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Tomomi Nakao First Department of Internal Medicine, Wakayama Medical University, Wakayama City, Wakayama, Japan

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Ken Takeshima First Department of Internal Medicine, Wakayama Medical University, Wakayama City, Wakayama, Japan

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Hiroyuki Ariyasu First Department of Internal Medicine, Wakayama Medical University, Wakayama City, Wakayama, Japan

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Chiaki Kurimoto First Department of Internal Medicine, Wakayama Medical University, Wakayama City, Wakayama, Japan

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Shinsuke Uraki First Department of Internal Medicine, Wakayama Medical University, Wakayama City, Wakayama, Japan

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Shuhei Morita First Department of Internal Medicine, Wakayama Medical University, Wakayama City, Wakayama, Japan

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Yasushi Furukawa First Department of Internal Medicine, Wakayama Medical University, Wakayama City, Wakayama, Japan

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Hiroshi Iwakura First Department of Internal Medicine, Wakayama Medical University, Wakayama City, Wakayama, Japan

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Takashi Akamizu First Department of Internal Medicine, Wakayama Medical University, Wakayama City, Wakayama, Japan

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Summary

Thyroid storm (TS) is a life-threatening condition that may suffer thyrotoxic patients. Therapeutic plasma exchange (TPE) is a rescue approach for TS with acute hepatic failure, but it should be initiated with careful considerations. We present a 55-year-old male patient with untreated Graves’ disease who developed TS. Severe hyperthyroidism and refractory atrial fibrillation with congestive heart failure aggregated to multiple organ failure. The patient was recovered by intensive multimodal therapy, but we had difficulty in introducing TPE treatment considering the risk of exacerbation of congestive heart failure due to plasma volume overload. In addition, serum total bilirubin level was not elevated in the early phase to the level of indication for TPE. The clinical course of this patient instructed delayed elevation of bilirubin until the level of indication for TPE in some patients and also demonstrated the risk of exacerbation of congestive heart failure by TPE.

Learning points:

  • Our patient with thyroid storm could be diagnosed and treated promptly using Japan Thyroid Association guidelines for thyroid storm.

  • Delayed elevation of serum bilirubin levels could make the decision of introducing therapeutic plasma exchange difficult in cases of thyroid storm with acute hepatic failure.

  • The risk of worsening congestive heart failure should be considered carefully when performing therapeutic plasma exchange.

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Fumiaki Kawano Departments of Surgery, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

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Tadato Yonekawa Neurology, Respirology, Endocrinology and Metabolism, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

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Hideki Yamaguchi Neurology, Respirology, Endocrinology and Metabolism, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

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Nobuhiro Shibata Clinical Oncology, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

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Kousei Tashiro Departments of Surgery, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

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Makoto Ikenoue Departments of Surgery, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

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Shun Munakata Departments of Surgery, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

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Kazuhiro Higuchi Departments of Surgery, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

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Hiroyuki Tanaka Diagnostic Pathology, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

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Yuichiro Sato Diagnostic Pathology, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

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Ayumu Hosokawa Clinical Oncology, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

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Shinsuke Takeno Departments of Surgery, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

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Kunihide Nakamura Departments of Surgery, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

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Atsushi Nanashima Departments of Surgery, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

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Summary

A 54-year-old woman was referred to our hospital with a cervical tumor. CT revealed a cervical tumor extending to the upper mediastinum, tracheal deviation and tumor infiltration in the cervical vessels. She was followed-up because no diagnosis of malignancy was made by cytology. However, 2 months later, a CT scan showed enlargement of the tumor and tracheal stenosis, and a surgical biopsy was performed and she was diagnosed with anaplastic thyroid cancer (ATC). The tracheal tube with tracheal stenosis could not be removed due to the rapid growth of the tumor, necessitating management by mechanical ventilation. Due to the difficulty of surgical resection, she was treated with lenvatinib. A lenvatinib solution was made and administered via a nasogastric tube. After lenvatinib treatment, the tumor volume decreased and the tracheal stenosis improved. The tracheal tube was removed and oral intake became possible. She was discharged and received ambulatory lenvatinib therapy. The tumor was significantly reduced in size, but gradually grew and was exposed through the cervical wound 6 months later. Esophageal perforation occurred 10 months after the start of treatment. Lenvatinib was re-administered via a nasogastric tube. Eleven months later, the patient died of massive bleeding from the exposed cervical tumor. Patients with advanced ATC may require management with mechanical ventilation for airway stenosis or with a nasogastric tube for esophageal stenosis and perforation. We experienced a case in which lenvatinib was safely administered via a nasogastric tube while performing mechanical ventilation.

