Browse

You are looking at 1 - 10 of 52 items for :

  • Gland/Organ x
  • Clinical Overview x
Clear All
Hakan Ozoran Oxford Centre for Diabetes, Endocrinology and Metabolism, University of Oxford, Oxford, UK
Clinical Medical School, University of Oxford, Oxford, UK
Green Templeton College, University of Oxford, Oxford, UK

Search for other papers by Hakan Ozoran in
Google Scholar
PubMed
Close
,
Phoenix Guwa Clinical Medical School, University of Oxford, Oxford, UK
Green Templeton College, University of Oxford, Oxford, UK

Search for other papers by Phoenix Guwa in
Google Scholar
PubMed
Close
,
Pam Dyson Oxford Centre for Diabetes, Endocrinology and Metabolism, University of Oxford, Oxford, UK

Search for other papers by Pam Dyson in
Google Scholar
PubMed
Close
,
Garry D Tan Oxford Centre for Diabetes, Endocrinology and Metabolism, Oxford University Hospitals Foundation Trust, Oxford, UK
NIHR Biomedical Research Centre, Oxford University Hospitals Foundation Trust, Oxford, UK

Search for other papers by Garry D Tan in
Google Scholar
PubMed
Close
, and
Fredrik Karpe Oxford Centre for Diabetes, Endocrinology and Metabolism, University of Oxford, Oxford, UK
NIHR Biomedical Research Centre, Oxford University Hospitals Foundation Trust, Oxford, UK

Search for other papers by Fredrik Karpe in
Google Scholar
PubMed
Close

Summary

The use of a low-carbohydrate diet (LCD) reduces insulin requirements in insulinopenic states such as type 1 diabetes mellitus (T1DM). However, the use of potentially ketogenic diets in this clinical setting is contentious and the mechanisms underlying their impact on glycaemic control are poorly understood. We report a case of a patient with a late-onset classic presentation of T1DM who adopted a very low-carbohydrate diet and completely avoided insulin therapy for 18 months, followed by tight glycaemic control on minimal insulin doses. The observations suggest that adherence to an LCD in T1DM, implemented soon after diagnosis, can facilitate an improved and less variable glycaemic profile in conjunction with temporary remission in some individuals. Importantly, these changes occurred in a manner that did not lead to a significant increase in blood ketone (beta-hydroxybutyrate) concentrations. This case highlights the need for further research in the form of randomised controlled trials to assess the long-term safety and sustainability of carbohydrate-reduced diets in T1DM.

Learning points

  • This case highlights the potential of low-carbohydrate diets (LCDs) in type 1 diabetes mellitus (T1DM) to mediate improved diabetes control and possible remission soon after diagnosis.

  • Could carbohydrate-reduced diets implemented early in the course of T1DM delay the decline in endogenous insulin production?

  • Adherence to an LCD in T1DM can facilitate an improved and less variable glycaemic profile.

  • This case suggests that LCDs in T1DM may not be associated with a concerning supraphysiological ketonaemia.

Open access
Sandra Martens Ghent University Hospital, Ghent, Belgium

Search for other papers by Sandra Martens in
Google Scholar
PubMed
Close
and
Bruno Lapauw Ghent University Hospital, Ghent, Belgium
Ghent University, Ghent, Belgium

Search for other papers by Bruno Lapauw in
Google Scholar
PubMed
Close

Summary

Mitotane is used for treatment of advanced adrenocortical carcinoma. It is administered when the carcinoma is unresectable, metastasized, or at high-risk of recurrence after resection. In addition, mitotane is considered to have direct adrenolytic effects. Because of its narrow therapeutic–toxic range, therapeutic drug monitoring (TDM) is warranted. In 2020, a left-sided adrenal gland tumor was found (5.8 cm) in a 38-year-old man. Considering the size of this lesion and inability to exclude an adrenocortical carcinoma on imaging, a laparoscopic adrenalectomy was performed. Histopathologic examination determined presence of an adrenocortical carcinoma (pT2N0M0 ENSAT stadium II; ki67 10–15%). There was no evidence for residual or metastatic disease but given the high risk of recurrence, adjuvant therapy with mitotane was initiated. During TDM, a sudden and spuriously high level of mitotane was observed but without signs or symptoms of toxicity. After exploration, it was found that this high concentration was completely due to uncontrolled hypertriglyceridemia. After correction thereof, mitotane levels were again in the therapeutic range. This observation underscores the importance of TDM sampling in a fasting state with concurrent control of prevalent or incident dyslipidemia.

