Evidence map›Paper›PMID 34560890›Full record

ArticleJournal of medical case reports2021

Graves' disease coexisted with resistance to thyroid hormone: a case report.

Hiroshi Akahori, Rika Usuda

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Article in Journal of medical case reports, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

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2citing papers in PubMed
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3 · Its place in the literature

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2 citing papers in PubMed.

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5 · Who and what money

Authors and funding

2 authors.

Hiroshi AkahoriDepartment of Endocrinology and Metabolism, Toyama Prefectural Central Hospital, 2-2-78 Nishinagae, Toyama-shi, Toyama, 930-8550, Japan. akahori0606@hotmail.com.
Rika UsudaDepartment of Endocrinology, Diabetology and Metabolism, Toyama Prefectural Rehabilitaion Hospital and Support Center for Children with Disabilities, 36 Shimo-Iino, Toyama-shi, Toyama, 931-8517, Japan.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundResistance to thyroid hormone is a rare autosomal dominant disorder characterized by reduced responsiveness to thyroid hormone and can cause syndrome of inappropriate secretion of thyroid stimulating hormone. Although Graves' disease is a common autoimmune thyroid disorder, the coexistence of these two diseases is extremely rare and makes the diagnosis and treatment complicated, leading to the delayed diagnosis of resistance to thyroid hormone. We describe the case of a Japanese man with resistance to thyroid hormone coexisting with Graves' disease, in which the correct diagnosis of resistance to thyroid hormone was delayed by masking of the signs of syndrome of inappropriate secretion of thyroid stimulating hormone, with final diagnosis 30 years after the initial treatment for Graves' disease. CASE PRESENTATION: A 30-year-old Japanese man presented with diffuse goiter and thyrotoxicosis. Anti-thyroid stimulating hormone receptor antibody was positive. He was diagnosed with Graves' disease. Anti-thyroid medication was chosen as the initial treatment for Graves' disease. However, this treatment failed to normalize the free triiodothyronine, free thyroxine, and thyroid stimulating hormone levels. His thyroid hormone levels indicated syndrome of inappropriate secretion of thyroid stimulating hormone. After cessation of methimazole treatment by remission of Graves' disease, his state of syndrome of inappropriate secretion of thyroid stimulating hormone persisted. Magnetic resonance imaging revealed no pituitary tumor lesions. The results of thyroid stimulating hormone-releasing hormone stimulation test showed a normal response of thyroid stimulating hormone. He was suspected to have resistance to thyroid hormone. Direct sequencing analysis of the thyroid hormone receptor β gene identified a heterozygous missense mutation, R282S. Coexistence of resistance to thyroid hormone with Graves' disease was confirmed. He has no signs of thyrotoxic symptoms, and is capable in activities of daily living at the present time.

conclusionWe described a rare case of resistance to thyroid hormone simultaneously existing with Graves' disease. This case demonstrated that these diseases can coexist, and indicated some of the difficulties in diagnosis of resistance to thyroid hormone with coexisting Graves' disease. The diagnosis of resistance to thyroid hormone did not become apparent until after anti-hyperthyroidism treatment. Although rare, careful follow-up after the initial treatment of Graves' disease is necessary. The coexistence of these two diseases should be considered in patients showing occasional syndrome of inappropriate secretion of thyroid stimulating hormone.

Indexed as

Activities of Daily LivingGraves DiseaseAdultHumansMaleMethimazoleThyrotropinTriiodothyronineMethimazoleThyrotropinTriiodothyronineGraves’ diseaseR282SResistance to thyroid hormoneSyndrome of inappropriate secretion of TSHThyroid hormone receptor β

Identifiers

PMID34560890
PMCPMC8464102

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