Learning points:

  • An anaplastic thyroid cancer patient under mechanical ventilator management was treated with lenvatinib via a nasogastric tube.

  • The lenvatinib solution can easily be prepared and administered via a nasogastric tube.

  • The lenvatinib solution was effective for a patient with difficulty in oral intake.

  • Lenvatinib could also improve the prognosis of an anaplastic thyroid cancer patient with severe airway and esophageal trouble.

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Agnieszka Łebkowska Department of Internal Medicine and Metabolic Diseases, Diabetology and Internal Medicine

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Anna Krentowska Department of Internal Medicine and Metabolic Diseases, Diabetology and Internal Medicine

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Agnieszka Adamska Department of Endocrinology, Diabetology and Internal Medicine

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Danuta Lipińska Department of Endocrinology, Diabetology and Internal Medicine

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Beata Piasecka Department of Endocrinology, Diabetology and Internal Medicine

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Otylia Kowal-Bielecka Department of Rheumatology and Internal Diseases, Medical University of Bialystok, Bialystok, Poland

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Maria Górska Department of Endocrinology, Diabetology and Internal Medicine

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Robert K Semple Centre for Cardiovascular Science, Queen’s Medical Research Institute, University of Edinburgh, Edinburgh, UK

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Irina Kowalska Department of Internal Medicine and Metabolic Diseases, Diabetology and Internal Medicine

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Summary

Type B insulin resistance syndrome (TBIR) is characterised by the rapid onset of severe insulin resistance due to circulating anti-insulin receptor antibodies (AIRAs). Widespread acanthosis nigricans is normally seen, and co-occurrence with other autoimmune diseases is common. We report a 27-year-old Caucasian man with psoriasis and connective tissue disease who presented with unexplained rapid weight loss, severe acanthosis nigricans, and hyperglycaemia punctuated by fasting hypoglycaemia. Severe insulin resistance was confirmed by hyperinsulinaemic euglycaemic clamping, and immunoprecipitation assay demonstrated AIRAs, confirming TBIR. Treatment with corticosteroids, metformin and hydroxychloroquine allowed withdrawal of insulin therapy, with stabilisation of glycaemia and diminished signs of insulin resistance; however, morning fasting hypoglycaemic episodes persisted. Over three years of follow-up, metabolic control remained satisfactory on a regimen of metformin, hydroxychloroquine and methotrexate; however, psoriatic arthritis developed. This case illustrates TBIR as a rare but severe form of acquired insulin resistance and describes an effective multidisciplinary approach to treatment.

Learning points:

  • We describe an unusual case of type B insulin resistance syndrome (TBIR) in association with mixed connective tissue disease and psoriasis.

  • Clinical evidence of severe insulin resistance was corroborated by euglycaemic hyperinsulinaemic clamp, and anti-insulin receptor autoantibodies were confirmed by immunoprecipitation assay.

  • Treatment with metformin, hydroxychloroquine and methotrexate ameliorated extreme insulin resistance.