Learning points

  • TDM of mitotane is advocated to achieve therapeutic levels while avoiding toxicity. For correct TDM, sampling should be done at least 12 h after last intake of mitotane.

  • Although sampling in fasting conditions in not explicitly mentioned in the guidelines, fasting state should be considered as elevated serum triglyceride levels might cause spuriously high mitotane levels.

  • In patients undergoing treatment with mitotane and presenting with too high or unexplained fluctuating mitotane levels without signs or symptoms of toxicity, hypertriglyceridemia as a possible cause should be investigated.

  • If dyslipidemia occurs in patients under mitotane treatment, other causes than mitotane (e.g. alcohol abuse and diabetes) should be considered and appropriate treatment should be initiated.

Open access
Ishara Ranathunga Department of Diabetes and Endocrinology, North Cumbria Integrated Care NHS Foundation Trust, Whitehaven, UK

Search for other papers by Ishara Ranathunga in
Google Scholar
PubMed
Close
and
Chandima Idampitiya Department of Diabetes and Endocrinology, North Cumbria Integrated Care NHS Foundation Trust, Whitehaven, UK

Search for other papers by Chandima Idampitiya in
Google Scholar
PubMed
Close

Summary

Type 1 diabetes mellitus (T1DM) is an autoimmune disorder caused by the destruction of the pancreatic beta cells, which produce insulin. Individuals with T1DM usually require at least 3-5 years to develop microvascular complications in comparison to people with type 2 diabetes (T2DM), who may develop complications even before the diagnosis of diabetes. We discuss a patient who presented with proliferative diabetic retinopathy subsequently diagnosed with T1DM and diabetic neuropathy following investigations. Diabetic retinopathy or other microvascular complications as the presenting feature of T1DM is rarely known or reported in the literature. A 33-year-old healthcare worker had been seen by the opticians due to 1-week history of blurred vision. The ophthalmology assessment had confirmed proliferative retinopathy in the right eye and severe non-proliferative retinopathy in the left eye with bilateral clinically significant macular oedema. His BMI was 24.9 kg/m2. The nervous system examination revealed bilateral stocking type peripheral neuropathy. The random venous glucose was 24.9 mmol/L. Plasma ketones were 0.7 mmol/L and HbA1c was 137 mmol/mol. On further evaluation, the anti-glutamic acid decarboxylase (GAD) antibody was positive, confirming the diagnosis of T1DM. He was started on aflibercept injections in both eyes, followed by panretinal photocoagulation. Subsequent nerve conduction studies confirmed the presence of symmetrical polyneuropathy. The pathogenesis of the development of microvascular complications in T1DM is multifactorial. Usually, the development of complications is seen at least a few years following the diagnosis. The occurrence of microvascular complications at presentation is rare. This makes the management challenging and extremely important in preventing the progression of the disease.

Learning points

  • The pathogenesis of the development of microvascular complications in type 1 diabetes mellitus is multifactorial.

  • The development of complications is seen at least a few years following the diagnosis.

  • Occurrence of microvascular complications at presentation is rare.

  • This makes the management challenging and extremely important to prevent the progression of the disease.

