRB)) was negative. Thyroidectomy disclosed five Hürthle adenomas and three hyperplasic nodules. Euthyroidism was achieved after surgery with 6.1 μg/kg per day of l-T4.

Conclusion: RTH may be a risk factor that predisposes to the development of multiple Hürthle cell adenomas. To our knowledge, this is the first case of multiple Hürthle cell adenomas in a patient with RTH.

Learning points

  • High TSH levels, as described in RTH syndrome, are known to be associated with an increased risk of developing thyroid nodules, with subsequent growth and malignancy.

  • The exact role of TR β mutants in thyroid carcinogenesis is still undefined.

  • We report the first case of multiple Hürthle cell adenomas associated with RTH.

Open access
Takuma Hara Department of Neurosurgery, Faculty of Medicine, University of Tsukuba, 1-1-1 Tennodai, Tsukuba, Ibaraki 305-0006, Japan

Search for other papers by Takuma Hara in
Google Scholar
PubMed
Close
,
Hiroyoshi Akutsu Department of Neurosurgery, Faculty of Medicine, University of Tsukuba, 1-1-1 Tennodai, Tsukuba, Ibaraki 305-0006, Japan

Search for other papers by Hiroyoshi Akutsu in
Google Scholar
PubMed
Close
,
Tetsuya Yamamoto Department of Neurosurgery, Faculty of Medicine, University of Tsukuba, 1-1-1 Tennodai, Tsukuba, Ibaraki 305-0006, Japan

Search for other papers by Tetsuya Yamamoto in
Google Scholar
PubMed
Close
,
Eiichi Ishikawa Department of Neurosurgery, Faculty of Medicine, University of Tsukuba, 1-1-1 Tennodai, Tsukuba, Ibaraki 305-0006, Japan

Search for other papers by Eiichi Ishikawa in
Google Scholar
PubMed
Close
,
Masahide Matsuda Department of Neurosurgery, Faculty of Medicine, University of Tsukuba, 1-1-1 Tennodai, Tsukuba, Ibaraki 305-0006, Japan

Search for other papers by Masahide Matsuda in
Google Scholar
PubMed
Close
, and
Akira Matsumura Department of Neurosurgery, Faculty of Medicine, University of Tsukuba, 1-1-1 Tennodai, Tsukuba, Ibaraki 305-0006, Japan

Search for other papers by Akira Matsumura in
Google Scholar
PubMed
Close

Summary

Gastrointestinal perforation is a complication associated with steroid therapy or hypercortisolism, but it is rarely observed in patients with Cushing's disease in clinical practice, and only one case has been reported as a presenting symptom. Herein, we report a rare case of Cushing's disease in which a patient presented with gastrointestinal perforation as a symptom. A 79-year-old man complained of discomfort in the lower abdomen for 6 months. Based on the endocrinological and gastroenterological examinations, he was diagnosed with Cushing's disease with a perforation of the descending colon. After consultation with a gastroenterological surgeon, it was decided that colonic perforation could be conservatively observed without any oral intake and treated with parenteral administration of antibiotics because of the mild systemic inflammation and lack of abdominal guarding. Despite the marked elevated levels of serum cortisol, oral medication was not an option because of colonic perforation. Therefore, the patient was submitted to endonasal adenomectomy to normalize the levels of serum cortisol. Subsequently, a colostomy was successfully performed. Despite its rarity, physicians should be aware that gastrointestinal perforation may be associated with hypercortisolism, especially in elderly patients, and immediate diagnosis and treatment of this life-threatening condition are essential. If a perforation can be conservatively observed, endonasal adenomectomy prior to laparotomy is an alternative treatment option for hypercortisolism.

Learning points

  • Thus far, only one case of gastrointestinal perforation as a presenting clinical symptom of Cushing's disease has been reported.

  • Physicians should be aware that gastrointestinal perforation might be associated with hypercortisolism in elderly patients because elevated levels of serum cortisol may mask the clinical signs of perforation. Because of this masking effect, the diagnosis of the perforation also tends to be delayed.

  • Although parenteral administration of etomidate is a standard treatment option for decreasing the elevated levels of serum cortisol, endonasal adenomectomy prior to laparotomy is an alternative treatment option if etomidate therapy is unavailable.

Open access
Vivienne Yoon Department of Internal Medicine/Division of Endocrinology and Metabolism, VA North Texas Health Care System and the University of Texas Southwestern Medical Center, 5323 Harry Hines Boulevard, Dallas, Texas 75390-8857, USA

Search for other papers by Vivienne Yoon in
Google Scholar
PubMed
Close
,
Aliya Heyliger Department of Internal Medicine/Division of Endocrinology and Metabolism, VA North Texas Health Care System and the University of Texas Southwestern Medical Center, 5323 Harry Hines Boulevard, Dallas, Texas 75390-8857, USA

Search for other papers by Aliya Heyliger in
Google Scholar
PubMed
Close
,
Takashi Maekawa Department of Pathology, Tohoku University School of Medicine and Hospital at Sendai, Sendai, Japan

Search for other papers by Takashi Maekawa in
Google Scholar
PubMed
Close
,
Hironobu Sasano Department of Pathology, Tohoku University School of Medicine and Hospital at Sendai, Sendai, Japan

Search for other papers by Hironobu Sasano in
Google Scholar
PubMed
Close
,
Kelley Carrick Departments of Pathology

Search for other papers by Kelley Carrick in
Google Scholar
PubMed
Close
,
Stacey Woodruff Surgery, University of Texas Southwestern Medical Center, Dallas, Texas, USA

