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      A rare case showing subacute thyroiditis-like symptoms with amyloid goiter after anti-tumor necrosis factor therapy

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          Summary

          Anti-tumor necrosis factor (TNF)-α therapy is established as a new standard for the treatment of various autoimmune inflammatory diseases. We report the first case showing subacute thyroiditis-like symptoms with an amyloid goiter after anti-TNF-α therapy. A 56-year-old man with Crohn's disease presented with fever and a diffuse, tender goiter. To control the diarrhea, anti-TNF therapy (infliximab) was administered 4 weeks before the thyroid symptoms emerged. The patient reported a swollen neck with tenderness on the right side and fever 4 days after the second infliximab injection. An elevated serum C-reactive protein (CRP) and serum thyroid hormone level with suppressed serum thyrotropin were observed. The thyroid-stimulating antibody was not elevated. An ultrasonograph of the thyroid revealed an enlarged goiter with posterior echogenicity attenuation and a low echoic region that was tender. The thyroid uptake value on technetium-99m scintigraphy was near the lower limit of the normal range. The patient was initially diagnosed with thyrotoxicosis resulting from subacute thyroiditis. Administration of oral prednisolone improved the fever, thyroid pain, and thyroid function, but his thyroid remained swollen. The patient developed diarrhea after prednisolone withdrawal; therefore, adalimumab, another TNF inhibitor, was administered. After three injections, his abdominal symptoms were alleviated, but the thyroid pain and fever recurred. Elevated serum CRP levels in the absence of thyroid dysfunction were observed. The patient's symptoms resolved after prednisolone retreatment, but an elastic, firm goiter persisted. A fine-needle biopsy revealed amyloid deposition in the thyroid.

          Learning points

          • Many cases with thyroid dysfunction accompanied by amyloid goiter have been reported.

          • There are cases that develop amyloid goiter with subacute thyroiditis-like symptoms after anti-TNF therapy.

          • When the thyroid remains swollen after improvement of thyrotoxicosis following treatment with prednisolone, it should be assessed to differentiate between an amyloid goiter and common subacute thyroiditis.

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          Most cited references14

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          Clinical features and outcome of subacute thyroiditis in an incidence cohort: Olmsted County, Minnesota, study.

          Subacute thyroiditis (SAT), or granulomatous thyroiditis, is an inflammatory thyroid condition associated with pain and systemic symptoms. Few community studies are available. We studied the 160 patients with SAT in Olmsted County, Minnesota, seen between January 1, 1960, and December 30, 1997. Subjects were identified through the medical diagnostic index of the Rochester Epidemiology Project. The overall age- and sex-adjusted incidence from 1960 through 1997 was 4.9 cases per 100,000/yr. In the most recent 28-yr period (1970-1997), 94 patients were identified. In this group, pain was the presenting symptom in 96%. SAT recurred in 4% of the patients 6-21 yr after the initial episode. Corticosteroid therapy was given to 36%. Early-onset hypothyroidism occurred both in patients receiving corticosteroid therapy (29%) and in those not receiving corticosteroid therapy (37%). At latest follow-up, significantly more patients who had received corticosteroid therapy had a diagnosis of hypothyroidism than the group without corticosteroid therapy (25% vs. 10%, P < 0.05; overall rate of hypothyroidism, 15%). Early transient hypothyroidism is common in SAT. Permanent hypothyroidism is less common, and only 15% of the patients are receiving T(4) therapy after 28 yr of follow-up. Symptomatic relief is achieved with corticosteroid therapy, but such therapy does not prevent early- and late-onset thyroid dysfunction.
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            Drugs and thyroid function.

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              Clinical characteristics of subacute thyroiditis classified according to human leukocyte antigen typing.

                Author and article information

                Journal
                Endocrinol Diabetes Metab Case Rep
                Endocrinol Diabetes Metab Case Rep
                edm
                EDM Case Reports
                Endocrinology, Diabetes & Metabolism Case Reports
                Bioscientifica Ltd (Bristol )
                2052-0573
                1 May 2015
                2015
                : 2015
                : 140117
                Affiliations
                [1 ]Department of Metabolic Medicine, Faculty of Life Sciences, Kumamoto University , 1-1-1 Honjo, Chuo-ku, Kumamoto, 860-8556, Japan
                [2 ]Department of Gastroenterology and Hepatology, Faculty of Life Sciences, Kumamoto University , 1-1-1 Honjo, Chuo-ku, Kumamoto, 860-8556, Japan
                Author notes
                Correspondence should be addressed to J Kawashima Email: jkawashima@ 123456fc.kuh.kumamoto-u.ac.jp
                Article
                EDM140117
                10.1530/EDM-14-0117
                4424874
                94d1a64f-1886-4a7f-a75e-3dd65b2ee9e9
                © 2015 The authors

                This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivs 3.0 Unported License.

                History
                : 1 April 2015
                : 8 April 2015
                Categories
                Unique/Unexpected Symptoms or Presentations of a Disease

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