Open access
Rikako Nakajima Department of Endocrinology and Metabolism, Tsukuba University Hospital Mito Clinical Education and Training Center, Mito Kyodo General Hospital, Miyamachi, Mito, Ibaraki, Japan

Search for other papers by Rikako Nakajima in
Google Scholar
PubMed
Close
,
Hiroto Idesawa Department of Endocrinology and Metabolism, Tsukuba University Hospital Mito Clinical Education and Training Center, Mito Kyodo General Hospital, Miyamachi, Mito, Ibaraki, Japan

Search for other papers by Hiroto Idesawa in
Google Scholar
PubMed
Close
,
Daisuke Sato Department of Endocrinology and Metabolism, Tsukuba University Hospital Mito Clinical Education and Training Center, Mito Kyodo General Hospital, Miyamachi, Mito, Ibaraki, Japan

Search for other papers by Daisuke Sato in
Google Scholar
PubMed
Close
,
Jun Ito Department of Endocrinology and Metabolism, Tsukuba University Hospital Mito Clinical Education and Training Center, Mito Kyodo General Hospital, Miyamachi, Mito, Ibaraki, Japan

Search for other papers by Jun Ito in
Google Scholar
PubMed
Close
,
Kei Ito Department of Endocrinology and Metabolism, Tsukuba University Hospital Mito Clinical Education and Training Center, Mito Kyodo General Hospital, Miyamachi, Mito, Ibaraki, Japan

Search for other papers by Kei Ito in
Google Scholar
PubMed
Close
,
Masanao Fujii Department of Endocrinology and Metabolism, Tsukuba University Hospital Mito Clinical Education and Training Center, Mito Kyodo General Hospital, Miyamachi, Mito, Ibaraki, Japan

Search for other papers by Masanao Fujii in
Google Scholar
PubMed
Close
,
Takamichi Suzuki Department of Gastrointestinal Surgery, Tsukuba University Hospital Mito Clinical Education and Training Center, Mito Kyodo General Hospital, Miyamachi, Mito, Ibaraki, Japan

Search for other papers by Takamichi Suzuki in
Google Scholar
PubMed
Close
,
Tomoaki Furuta Department of Gastrointestinal Surgery, Tsukuba University Hospital Mito Clinical Education and Training Center, Mito Kyodo General Hospital, Miyamachi, Mito, Ibaraki, Japan

Search for other papers by Tomoaki Furuta in
Google Scholar
PubMed
Close
,
Hitomi Kawai Department of Pathology, Tsukuba University Hospital Mito Clinical Education and Training Center, Mito Kyodo General Hospital, Miyamachi, Mito, Ibaraki, Japan

Search for other papers by Hitomi Kawai in
Google Scholar
PubMed
Close
,
Norio Takayashiki Department of Pathology, Tsukuba University Hospital Mito Clinical Education and Training Center, Mito Kyodo General Hospital, Miyamachi, Mito, Ibaraki, Japan

Search for other papers by Norio Takayashiki in
Google Scholar
PubMed
Close
,
Masanao Kurata Department of Gastrointestinal Surgery, Tsukuba University Hospital Mito Clinical Education and Training Center, Mito Kyodo General Hospital, Miyamachi, Mito, Ibaraki, Japan

Search for other papers by Masanao Kurata in
Google Scholar
PubMed
Close
, and
Hiroaki Yagyu Department of Endocrinology and Metabolism, Tsukuba University Hospital Mito Clinical Education and Training Center, Mito Kyodo General Hospital, Miyamachi, Mito, Ibaraki, Japan