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Anna Luiza Galeazzi Rech Kantonsspital Sankt Gallen, Klinik für Allgemeine Innere Medizin/Hausarztmedizin, Sankt Gallen, Switzerland

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Yvon Stüve Kantonsspital Sankt Gallen, Klinik für Allgemeine Innere Medizin/Hausarztmedizin, Sankt Gallen, Switzerland

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Andreas Toepfer Kantonsspital Sankt Gallen, Klinik für Orthopädische Chirurgie und Traumatologie des Bewegungsapparts, Sankt Gallen, Switzerland

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Katrin E Schimke Kantonsspital Sankt Gallen, Klinik für Allgemeine Innere Medizin/Hausarztmedizin, Sankt Gallen, Switzerland

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Summary

Acute Charcot neuropathic osteoarthropathy (CN) is a clinical entity which can easily go unrecognized in its acute early stages due to lack of awareness and unspecific presentation. However, missing early diagnosis can lead to severe complications. We present the case of a 72-year-old male patient who went through the natural course of the disease unnoticed before the very eyes of his physicians leading to a tragic end. We aim to raise awareness for this rare diabetic complication, emphasizing the necessity of early diagnosis and adequate, interdisciplinary treatment.

Learning points:

  • Clinical signs and symptoms of acute Charcot neuropathic osteoarthropathy (CN).

  • Red flags.

  • Importance of early diagnosis and correct treatment.

  • Diagnostic challenges of acute CN.

  • Awareness of high morbidity and mortality.

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Waralee Chatchomchaun Diabetes and Thyroid Center, Theptarin Hospital, Bangkok, Thailand

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Yotsapon Thewjitcharoen Diabetes and Thyroid Center, Theptarin Hospital, Bangkok, Thailand

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Karndumri Krittadhee Diabetes and Thyroid Center, Theptarin Hospital, Bangkok, Thailand

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Veekij Veerasomboonsin Diabetes and Thyroid Center, Theptarin Hospital, Bangkok, Thailand

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Soontaree Nakasatien Diabetes and Thyroid Center, Theptarin Hospital, Bangkok, Thailand

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Sirinate Krittiyawong Diabetes and Thyroid Center, Theptarin Hospital, Bangkok, Thailand

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Sriurai Porramatikul Diabetes and Thyroid Center, Theptarin Hospital, Bangkok, Thailand

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Ekgaluck Wanathayanoroj Diabetes and Thyroid Center, Theptarin Hospital, Bangkok, Thailand

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Auchai Kanchanapituk Diabetes and Thyroid Center, Theptarin Hospital, Bangkok, Thailand

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Pairoj Junyangdikul Department of Pathology, Samitivej Srinakarin Hospital, Bangkok Hospital Group, Bangkok, Thailand

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Thep Himathongkam Diabetes and Thyroid Center, Theptarin Hospital, Bangkok, Thailand

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Summary

In this case report, we describe a 37-year-old male who presented with fever and tender neck mass. Neck ultrasonography revealed a mixed echogenic multiloculated solid-cystic lesion containing turbid fluid and occupying the right thyroid region. Thyroid function tests showed subclinical hyperthyroidism. The patient was initially diagnosed with thyroid abscess and he was subsequently treated with percutaneous aspiration and i.v. antibiotics; however, his clinical symptoms did not improve. Surgical treatment was then performed and a pathological examination revealed a ruptured epidermoid cyst with abscess formation. No thyroid tissue was identified in the specimen. The patient was discharged uneventfully. However, at the 3-month and 1-year follow-ups, the patient was discovered to have developed subclinical hypothyroidism. Neck ultrasonography revealed a normal thyroid gland. This report demonstrates a rare case of epidermoid cyst abscess in the cervical region, of which initial imaging and abnormal thyroid function tests led to the erroneous diagnosis of thyroid abscess.

Learning points:

  • Epidermoid cyst abscess at the cervical region can mimic thyroid abscess.

  • Neck ultrasonography cannot distinguish thyroid abscess from epidermoid cyst abscess.

  • Thyroid function may be altered due to the adjacent soft tissue inflammation.

