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      Fulminant hepatitis and elevated levels of sIL-2R in thyroid storm

      research-article
      1 , 1 , 1 , 1 , 1 , 1
      Endocrinology, Diabetes & Metabolism Case Reports
      Bioscientifica Ltd
      Adult, Male, Asian - Japanese, Japan, Heart, Liver, Thyroid, Thyroxine (T4), Triiodothyronine (T3), TSH, Liver failure, Fulminant hepatitis*, Thyroid storm, Graves' disease, Fatigue, Pyrexia, Breathing difficulties, Dyspnoea, Weight loss, Tachycardia, Heart failure, Atrial fibrillation, Hyperthyroidism, Cardiogenic shock, Cardiomegaly, Liver dysfunction*, Goitre, Hyperhidrosis, Oedema, Hypoglycaemia, Renal failure, Coagulopathy, Hyperammonemia*, Jaundice, Hypoxia, Metabolic acidosis, Soluble IL-2 receptor*, Alanine aminotransferase, Aspartate transaminase, FT4, CT scan, Bilirubin, Transaminase, X-ray, Echocardiogram, Albumin, Lactate dehydrogenase, Alkaline phosphatase, Glucose (blood), Creatinine, Calcium (serum), C-reactive protein, Potassium, Ammonia, Brain natriuretic peptide, FT3, TSH, Thyroid antibodies, Prothrombin time, Plasma exchange, Dialysis, Intra-aortic balloon pumping*, Methimazole, Potassium iodide, Beta-blockers, Hydrocortisone, Glucocorticoids, Landiolol hydrochloride*, Glucose, Propylthiouracil, Cardiology, Gastroenterology, Unique/unexpected symptoms or presentations of a disease, September, 2019

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          Summary

          We report the case of a 48-year-old man with thyroid storm associated with fulminant hepatitis and elevated levels of soluble interleukin-2 receptor (sIL-2R). Fatigue, low-grade fever, shortness of breath, and weight loss developed over several months. The patient was admitted to the hospital because of tachycardia-induced heart failure and liver dysfunction. Graves’ disease with heart failure was diagnosed. He was treated with methimazole, inorganic iodide, and a β-blocker. On the day after admission, he became unconscious with a high fever and was transferred to the intensive care unit. Cardiogenic shock with atrial flutter was treated with intra-aortic balloon pumping and cardioversion. Hyperthyroidism decreased over 10 days, but hepatic failure developed. He was diagnosed with thyroid storm accompanied by fulminant hepatitis. Laboratory investigations revealed elevated levels of sIL-2R (9770 U/mL). The fulminant hepatitis was refractory to plasma exchange and plasma filtration with dialysis, and no donors for liver transplantation were available. He died of hemoperitoneum and gastrointestinal hemorrhage due to fulminant hepatitis 62 days after admission. Elevated circulating levels of sIL-2R might be a marker of poor prognosis in thyroid storm with fulminant hepatitis.

          Learning points:
          • The prognosis of thyroid storm when fulminant hepatitis occurs is poor.

          • Liver transplantation is the preferred treatment for fulminant hepatitis induced by thyroid storm refractory to plasma exchange.

          • Elevated levels of soluble interleukin-2 receptor might be a marker of poor prognosis in patients with thyroid storm.

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          Most cited references6

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          The relationship between the thyroid gland and the liver.

          Thyroxine and tri-iodothyronine are essential for normal organ growth, development and function. These hormones regulate the basal metabolic rate of all cells, including hepatocytes, and thereby modulate hepatic function; the liver in turn metabolizes the thyroid hormones and regulates their systemic endocrine effects. Thyroid dysfunction may perturb liver function, liver disease modulates thyroid hormone metabolism, and a variety of systemic diseases affect both organs. We highlight the intricate relations between the thyroid gland and the liver in health and disease.
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            Thyroid Storm: A Japanese Perspective

            Background: Thyroid storm (TS) is life threatening. In the mid-2000s, its incidence was poorly defined, peer-reviewed diagnostic criteria were not available, and management and treatment did not seem to be verified based upon evidence and latest advances in medicine. Methods: First, diagnostic criteria were developed based on 99 patients in the literature and seven patients in this study. Then, initial and follow-up surveys were conducted from 2004 through 2008, targeting all hospitals in Japan to obtain and verify information on patients who met diagnostic criteria for TS. Based on these data, the diagnostic criteria were revised, and management and treatment guidelines were created. Results: The incidence of TS in hospitalized patients in Japan was estimated to be 0.20 per 100,000 per year and 0.22% of all thyrotoxic patients. The mortality rate was 10.7%. Multiple organ failure was the most common cause of death, followed by congestive heart failure, respiratory failure, and arrhythmia. In the final diagnostic criteria for TS, the definition of jaundice as serum bilirubin concentration >3 mg/dL was added. Based upon nationwide surveys and the latest information, guidelines for the management and treatment for TS were extensively revised and algorithms were developed. Conclusions: TS remains a life-threatening disorder, with >10% mortality in Japan. New peer-reviewed diagnostic criteria for TS are presented and its clinical features, prognosis, and incidence are clarified based on nationwide surveys. Furthermore, this information helped to establish detailed guidelines for the management and treatment of TS. A prospective prognostic study to validate the guidelines is eagerly anticipated.
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              Role of plasma exchange in the thyroid storm.