Search for other papers by Hiroaki Yagyu in
Google Scholar
PubMed
Close

Summary

Unawareness of postprandial hypoglycemia for 5 years was identified in a 66-year-old man at a local clinic. The patient was referred to our hospital because of this first awareness of hypoglycemia (i.e. lightheadedness and impaired consciousness) developing after lunch. In a 75 g oral glucose tolerance test, the plasma glucose concentration was decreased to 32 mg/dL (1.8 mmol/L) at 150 min with relatively high concentrations of insulin (8.1 μU/mL), proinsulin (70.3 pmol/L), and C-peptide (4.63 ng/mL). In a prolonged fasting test, the plasma glucose concentration was decreased to 43 mg/dL (2.4 mmol/L) at 66 h with an insulin concentration of 1.4 μU/mL and a C-peptide concentration of 0.49 ng/mL. Computed tomography showed an 18 mm hyperenhancing tumor in the uncinate process of the pancreas. A selective arterial calcium stimulation test showed an elevated serum insulin concentration in the superior mesenteric artery. The patient was then diagnosed with insulinoma and received pancreaticoduodenectomy. Continuous glucose monitoring (CGM) using the Dexcom G6 system showed unawareness of hypoglycemia mainly during the daytime before surgery. When the sensor glucose value was reduced to 55 mg/dL (3.1 mmol/L), the Dexcom G6 system emitted an urgent low glucose alarm to the patient four times for 10 days. Two months after surgery, an overall increase in daily blood glucose concentrations and resolution of hypoglycemia were shown by CGM. We report a case of insulinoma with unawareness of postprandial hypoglycemia in the patient. The Dexcom G6 system was helpful for assessing preoperative hypoglycemia and for evaluating outcomes of treatment by surgery.

Learning points

  • Insulinoma occasionally leads to postprandial hypoglycemia.

  • The CGM system is useful for revealing the presence of unnoticed hypoglycemia and for evaluating treatment outcomes after surgical resection.

  • The Dexcom G6 system has an urgent low glucose alarm, making it particularly suitable for patients who are unaware of hypoglycemia.

Open access
Clemens Gardemann FH Münster Oecotrophologie, Münster, Germany
Clinic for Pediatrics and Adolescent Medicine/Metabolism Laboratory, Universitätsklinikum Münster, Münster, Germany

Search for other papers by Clemens Gardemann in
Google Scholar
PubMed
Close
,
Sonja Knowles FH Münster Oecotrophologie, Münster, Germany

Search for other papers by Sonja Knowles in
Google Scholar
PubMed
Close
, and
Thorsten Marquardt Clinic for Pediatrics and Adolescent Medicine/Metabolism Laboratory, Universitätsklinikum Münster, Münster, Germany

Search for other papers by Thorsten Marquardt in
Google Scholar
PubMed
Close

Summary

Traditional guidelines for type 1 diabetics do not restrict carbohydrates to improve clinical outcomes for patients. This paper highlights the favorable blood glucose control outcomes when a type 1 diabetic focuses on caloric intake from protein and healthy fats instead of the traditional carbohydrate-focused meals. We followed a male type 1 diabetic in his 20s adopting a ketogenic diet through a process of slowly lowering total daily carbohydrate intake. Diabetes-related biomarkers were measured throughout the process. Diabetes-related biomarkers saw massive improvements and ended up in the official non-diabetic range. Total daily insulin requirements dropped by 70%. The patient also experienced great improvements in his quality of life. This study demonstrates the possibility of improving diabetes-related biomarkers through dietary changes, which have positive effects on health outcomes in patients living with this disease.

Learning points

  • The adaptation of a ketogenic diet improved diabetes-related biomarkers in this patient.

  • Diabetes-related biomarkers, such as HbA1c, are the main risk factors for developing complications in diabetics.

  • The ketogenic diet is a feasible approach to minimizing the risk of developing complications in diabetics.

  • Total daily insulin requirements dropped by 67% adapting a ketogenic diet.

  • The patient experienced enormous changes in the quality of life after adapting to the new diet.

  • The safe and physiological state of ketosis might be associated with additional benefits for the patient

Open access
Dimitra Stathi Department of Endocrinology and Diabetes, Guy’s and St Thomas’ NHS Trust, London, UK
School of Cardiovascular Medicine & Sciences, King's College London, London, UK

Search for other papers by Dimitra Stathi in
Google Scholar
PubMed
Close
,
Sufyan Hussain Department of Endocrinology and Diabetes, Guy’s and St Thomas’ NHS Trust, London, UK

Search for other papers by Sufyan Hussain in
Google Scholar
PubMed
Close
,
Danielle Crawley Department of Oncology, Guy’s and St Thomas’ NHS Trust, London, UK

Search for other papers by Danielle Crawley in
Google Scholar
PubMed
Close
, and
Janaka Karalliedde Department of Endocrinology and Diabetes, Guy’s and St Thomas’ NHS Trust, London, UK
School of Cardiovascular Medicine & Sciences, King's College London, London, UK

