High Prevalence of Hypothyroidism in Patients with Autoimmune Pancreatitis

Komatsu, Kenichi; Hamano, Hideaki; Ochi, Yasuhide; Takayama, Mari; Muraki, Takamura; Yoshizawa, Kaname; Sakurai, Akihiro; Ôta, Masao; Kawa, Shigeyuki

doi:10.1007/s10620-005-2703-9

Cited by 123 publications

(79 citation statements)

References 33 publications

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“…Patients with IgG4-related disease that included AIP have hypothyroidism in 27% of cases (14) and that finding led investigators to search for a relationship between HT and that systemic disease.…”

Section: Discussionmentioning

confidence: 99%

IgG4-related Hashimoto’s thyroiditis – A new variant of a well known disease

Luiz

Gonçalves

Silva

et al. 2014

Arq Bras Endocrinol Metab

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SUMMARYHashimoto's thyroiditis (HT) has been characterized for many years as a well-defined clinicopathologic entity, but is now considered a heterogeneous disease. IgG4-related HT is a new subtype characterized by thyroid inflammation rich in IgG4-positive plasma cells and marked fibrosis. It may be part of the systemic IgG4-related disease. We report a case of a 56-year-old Portuguese man who presented with a one-month history of progressive neck swelling and dysphagia. Laboratory testing revealed increased inflammatory parameters, subclinical hypothyroidism and very high levels of thyroid autoantibodies. Cervical ultrasound (US) demonstrated an enlarged and heterogeneous thyroid gland and two hypoechoic nodules. US-guided fine needle aspiration cytology was consistent with lymphocytic thyroiditis. The patient was submitted to total thyroidectomy and microscopic examination identified typical findings of HT, marked fibrosis limited within the thyroid capsule and lymphoplasmacytic infiltration, with > 50 IgG4-positive plasma cells per high-power field and an IgG4/ IgG ratio of > 40%. After surgery, serum IgG4 concentration was high-normal. Symptoms relief and reduction in laboratory inflammatory parameters were noticed. Thyroid function is controlled with levothyroxine. To our knowledge we report the first case of IgG4-related HT in a non-Asian patient. We also perform a review of the literature regarding IgG4-related disease and IgG4-related HT. Our case highlights this new variant of the well known HT, and helps physicians in recognizing its main clinical features, allowing for proper diagnosis and treatment. Arq Bras Endocrinol Metab. 2014;58(8):862-8 SUMÁRIO A tireoidite de Hashimoto (TH) foi caracterizada durante muitos anos como uma entidade clinicopatológica bem definida, mas é atualmente considerada uma patologia heterogênea. A TH associada a IgG4 apresenta-se como um novo subtipo, sendo caracterizada por inflamação da tireoide com numerosos plasmócitos IgG4-positivos e fibrose extensa. É possível que pertença ao espectro da doença sistêmica associada a IgG4. Relatamos o caso de um homem português de 56 anos que se apresentou com aumento progressivo do volume cervical e disfagia, com um mês de evolução. A avaliação laboratorial revelou elevação dos parâmetros inflamatórios, hipotireoidismo subclínico e níveis muito elevados de autoanticorpos tireoidianos. Por ultrassonografia cervical demonstrou-se tireoide aumentada, heterogênea, com dois nódulos hipoecoicos. Foi realizada citologia aspirativa com agulha fina guiada por ultrassom, compatível com tireoidite linfocítica. O doente foi submetido à tireoidectomia total e o exame histológico revelou achados típicos de TH, extensa fibrose localizada dentro da cápsula tireoidiana e infiltrado linfoplasmocitário, com > 50 plasmócitos IgG4-positivos por campo de grande ampliação e uma relação IgG4/IgG > 40%. Após cirurgia, a concentração sérica de IgG4 encontrava-se no limite superior do normal. Ocorreu melhoria sintomática e redução dos parâmetros inflamatór...