              Inadequately treated thyroid storm can lead to death. Therapeutic plasma exchange (TPE) is a suggested treatment when conventional treatments fail, but its indication is not well codified. We report our experience through three explicit cases. Three elderly patients were admitted to our hospital for cardiac or neurologic symptoms due to thyroid storm. After initiation of conventional therapy, TPE was performed with clinical and biological improvement. The speed of symptom resolution varies depending on the severity. This technique must be carried out by experienced medical staff as many complications can occur; nevertheless, in our patients with severe comorbidities, no complications occurred. The action of TPE mainly results from plasma removal of cytokines, putative antibodies, and thyroid hormones and their bound proteins. TPE has a transitory effect and thus should be associated with other thyroid blockers. When there are threatening symptoms, TPE should be done early, without waiting for the efficiency of conventional treatment, since it is the fastest method known for the improvement of the clinical condition. We also suggest starting TPE in case of neurologic symptoms because of very slow and incomplete regression. The Burch and Wartofsky score seems to be a helpful tool in establishing the diagnosis of thyroid storm and for deciding on when to initiate TPE.

                Author and article information

                Journal
                Endocrinol Diabetes Metab Case Rep
                Endocrinol Diabetes Metab Case Rep
                EDM
                Endocrinology, Diabetes & Metabolism Case Reports
                Bioscientifica Ltd (Bristol )
                2052-0573
                06 September 2019
                2019
                : 2019
                : 19-0078
                Affiliations
                [1 ]Division of Neurology , Respirology, Endocrinology and Metabolism, Department of Internal Medicine, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan
                Author notes
                Correspondence should be addressed to H Yamaguchi; Email: yamahide@ 123456med.miyazaki-u.ac.jp

                *(Y Tanaka and T Uchida contributed equally to this work)

                Article
                EDM190078
                10.1530/EDM-19-0078
                6765318
                c8a840c4-f931-47f5-a392-141356f05105
                © 2019 The authors

                This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License..

                History
                : 21 August 2019
                : 06 September 2019
                Categories
                Adult
                Male
                Asian - Japanese
                Japan
                Heart
                Liver
                Thyroid
                Thyroxine (T4)
                Triiodothyronine (T3)
                TSH
                Liver failure
                Fulminant hepatitis*
                Thyroid storm
                Graves' disease
                Fatigue
                Pyrexia
                Breathing difficulties
                Dyspnoea
                Weight loss
                Tachycardia
                Heart failure
                Atrial fibrillation
                Hyperthyroidism
                Cardiogenic shock
                Cardiomegaly
                Liver dysfunction*
                Goitre
                Hyperhidrosis
                Oedema
                Hypoglycaemia
                Renal failure
                Coagulopathy
                Hyperammonemia*
                Jaundice
                Hypoxia
                Metabolic acidosis
                Soluble IL-2 receptor*
                Alanine aminotransferase
                Aspartate transaminase
                FT4
                CT scan
                Bilirubin
                Transaminase
                X-ray
                Echocardiogram
                Albumin
                Lactate dehydrogenase
                Alkaline phosphatase
                Glucose (blood)
                Creatinine
                Calcium (serum)
                C-reactive protein
                Potassium
                Ammonia
                Brain natriuretic peptide
                FT3
                TSH
                Thyroid antibodies
                Prothrombin time
                Plasma exchange
                Dialysis
                Intra-aortic balloon pumping*
                Methimazole
                Potassium iodide
                Beta-blockers
                Hydrocortisone
                Glucocorticoids
                Landiolol hydrochloride*
                Glucose
                Propylthiouracil
                Cardiology
                Gastroenterology
                Unique/Unexpected Symptoms or Presentations of a Disease
                Unique/Unexpected Symptoms or Presentations of a Disease

                adult,male,asian - japanese,japan,heart,liver,thyroid,thyroxine (t4),triiodothyronine (t3),tsh,liver failure,fulminant hepatitis*,thyroid storm,graves' disease,fatigue,pyrexia,breathing difficulties,dyspnoea,weight loss,tachycardia,heart failure,atrial fibrillation,hyperthyroidism,cardiogenic shock,cardiomegaly,liver dysfunction*,goitre,hyperhidrosis,oedema,hypoglycaemia,renal failure,coagulopathy,hyperammonemia*,jaundice,hypoxia,metabolic acidosis,soluble il-2 receptor*,alanine aminotransferase,aspartate transaminase,ft4,ct scan,bilirubin,transaminase,x-ray,echocardiogram,albumin,lactate dehydrogenase,alkaline phosphatase,glucose (blood),creatinine,calcium (serum),c-reactive protein,potassium,ammonia,brain natriuretic peptide,ft3,thyroid antibodies,prothrombin time,plasma exchange,dialysis,intra-aortic balloon pumping*,methimazole,potassium iodide,beta-blockers,hydrocortisone,glucocorticoids,landiolol hydrochloride*,glucose,propylthiouracil,cardiology,gastroenterology,unique/unexpected symptoms or presentations of a disease,september,2019

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