Search for other papers by Janaka Karalliedde in
Google Scholar
PubMed
Close

Summary

A Caucasian man in his 60s with recent diagnosis of metastatic renal cell carcinoma presented to the emergency department with a 5-day history of severe polyuria, polydipsia and fatigue and 1-day history of confusion, abdominal pain, nausea and vomiting. Investigations revealed an overlap of diabetic ketoacidosis (DKA) and hyperosmolar hyperglycaemic state (HHS). He had received the first dose of immunotherapy with nivolumab and ipilimumab 3 weeks prior to this attendance. New-onset type 1 diabetes (T1DM) was confirmed based on the clinical features at presentation, seropositivity for glutamic acid decarboxylase antibodies and significant insulin deficiency. He is currently on a multiple daily injections of insulin and uses intermittent-scanned glucose monitoring. Given the irreversible impact on beta-cell function and clinical response with insulin resulting in improved diabetes control, immunotherapy was resumed for his metastatic cancer with good radiological response. Although rare, new-onset T1DM can present with DKA and HSS overlap after a single dose of nivolumab/ipilimumab in individuals without pre-existing history of diabetes.

Learning points

  • Although rare, new onset of T1DM after immunotherapy can present with DKA and HSS overlap after a single dose of nivolumab/ipilimumab in individuals without pre-existing history of diabetes and normal glycaemic parameters.

  • Due to the irreversible destruction of beta-cells, treatment with steroids is not indicated in contrast to other settings such as immunotherapy-induced hypophysitis.

  • Presence of low c-peptide levels post-acute presentation is indicative of an irreversible impact on beta-cell function and supports resuming immunotherapy given the significant benefits on cancer prognosis.

  • Clinicians must maintain a high index of suspicion in regards to diagnosis and management of new-onset type 1 diabetes and advice patients on reporting symptoms suggestive of diabetes and/or diabetes-related hyperglycaemic emergencies.

Open access
Toshitaka Sawamura Department of Internal Medicine, Asanogawa General Hospital, Kosakamachinaka, Kanazawa, Ishikawa, Japan
Department of Endocrinology and Metabolism, Kanazawa University Graduate School of Medicine, Takaramachi, Kanazawa, Japan
Department of Health Promotion and Medicine of the Future, Kanazawa University, Takaramachi, Kanazawa, Japan

Search for other papers by Toshitaka Sawamura in
Google Scholar
PubMed
Close
,
Shigehiro Karashima Department of Health Promotion and Medicine of the Future, Kanazawa University, Takaramachi, Kanazawa, Japan

Search for other papers by Shigehiro Karashima in
Google Scholar
PubMed
Close
,
Ai Ohmori Department of Internal Medicine, Asanogawa General Hospital, Kosakamachinaka, Kanazawa, Ishikawa, Japan
Department of Endocrinology and Metabolism, Kanazawa University Graduate School of Medicine, Takaramachi, Kanazawa, Japan

Search for other papers by Ai Ohmori in
Google Scholar
PubMed
Close
,
Kei Sawada Department of Internal Medicine, Asanogawa General Hospital, Kosakamachinaka, Kanazawa, Ishikawa, Japan

Search for other papers by Kei Sawada in
Google Scholar
PubMed
Close
,
Daisuke Aono Department of Endocrinology and Metabolism, Kanazawa University Graduate School of Medicine, Takaramachi, Kanazawa, Japan
Department of Health Promotion and Medicine of the Future, Kanazawa University, Takaramachi, Kanazawa, Japan

Search for other papers by Daisuke Aono in
Google Scholar
PubMed
Close
,
Mitsuhiro Kometani Department of Endocrinology and Metabolism, Kanazawa University Graduate School of Medicine, Takaramachi, Kanazawa, Japan
Department of Health Promotion and Medicine of the Future, Kanazawa University, Takaramachi, Kanazawa, Japan