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Aishah Ekhzaimy Department of Medicine and College of Medicine, King Saud University, Riyadh, Saudi Arabia

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Afshan Masood Obesity Research Center, and College of Medicine, King Saud University, Riyadh, Saudi Arabia

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Seham Alzahrani Department of Medicine and College of Medicine, King Saud University, Riyadh, Saudi Arabia

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Waleed Al-Ghamdi Department of Medicine and College of Medicine, King Saud University, Riyadh, Saudi Arabia

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Daad Alotaibi Department of Medicine and College of Medicine, King Saud University, Riyadh, Saudi Arabia

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Muhammad Mujammami Department of Medicine and College of Medicine, King Saud University, Riyadh, Saudi Arabia

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Summary

Central diabetes insipidus (CDI) and several endocrine disorders previously classified as idiopathic are now considered to be of an autoimmune etiology. Dermatomyositis (DM), a rare autoimmune condition characterized by inflammatory myopathy and skin rashes, is also known to affect the gastrointestinal, pulmonary, and rarely the cardiac systems and the joints. The association of CDI and DM is extremely rare. After an extensive literature search and to the best of our knowledge this is the first reported case in literature, we report the case of a 36-year-old male with a history of CDI, who presented to the hospital’s endocrine outpatient clinic for evaluation of a 3-week history of progressive facial rash accompanied by weakness and aching of the muscles.

Learning points:

  • Accurate biochemical diagnosis should always be followed by etiological investigation.

  • This clinical entity usually constitutes a therapeutic challenge, often requiring a multidisciplinary approach for optimal outcome.

  • Dermatomyositis is an important differential diagnosis in patients presenting with proximal muscle weakness.

  • Associated autoimmune conditions should be considered while evaluating patients with dermatomyositis.

  • Dermatomyositis can relapse at any stage, even following a very long period of remission.

  • Maintenance immunosuppressive therapy should be carefully considered in these patients.

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Maria Tomkins Department of Endocrinology and Diabetes, Beaumont Hospital Dublin, Dublin, Ireland

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Roxana Maria Tudor Department of Endocrinology and Diabetes, Beaumont Hospital Dublin, Dublin, Ireland

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Diarmuid Smith Department of Endocrinology and Diabetes, Beaumont Hospital Dublin, Dublin, Ireland

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Amar Agha Department of Endocrinology and Diabetes, Beaumont Hospital Dublin, Dublin, Ireland

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Summary

This case is the first to describe a patient who experienced concomitant agranulocytosis and anti-neutrophil cytoplasmic antibody (ANCA)-associated vasculitis as an adverse effect of propylthiouracil treatment for Graves’ disease. A 42-year-old female with Graves’ disease presented to the emergency department (ED) with a 2-week history of fevers, night sweats, transient lower limb rash, arthralgia, myalgia and fatigue. She had been taking propylthiouracil for 18 months prior to presentation. On admission, agranulocytosis was evident with a neutrophil count of 0.36 × 109/L and immediately propylthiouracil was stopped. There was no evidence of active infection and the patient was treated with broad-spectrum antibodies and one dose of granulocyte colony-stimulation factor, resulting in a satisfactory response. On further investigation, ANCAs were positive with dual positivity for proteinase 3 and myeloperoxidase. There was no evidence of end-organ damage secondary to vasculitis, and the patient’s constitutional symptoms resolved completely on discontinuation of the drug precluding the need for immunosuppressive therapy.

Learning points:

  • Continued vigilance and patient education regarding the risk of antithyroid drug-induced agranulocytosis is vital throughout the course of treatment.

  • ANCA-associated vasculitis is a rare adverse effect of antithyroid drug use.

  • Timely discontinuation of the offending drug is vital in reducing end-organ damage and the need for immunosuppressive therapy in drug-induced ANCA-associated vasculitis.

  • Similarities in the pathogenesis of agranulocytosis and drug-induced ANCA-associated vasculitis may offer insight into an improved understanding of vasculitis and agranulocytosis.