Search for other papers by Stacey Woodruff in
Google Scholar
PubMed
Close
,
Jennifer Rabaglia Surgery, University of Texas Southwestern Medical Center, Dallas, Texas, USA

Search for other papers by Jennifer Rabaglia in
Google Scholar
PubMed
Close
,
Richard J Auchus Department of Internal Medicine/Division of Metabolism, Endocrinology and Diabetes, University of Michigan Health System, Ann Arbor, Michigan, USA

Search for other papers by Richard J Auchus in
Google Scholar
PubMed
Close
, and
Hans K Ghayee Department of Internal Medicine/Division of Endocrinology and Metabolism, VA North Texas Health Care System and the University of Texas Southwestern Medical Center, 5323 Harry Hines Boulevard, Dallas, Texas 75390-8857, USA

Search for other papers by Hans K Ghayee in
Google Scholar
PubMed
Close

Summary

Objective: To recognize that benign adrenal adenomas can co-secrete excess aldosterone and cortisol, which can change clinical management.

Methods: We reviewed the clinical and histological features of an adrenal tumor co-secreting aldosterone and cortisol in a patient. Biochemical testing as well as postoperative immunohistochemistry was carried out on tissue samples for assessing enzymes involved in steroidogenesis.

Results: A patient presented with hypertension, hypokalemia, and symptoms related to hypercortisolism. The case demonstrated suppressed renin concentrations with an elevated aldosterone:renin ratio, abnormal dexamethasone suppression test results, and elevated midnight salivary cortisol concentrations. The patient had a right adrenal nodule with autonomous cortisol production and interval growth. Right adrenalectomy was carried out. Postoperatively, the patient tolerated the surgery, but he was placed on a short course of steroid replacement given a subnormal postoperative serum cortisol concentration. Long-term follow-up of the patient showed that his blood pressure and glucose levels had improved. Histopathology slides showed positive staining for 3β-hydroxysteroid dehydrogenase, 11β-hydroxylase, and 21 hydroxylase.

Conclusion: In addition to the clinical manifestations and laboratory values, the presence of these enzymes in this type of tumor provides support that the tumor in this patient was able to produce mineralocorticoids and glucocorticoids. The recognition of patients with a tumor that is co-secreting aldosterone and cortisol can affect decisions to treat with glucocorticoids perioperatively to avoid adrenal crisis.

Learning points

  • Recognition of the presence of adrenal adenomas co-secreting mineralocorticoids and glucocorticoids.

  • Consideration for perioperative and postoperative glucocorticoid use in the treatment of co-secreting adrenal adenomas.

Open access
Rajesh Rajendran Department of Diabetes and Endocrinology, The Ipswich Hospital NHS Trust, Ipswich IP4 5PD, UK

Search for other papers by Rajesh Rajendran in
Google Scholar
PubMed
Close
,
Sarita Naik Department of Diabetes and Endocrinology, Royal United Hospital Bath NHS Trust, Bath BA1 3NG, UK

Search for other papers by Sarita Naik in
Google Scholar
PubMed
Close
,
Derek D Sandeman Department of Diabetes and Endocrinology, University Hospital of Southampton NHS Foundation Trust, Southampton SO16 6YD, UK

Search for other papers by Derek D Sandeman in
Google Scholar
PubMed
Close
, and
Azraai B Nasruddin Department of Diabetes and Endocrinology, University Hospital of Southampton NHS Foundation Trust, Southampton SO16 6YD, UK

Search for other papers by Azraai B Nasruddin in
Google Scholar
PubMed
Close

Summary

We report the use of pasireotide in a rare and unusual case of pituitary macroadenoma co-secreting GH, prolactin and ACTH. A 62-year-old Caucasian man presented with impotence. Clinically, he appeared acromegalic and subsequent investigations confirmed GH excess and hyperprolactinaemia. Magnetic resonance imaging (MRI) of pituitary revealed a large pituitary macroadenoma. He underwent trans-sphenoidal surgery and histology confirmed an adenoma with immunohistochemistry positive for ACTH, GH and prolactin. Acromegaly was not cured following surgery and inadequately controlled despite subsequent octreotide therapy. He underwent further debulking pituitary surgery, following which IGF1 levels improved but still high. This time adenoma cells showed immunohistochemistry positivity for ACTH only, following which subsequent investigations confirmed intermittent hypercortisolaemia compatible with pituitary Cushing's disease. We recommended radiotherapy, but in view of the pluripotential nature of the tumour, we proceeded with a trial of s.c. pasireotide therapy on the basis that it may control both his acromegaly and Cushing's disease. After 3 months of pasireotide therapy, his mean GH and IGF1 levels improved significantly, with improvement in his symptoms but intermittent hypercortisolaemia persists. His glycaemic control deteriorated requiring addition of new anti-diabetic medication. MRI imaging showed loss of contrast uptake within the tumour following pasireotide therapy but no change in size. We conclude that our patient has had a partial response to pasireotide therapy. Long-term follow-up studies are needed to establish its safety and efficacy in patients with acromegaly and/or Cushing's disease.

Learning points

  • Plurihormonal pituitary adenomas are rare and unusual.

  • Patients with pituitary adenomas co-secreting ACTH and GH are more likely to present with acromegaly because GH excess can mask hypercortisolaemia.

  • Pasireotide holds potential where conventional somatostatin analogues are not effective in acromegaly due to higher affinity for somatostatin receptor subtypes 1, 2, 3 and 5.

  • Significant deterioration in glycaemic control remains a concern in the use of pasireotide.

  • Currently, long-term safety and efficacy of pasireotide in patients with acromegaly and/or Cushing's disease are not fully clear.

Open access