Search for other papers by Mitsuhiro Kometani in
Google Scholar
PubMed
Close
,
Yoshiyu Takeda Department of Internal Medicine, Asanogawa General Hospital, Kosakamachinaka, Kanazawa, Ishikawa, Japan

Search for other papers by Yoshiyu Takeda in
Google Scholar
PubMed
Close
, and
Takashi Yoneda Department of Endocrinology and Metabolism, Kanazawa University Graduate School of Medicine, Takaramachi, Kanazawa, Japan
Department of Health Promotion and Medicine of the Future, Kanazawa University, Takaramachi, Kanazawa, Japan

Search for other papers by Takashi Yoneda in
Google Scholar
PubMed
Close

Summary

Fulminant type 1 diabetes (FT1D) is a subtype of diabetes characterized by rapid progression of β-cell destruction, hyperglycemia, and diabetic ketoacidosis (DKA). The pathogenesis of this disease remains unclear. However, viral infections, HLA genes, and immune checkpoint inhibitor use were reportedly involved in this disease. A 51-year-old Japanese man with no chronic medical condition was admitted to our hospital with complaints of nausea and vomiting. Cough, sore throat, nasal discharge, and diarrhea were not noted. He had a medical history of at least two influenza infections. His vaccination history was notable for receiving an inactive split influenza vaccine 12 days prior to developing these symptoms. He was diagnosed with DKA associated with FT1D. His HLA class II genotypes were nonsusceptible to FT1D, and he had a negative history of immune checkpoint inhibitor use. The destruction of the pancreas by cytotoxic T cells is reported to be involved in FT1D. Inactive split influenza vaccines do not directly activate cytotoxic T cells. However, these could activate the redifferentiation of memory CD8-positive T cells into cytotoxic T cells and induce FT1D, as this patient had a history of influenza infections.

Learning points

  • Influenza split vaccination could cause fulminant type 1 diabetes (FT1D).

  • The mechanism of influenza split vaccine-induced FT1D might be through the redifferentiation of CD8-positive memory T cells into cytotoxic T cells.

Open access
Valerie Lai Department of Medicine, University of Alberta, Edmonton, AB, Canada

Search for other papers by Valerie Lai in
Google Scholar
PubMed
Close
,
Mariam Shahidi Department of Medicine, University of Alberta, Edmonton, AB, Canada
Division of Endocrinology and Metabolism, University of Alberta, Edmonton, AB, Canada

Search for other papers by Mariam Shahidi in
Google Scholar
PubMed
Close
,
Alicia Chan Department of Medical Genetics, University of Alberta, Edmonton, AB, Canada

Search for other papers by Alicia Chan in
Google Scholar
PubMed
Close
, and
Shailly Jain-Ghai Department of Medical Genetics, University of Alberta, Edmonton, AB, Canada

Search for other papers by Shailly Jain-Ghai in
Google Scholar
PubMed
Close

Summary

3-hydroxy-3-methylglutaryl coenzyme A (HMG-CoA) lyase deficiency is an inborn error of metabolism resulting in a lack of ketogenesis and leucine catabolism. Hallmarks of decompensation include hypoglycemia without ketosis (or hypoketosis), metabolic acidosis, and hyperammonemia. Management includes avoiding fasting and restricting dietary protein and fat. Conversely, type 2 diabetes mellitus (T2DM) requires carbohydrate restriction and/or anti-hyperglycemic agents; thus, managing these co-existing disorders is challenging. A 36-year-old male with HMG-CoA lyase deficiency and T2DM (Hemoglobin A1c (HbA1c): 7.9%) presented with confusion and shock. Blood work revealed metabolic acidosis, hyperammonemia, hyperglycemia, and hypoketosis. The patient was diagnosed with hyperosmolar non-ketotic hyperglycemia and hyperammonemia secondary to HMG-CoA lyase metabolic decompensation requiring intensive care unit admission. Hyperammonemia management was challenging because alternative calories with i.v. dextrose (due to hyperglycemia) and i.v. lipids (due to HMG-CoA lyase deficiency) could not be provided as usual. The patient was started on hemodialysis and i.v. insulin with marked improvement. Once stabilized, metformin and insulin were initiated. T2DM impaired cellular glucose uptake and produced a state similar to hypoglycemia, despite the patient being profoundly hyperglycemic, which led to metabolic decompensation of HMG-CoA lyase deficiency. Managing T2DM and HMG-CoA lyase deficiency warrants special considerations due to the potential for metabolic decompensation with both hyperglycemia and hypoglycemia.