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Section: Discussionmentioning

confidence: 99%

IgG4-related Hashimoto’s thyroiditis – A new variant of a well known disease

Luiz

Gonçalves

Silva

et al. 2014

Arq Bras Endocrinol Metab

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“…Approximately 20 % of patients have a history of allergic disorders such as bronchial asthma, chronic sinusitis, and drug allergies. Up to 15 % of patients with IgG4-RD also have hypothyroidism and/or Hashimoto's thyroiditis [26]. Although some patients rarely have other autoimmune diseases (e.g., rheumatoid arthritis and inflammatory bowel disease), they are more likely to be an incidental association with no proven pathogenetic relevance.…”

Section: Clinical Featuresmentioning

confidence: 99%

IgG4-related sclerosing cholangitis: all we need to know

2016

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Our knowledge and experience of IgG4-related sclerosing cholangitis (ISC) have expanded in the last decade. ISC is one of the common organ manifestations of IgG4-related disease (IgG4-RD); approximately 60 % of patients with this systemic condition have ISC in the proximal and/or distal bile ducts. ISC needs to be discriminated from primary sclerosing cholangitis, cholangiocarcinoma, and other rare forms of lymphoplasmacytic cholangiopathy (e.g., follicular cholangitis and sclerosing cholangitis with granulocytic epithelial lesions). Its diagnosis requires a multidisciplinary approach, in which serology, histology, and imaging play crucial roles. Treatments with high-dose corticosteroids typically lead to the rapid and consistent induction of disease remission. Another promising therapeutic approach is B-cell depletion with rituximab. Although disease relapse is relatively common, provided that appropriate treatments are administered, ISC is considered a ''benign'' disease with a low risk of liver failure and biliary malignancy. Its molecular pathology is characterized by Th2-dominant immune reactions, regulatory T-cell activation, and CCL1-CCR8 interactions. Particular subsets of B cells such as plasmablasts and regulatory B cells also expand. A recent global proteomic study demonstrated that three significantly activated immunological cascades in ISC were all B-cell-or immunoglobulin-related (Fc-gamma receptormediated phagocytosis, B-cell receptor signaling pathway, and Fc-epsilon receptor I signaling pathway), suggesting the crucial roles of B cells in the underlying immune reactions. Despite the expansion of our knowledge of the pathophysiology of ISC, the exact role of IgG4 remains unclear. A better understanding of its immunopathology will offer some potential drug targets for this emerging biliary disease.

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“…Anti-mitochondria antibody (AMA), a specific marker of PBC, is also very rarely detected in AIP patients. It is reported that one fourth of AIP patients show a decrease in thyroid function and 34% and 17% of them are associated with positive anti-thyroglobulin antibody and anti-thyroid peroxidase (TPO) antibody, respectively [54].…”

Section: Antibodiesmentioning

confidence: 99%

“…It disappears shortly after starting steroid treatment (b) retroperitoneal fibrosis [68], tubulointerstitial nephritis [69,70] and interstitial pneumonitis [71,72]. Other lesions reported include hypophysitis [73], chronic thyroiditis [54], pseudotumor of the liver [74,75], gastric ulcer [76], prostitis [77], Schönlein-Henoch purpura and autoimmune thrombocytopenia [78]. It is unknown whether all of them are real complications of AIP.…”

Section: Extrapancreatic Organ Involvementmentioning

confidence: 99%

Autoimmune pancreatitis in Japan: overview and perspective

Shimosegawa

Kanno

2009

J Gastroenterol

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Since the rediscovery and definition of autoimmune pancreatitis (AIP) by Yoshida et al. in 1995, the disease has been attracting attention because of its unique clinical features and practical issues. This disease shows very impressive imaging findings, serological changes, and characteristic histopathology. It occurs most commonly in elderly males with painless jaundice or mild abdominal pain; resemblance in imaging findings between AIP and pancreatobiliary cancers poses an important practical issue of differentiation. With increasing recognition of AIP and accumulation of cases, another important feature of this disease has been revealed, i.e

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High Prevalence of Hypothyroidism in Patients with Autoimmune Pancreatitis

Cited by 123 publications

References 33 publications

IgG4-related Hashimoto’s thyroiditis – A new variant of a well known disease

IgG4-related Hashimoto’s thyroiditis – A new variant of a well known disease

IgG4-related sclerosing cholangitis: all we need to know

Autoimmune pancreatitis in Japan: overview and perspective

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