Open access
Masato Kotani Center for Diabetes, Endocrinology and Metabolism
Research Support Center, Shizuoka General Hospital, Shizuoka, Shizuoka, Japan
Asahina Shinryoujo, Fujieda, Shizuoka, Japan

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Naohisa Tamura Center for Diabetes, Endocrinology and Metabolism
Research Support Center, Shizuoka General Hospital, Shizuoka, Shizuoka, Japan

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Tatsuhide Inoue Center for Diabetes, Endocrinology and Metabolism

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Issei Tanaka Center for Diabetes, Endocrinology and Metabolism

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Summary

Type B insulin resistance syndrome is characterized by the presence of autoantibodies to the insulin receptor. We present a 57-year-old male admitted to a hospital due to body weight loss of 16 kg and hyperglycemia of 13.6 mmol/L. He was diagnosed with type B insulin resistance syndrome because the anti-insulin receptor antibodies were positive. We informed him that some hyperglycemic cases of this syndrome had been reported to be spontaneously remitted in 5 years, and he did not agree to be treated with high-dose glucocorticoids and/or immunosuppressive agents due to his concern for their adverse effects such as hyperglycemia and immunosuppression. He chose to be treated with insulin and voglibose, but fair glucose control could not be obtained. Six years later, he agreed to be treated with low-dose glucocorticoids practicable in outpatient settings. One milligram per day of betamethasone was tried orally and reduced gradually according to the values of glycated hemoglobin. After 30 months of glucocorticoid treatment, the anti-insulin receptor antibodies became undetectable and his fasting plasma glucose and glycated hemoglobin were normalized. This case suggests that low-dose glucocorticoids could be a choice to treat type B insulin resistance syndrome in outpatient settings.

Learning points:

  • Type B insulin resistance syndrome is an acquired autoimmune disease for insulin receptors.

  • This case suggested the possibility of long-lasting, low-dose glucocorticoid therapy for the syndrome as an alternative for high-dose glucocorticoids or immunosuppressive agents.

  • Since the prevalence of autoimmune nephritis is high in the syndrome, a delay of immunosuppressive therapy initiation might result in an exacerbation of nephropathy.

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

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

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

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

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

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

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

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Summary

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

Learning points:

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

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

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

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

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Huilin Koh Department of Endocrinology, Singapore General Hospital, Singapore, Singapore

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Manish Kaushik Department of Renal Medicine, Singapore General Hospital, Singapore, Singapore

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Julian Kenrick Loh Department of Cardiology, National Heart Centre Singapore, Singapore, Singapore

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Chiaw Ling Chng Department of Endocrinology, Singapore General Hospital, Singapore, Singapore

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Summary

Thyroid storm with multi-organ failure limits the use of conventional treatment. A 44-year-old male presented with thyroid storm and experienced cardiovascular collapse after beta-blocker administration, with resultant fulminant multi-organ failure requiring inotropic support, mechanical ventilation, extracorporeal membrane oxygenation (ECMO) and continuous renal replacement therapy. Hepatic and renal failure precluded the use of conventional thyroid storm treatment and early plasma exchange was instituted. The patient underwent emergency thyroidectomy after four effective exchanges, with subsequent rapid reversal of multi-organ failure. The challenges of institution of plasma exchanges with ongoing ECMO support, dialysis and timing of thyroidectomy are discussed. This case highlights the important role of early therapeutic plasma exchange (TPE) as an effective salvage therapy for lowering circulating hormones and stabilization of patients in preparation for emergency thyroidectomy in patients with thyroid storm and fulminant multi-organ failure.

Learning points:

  • Administration of beta-blockers in thyroid storm presenting with congestive cardiac failure may precipitate cardiovascular collapse due to inhibition of thyroid-induced hyperadrenergic compensation which maintains cardiac output.

  • TPE can be an effective bridging therapy to emergency total thyroidectomy when conventional thyroid storm treatment is contraindicated.

  • End-organ support using ECMO and CRRT can be combined with TPE effectively in the management of critically ill cases of thyroid storm.

  • The effectiveness of plasma exchange in lowering thyroid hormones appears to wane after 44–48 h of therapy in this case, highlighting the importance early thyroidectomy.

Open access