Learning points

  • In a patient with 3-hydroxy-3-methylglutaryl coenzyme A (HMG-CoA) lyase deficiency and type 2 diabetes mellitus (T2DM), management principles include avoiding hypoglycemia to prevent metabolic decompensation, providing insulin for proper glucose utilization, and moderation of carbohydrate intake to prevent consequences of chronic hyperglycemia.

  • The development of insulin resistance in the form of T2DM in HMG-CoA lyase deficiency likely triggered a state similar to hypoglycemia, leading to cellular energy deficiency and subsequently metabolic decompensation.

  • It is important to avoid hypoglycemia in patients with HMG-CoA lyase deficiency and T2DM, as the risk of metabolic decompensation is increased due to the lack of ketogenesis in HMG-CoA lyase deficiency.

  • Selection of antidiabetic agents in this patient population requires careful consideration, and agents that have a higher risk of hypoglycemia should be avoided.

Open access
Raad Alwithenani Department of Medicine, Division of Endocrinology and Metabolism, University of Toronto, Toronto, Ontario, Canada
College of Medicine, King Saud Bin Abdulaziz University for Health Sciences, Jeddah, Saudi Arabia
King Abdullah International Medical Research Center, Jeddah, Saudi Arabia
Department of Medicine, King Abdulaziz Medical City, Ministry of National Guard Health Affairs, Jeddah, Saudi Arabia

Search for other papers by Raad Alwithenani in
Google Scholar
PubMed
Close
,
Danielle M Andrade Department of Medicine, Division of Neurology, University of Toronto, Toronto, Ontario, Canada

Search for other papers by Danielle M Andrade in
Google Scholar
PubMed
Close
,
Lingxin Zhang Department of Laboratory Medicine and Pathobiology, University of Toronto, Toronto, Canada
Department of Pathology and Laboratory Medicine, Mount Sinai Hospital, Toronto, Ontario, Canada

Search for other papers by Lingxin Zhang in
Google Scholar
PubMed
Close
, and
Karen E Gomez-Hernandez Department of Medicine, Division of Endocrinology and Metabolism, University of Toronto, Toronto, Ontario, Canada

Search for other papers by Karen E Gomez-Hernandez in
Google Scholar
PubMed
Close

Summary

Myopathy caused by thyrotoxicosis is not uncommon. Skeletal muscles are commonly involved, but dysphagia is a rare manifestation of thyrotoxicosis. We aim to raise awareness of dysphagia caused by hyperthyroidism and review similar cases in the literature. We present a case of severe dysphagia caused by hyperthyroidism. We also summarize similar case reports in the literature. Our patient is a 77-year-old man who presented with thyrotoxicosis related to Graves’ disease (GD), dysphagia to both liquid and solid food, and weight loss. Further investigations revealed severe esophageal dysphagia and a high risk for aspiration. He required the placement of a G-tube for feeding. After 8 weeks of methimazole treatment, his thyroid function normalized and his dysphagia improved significantly, leading to the removal of the feeding G-tube. We summarize 19 case reports published in the literature of hyperthyroidism leading to dysphagia. Patients with thyrotoxicosis and dysphagia are at higher risk for aspiration pneumonia and thyroid storm. Based on previous case reports, on average, approximately 3 weeks of treatment with anti-thyroidal drugs and beta-blockers is needed before patients can eat normally. We report a case of dysphagia associated with GD, which is rare and needs prompt recognition to restore euthyroid status. Dysphagia generally resolved with normalization of thyroid function.

Learning points

  • Myopathy caused by thyrotoxicosis is not uncommon.

