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

Abstract

SummaryAnti-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.

Concomitant Thyroid Disorders and Inflammatory Bowel Disease: A Literature Review

“…Standard treatment for SAT is administration of prednisolone. Moreover, SAT-like syndrome (symptoms) may have similar clinical symptoms as SAT and the thyroid function in SAT-like syndrome is within the normal range [ 41 , 50 , 77 ]. The development of SAT in IBD patients appears to be extremely rare.…”

“…The development of SAT in IBD patients appears to be extremely rare. A few cases of SAT or SAT-like syndrome have occurred in IBD patients [ 50 , 77 , 78 ]. One case report of SAT in IBD was reported by Horai et al [ 78 ].…”

“…This suggests that genetic factors may be associated with the occurrence of these conditions [ 78 ]. In another case, Kawashima et al [ 50 ] reported a middle-aged man with AG and CD, who developed hyperthyroidism caused by SAT after the administration of the tumor necrosis factor- (TNF-) α inhibitor infliximab, and subsequent SAT-like symptoms then developed after the administration of another TNF- α inhibitor, adalimumab (suspected drug-induced thyroiditis). However, after an examination of 36 rheumatic patients, Kaklamanos et al [ 79 ] reported that infliximab and rituximab did not cause any alterations in thyroid function and/or autoimmunity (thyroid antibody titer), even in patients with previously undiagnosed autoimmune thyroid diseases.…”

Subacute thyroiditis in a patient with psoriasis treated with a tumor necrosis factor‐α inhibitor

“…TNF‐ α is a cytokine that could be secreted by epithelial cells and fibroblasts of the thyroid gland and lymphocytes . TNF‐ α regulates thyroid gland functions and contributes to hyperthyroidism or hypothyroidism clinically. In addition, the use of biologics induces a relative immunosuppressive status, rendering patients susceptible to developing viral infection, which appears to be a triggering factor for SAT.…”

“…Anti‐TNF‐ α therapy may also contribute to other thyroid diseases, such as Graves' disease, Hashimoto's thyroiditis, amyloid goiter, and papillary thyroid carcinoma . Allanore et al .…”