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      Surgical Management of an Atypical Presentation of a Thyroid Storm

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          Thyroid storm is a rare complication of Graves' disease that can carry a poor prognosis. In order to prevent major complications, thyroid storm must be quickly identified in patients and treatment must be promptly implemented. Medical treatment is usually initiated with antithyroid medications, such as propylthiouracil (PTU), methimazole, and beta-blockers. However, some patients may experience adverse reactions to these medications and alternate treatment options must be explored.

          Case Presentation:

          We report a case of a 30-year-old female initiated on PTU after diagnosis with Graves' disease that later presented an acute thyroid storm.


          Therapy was changed to methimazole, yet the patient subsequently developed angioedema and dyspnea. Medical management was discontinued and emergent thyroidectomy was performed without complication.

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          Most cited references 19

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          Hyperthyroidism and other causes of thyrotoxicosis: management guidelines of the American Thyroid Association and American Association of Clinical Endocrinologists.

          Thyrotoxicosis has multiple etiologies, manifestations, and potential therapies. Appropriate treatment requires an accurate diagnosis and is influenced by coexisting medical conditions and patient preference. This article describes evidence-based clinical guidelines for the management of thyrotoxicosis that would be useful to generalist and subspeciality physicians and others providing care for patients with this condition. The development of these guidelines was commissioned by the American Thyroid Association in association with the American Association of Clinical Endocrinologists. The American Thyroid Association and American Association of Clinical Endocrinologists assembled a task force of expert clinicians who authored this report. The task force examined relevant literature using a systematic PubMed search supplemented with additional published materials. An evidence-based medicine approach that incorporated the knowledge and experience of the panel was used to develop the text and a series of specific recommendations. The strength of the recommendations and the quality of evidence supporting each was rated according to the approach recommended by the Grading of Recommendations, Assessment, Development, and Evaluation Group. Clinical topics addressed include the initial evaluation and management of thyrotoxicosis; management of Graves' hyperthyroidism using radioactive iodine, antithyroid drugs, or surgery; management of toxic multinodular goiter or toxic adenoma using radioactive iodine or surgery; Graves' disease in children, adolescents, or pregnant patients; subclinical hyperthyroidism; hyperthyroidism in patients with Graves' ophthalmopathy; and management of other miscellaneous causes of thyrotoxicosis. One hundred evidence-based recommendations were developed to aid in the care of patients with thyrotoxicosis and to share what the task force believes is current, rational, and optimal medical practice.
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            Life-threatening thyrotoxicosis. Thyroid storm.

            Although important strides in recognition and therapy have significantly reduced the mortality in this disorder from the nearly 100% fatality rate noted by Lahey, survival is by no means guaranteed. More recent series have yielded fatality rates between 20% and 50%. Although some authors have attributed this improvement, in part, to a relaxation of the diagnostic criteria for thyroid storm, it more likely represents improvements in early recognition and the beneficial effects of the serial addition of antithyroid, corticosteroid, and antiadrenergic therapies to the treatment of this disorder. Thyroid storm is a dreaded, fortunately rare complication of a very common disorder. Most cases of thyroid storm occur following a precipitating event or intercurrent illness. Effective management is predicated on a prompt recognition of impending thyroid storm which is, in turn, dependent on a thorough knowledge of both the typical and atypical presentations of this disorder. An unwavering commitment to an aggressive, multifaceted therapeutic intervention as outlined herein is critical to the obtainment of a satisfactory outcome.
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              Diagnostic criteria, clinical features, and incidence of thyroid storm based on nationwide surveys.

              Thyroid storm (TS) is life threatening. Its incidence is poorly defined, few series are available, and population-based diagnostic criteria have not been established. We surveyed TS in Japan, defined its characteristics, and formulated diagnostic criteria, FINAL-CRITERIA1 and FINAL-CRITERIA2, for two grades of TS, TS1, and TS2 respectively. We first developed diagnostic criteria based on 99 patients in the literature and 7 of our patients (LIT-CRITERIA1 for TS1 and LIT-CRITERIA2 for TS2). Thyrotoxicosis was a prerequisite for TS1 and TS2 as well as for combinations of the central nervous system manifestations, fever, tachycardia, congestive heart failure (CHF), and gastrointestinal (GI)/hepatic disturbances. We then conducted initial and follow-up surveys from 2004 through 2008, targeting all hospitals in Japan, with an eight-layered random extraction selection process to obtain and verify information on patients who met LIT-CRITERIA1 and LIT-CRITERIA2. We identified 282 patients with TS1 and 74 patients with TS2. Based on these data and information from the Ministry of Health, Labor, and Welfare of Japan, we estimated the incidence of TS in hospitalized patients in Japan to be 0.20 per 100,000 per year. Serum-free thyroxine and free triiodothyroine concentrations were similar among patients with TS in the literature, Japanese patients with TS1 or TS2, and a group of patients with thyrotoxicosis without TS (Tox-NoTS). The mortality rate was 11.0% in TS1, 9.5% in TS2, and 0% in Tox-NoTS patients. Multiple organ failure was the most common cause of death in TS1 and TS2, followed by CHF, respiratory failure, arrhythmia, disseminated intravascular coagulation, GI perforation, hypoxic brain syndrome, and sepsis. Glasgow Coma Scale results and blood urea nitrogen (BUN) were associated with irreversible damages in 22 survivors. The only change in our final diagnostic criteria for TS as compared with our initial criteria related to serum bilirubin concentration >3 mg/dL. TS is still a life-threatening disorder with more than 10% mortality in Japan. We present newly formulated diagnostic criteria for TS and clarify its clinical features, prognosis, and incidence based on nationwide surveys in Japan. This information will help diagnose TS and in understanding the factors contributing to mortality and irreversible complications.

                Author and article information

                Int J Endocrinol Metab
                Int J Endocrinol Metab
                International Journal of Endocrinology and Metabolism
                01 April 2014
                April 2014
                : 12
                : 2
                [1 ]Tufts University School of Medicine, Boston, USA
                [2 ]Department of Otolaryngology-HNS, Tufts Medical Center, Boston, USA
                [3 ]Department of Endocrinology and Diabetes, Tufts Medical Center, Boston, USA
                Author notes
                [* ]Corresponding author: Richard O. Wein, Otolaryngology Department, Tufts Medical Center, Boston, USA. Tel: +61-76368711, Fax:+ 61-7636-1479, E-mail: rwein@
                Copyright © 2014, Research Institute For Endocrine Sciences and Iran Endocrine Society; Published by Kowsar Corp.

                This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

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