  • Skeletal muscles are commonly involved, but dysphagia is a rare manifestation of thyrotoxicosis.

  • Dysphagia due to hyperthyroidism resolves with normalization of thyroid function.

  • Early recognition of dysphagia related to hyperthyroidism and early initiation of therapy may help reverse the dysphagia and prevent complications.

Open access
George Brown Department of Hepatobiliary & Pancreatic Surgery, University Hospital Southampton, Southampton, UK

Search for other papers by George Brown in
Google Scholar
PubMed
Close
,
Anthony Mark Monaghan Department of Hepatobiliary & Pancreatic Surgery, University Hospital Southampton, Southampton, UK

Search for other papers by Anthony Mark Monaghan in
Google Scholar
PubMed
Close
,
Richard Fristedt Department of Hepatobiliary & Pancreatic Surgery, University Hospital Southampton, Southampton, UK

Search for other papers by Richard Fristedt in
Google Scholar
PubMed
Close
,
Emma Ramsey Department of Hepatobiliary & Pancreatic Surgery, University Hospital Southampton, Southampton, UK

Search for other papers by Emma Ramsey in
Google Scholar
PubMed
Close
,
Ma’en Al-Mrayat Department of Endocrinology, University Hospital Southampton, Southampton, UK

Search for other papers by Ma’en Al-Mrayat in
Google Scholar
PubMed
Close
,
Rushda Rajak Department of Cellular Pathology, University Hospital Southampton, Southampton, UK

Search for other papers by Rushda Rajak in
Google Scholar
PubMed
Close
,
Thomas Armstrong Department of Hepatobiliary & Pancreatic Surgery, University Hospital Southampton, Southampton, UK

Search for other papers by Thomas Armstrong in
Google Scholar
PubMed
Close
, and
Arjun Takhar Department of Hepatobiliary & Pancreatic Surgery, University Hospital Southampton, Southampton, UK

Search for other papers by Arjun Takhar in
Google Scholar
PubMed
Close

Summary

Vasoactive intestinal peptide-secreting tumours (VIPomas) are an extremely rare form of functional pancreatic neuroendocrine tumour with an estimated annual incidence of 1 in 10 million. Associated tumour hypersecretion of other peptides, including pancreatic polypeptide (PPomas), may also be seen. These malignancies classically present with a defined triad of refractory diarrhoea, hypokalaemia and metabolic acidosis known as Verner–Morrison syndrome. Diagnosis is frequently delayed, and the majority of patients will have metastatic disease at presentation. Symptoms are usually well controlled with somatostatin analogue administration. Here we report a case of metastatic mixed VIPoma/PPoma-induced diarrhoea causing renal failure so severe that ultrafiltration was required to recover adequate renal function.

Learning points

  • Profuse, watery diarrhoea is a common presenting complaint with a multitude of aetiologies. This, combined with the rarity of these tumours, makes diagnosis difficult and frequently delayed. A functional neuroendocrine tumour should be suspected when diarrhoea is unusually extreme, prolonged and common causes have been promptly excluded.

  • These patients are likely to be profoundly unwell on presentation. They are extremely hypovolaemic with dangerous electrolyte and metabolic abnormalities. Aggressive initial rehydration and electrolyte replacement are imperative. A somatostatin analogue should be commenced as soon as the diagnosis is suspected.

  • This is an extreme example of Verner–Morrison syndrome. We are unaware of another case where renal failure secondary to diarrhoea and dehydration was so severe that renal replacement therapy was required to restore adequate renal function, further emphasising how critically unwell these patients can be.

  • Both the primary tumour and metastases showed a remarkably good and rapid response to somatostatin analogue administration. Cystic change and involution were noted on repeat imaging within days.

  • Prior to his illness, this patient was extremely high functioning with no medical history. His diagnosis was an enormous psychological shock, and the consideration and care for his psychological well-being were a crucial part of his overall management. It highlights the importance of a holistic approach to cancer care and the role of the clinical nurse specialist within the cancer multidisciplinary team.

Open access