Anti-pituitary antibodies against corticotrophs in IgG4-related hypophysitis

Iwata, Naoko; Iwama, Shintaro; Sugimura, Yoshihisa; Yasuda, Yoshinori; Nakashima, Kohtaro; Takeuchi, Seiji; Hagiwara, Daisuke; Ito, Yoshihiro; Suga, Hidetaka; Goto, Motomitsu; Banno, Ryoichi; Caturegli, Patrizio; Koike, Teruhiko; Oshida, Yoshiharu; Arima, Hiroshi

doi:10.1007/s11102-016-0780-8

Cited by 21 publications

(15 citation statements)

References 52 publications

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“…Recent studies have also shown that prevalence of IgG4-related hypophysistis has been underestimated [43,44]. Αutoimmunity seems to be involved in the pathogenesis of IgG4 hypophysitis and corticotrophs are the main antigenic target [44]. Glucocorticoid therapy is generally effective although rare cases refractory to glucocorticoids remitted with rituximab treatment as in our case [45].…”

Section: Treatment/patients Characteristicsmentioning

confidence: 60%

“…Although the precise mechanisms remain unclear, hypophysitis is associated with the detection of antithyrotroph, anticorticotroph, and antigonad-otroph antibodies [41,42]. Recent studies have also shown that prevalence of IgG4-related hypophysistis has been underestimated [43,44]. Αutoimmunity seems to be involved in the pathogenesis of IgG4 hypophysitis and corticotrophs are the main antigenic target [44].…”

Section: Treatment/patients Characteristicsmentioning

confidence: 99%

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Clinical, Endocrine and Imaging Characteristics of Patients with Primary Hypophysitis

et al. 2018

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Primary hypophysitis (PH) is a rare disease with a poorly-defined natural history. Our aim was to characterise patients with PH at presentation and during prolonged follow-up. Observational retrospective study of 22 patients was conducted from 3 centres. In 14 patients, PH was confirmed histologically and in the remaining 8 clinically, after excluding secondary causes of hypophysitis. All patients had hormonal and imaging investigations before any treatment. Median follow up was 48 months (25-75%: 3-60). There was a female predominance with a female/male ratio: 3.4:1. Eight out of 22 patients had another autoimmune disease. Headaches and gonadal dysfunction were the most common symptoms. Five patients presented with panhypopituitarism; 17 patients had anterior pituitary deficiency, and 7 had diabetes insipidus. At presentation, 9 patients were treated surgically, 5 received replacement hormonal treatment, and 8 high-dose glucocorticoids from whom 5 in association with other immunosuppressive agents. Six patients showed complete recovery of pituitary hormonal deficiencies while 6 showed a partial recovery during a 5-year follow-up period. No difference was found between patients treated with surgery and those treated medically. The overall relapse rate was 18%. PH can be manifested with a broad spectrum of clinical and hormonal disturbances. Long-term follow-up is required to define the natural history of the disease and response to treatment, since pituitary hormonal recovery or relapse may appear many years after initial diagnosis. We suggest that surgery and immunosuppressive therapy be reserved for exceptional cases.

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Section: Treatment/patients Characteristicsmentioning

confidence: 60%

Section: Treatment/patients Characteristicsmentioning

confidence: 99%

Clinical, Endocrine and Imaging Characteristics of Patients with Primary Hypophysitis

et al. 2018

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“…Although the prevalence of IgG4-related hypophysitis remains unknown, a study of 170 consecutive outpatients with hypopituitarism and/or central diabetes insipidus reported that IgG4related hypophysitis was detected in 30% (7 of 23) of hypophysitis cases and 4% of all cases (7 of 170), suggesting that IgG4-related hypophysitis is not so rare [18]. Although the roles of IgG4 in hypophysitis remain unknown, autoantigen candidates (growth hormone and proopiomelanocortin) [19] and the presence of antipituitary antibodies [20] have been reported in patients with biopsy-proven IgG4-related hypophysitis, suggesting that autoimmunity is involved, at least in part, in the pathogenesis of IgG4-related disease [15]. Anterior pituitary dysfunction and/or central diabetes insipidus accompanied by enlargement of the pituitary gland and/or thickened stalk are often observed in patients with IgG4related hypophysitis [16].…”

Section: Igg4-related Hypophysitismentioning

confidence: 99%

Diagnosis and treatment of autoimmune and IgG4-related hypophysitis: clinical guidelines of the Japan Endocrine Society

et al. 2020

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Hypophysitis, which is often accompanied by pituitary dysfunction, is classified into several subtypes based on the cause, histology, and the location of inflammation in the pituitary gland. A definitive diagnosis requires pituitary biopsy, which is invasive, and the process is limited to specialized clinical settings. In this opinion paper, we review the literature associated with hypophysitis, and provide the guidelines of the Japan Endocrine Society for the diagnosis and treatment of autoimmune and IgG4-related hypophysitis.

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“…In IgG4H, anterior pituitary deficiency and DI develop in 48% [59]. Patient history and clinical presentation are useful tools in differentiating IgG4H from LH [1, 60]. IgG4-RD can cause panhypopituitarism, anterior hypopituitarism, or isolated DI in 50, 25, and 18% of cases, respectively [61, 62].…”

Section: Resultsmentioning

confidence: 99%

Hypophysitis (Including IgG4 and Immunotherapy)

et al. 2020

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Hypophysitis is characterized by inflammation of the pituitary gland that can be primary (PH) or secondary (SH) to other diseases or following drug administration. It may also be classified according to anatomical and histopathological criteria, leading to variable degrees of hypopituitarism and/or compressive symptoms to nearby structures. There has recently been an increase in the number of hypophysitis cases, raising the interest on the spectrum of its pathogenesis, clinical, biochemical/endocrinological, and imaging features. However, the use of conventional biomarkers, including currently utilized pituitary autoantibodies, has relatively limited diagnostic accuracy. Lymphocytic hypophysitis (LH) is the commonest cause of PH, whereas IgG4-related hypophysitis is increasingly being recognized. Histiocytosis and granulomatous diseases are the most frequent causes of SH, although infections and lymphoma have also been reported. The increasing use of immune checkpoint inhibitors in oncology is associated with a high incidence of hypophysitis, providing further understanding of its pathogenesis. Hy-pophysitis can occur silently and be easily missed, potentially leading to substantial morbidity or mortality due to adrenal insufficiency, requiring a high index of clinical suspicion and timely initiation of appropriate treatment. In most cases of LH or drug-induced hypophysitis, active surveillance along with replacement of established hormonal deficiencies is needed. In the presence of compressive and/or evolving symptoms, treatment with glucocorticoids either alone or in combination with other immunosuppressive agents can be used. Surgical decompression is reserved for nonresponsive cases with threatened vital structures. Timely diagnosis and intervention are important to minimize disease-related morbidity and mortality. We aimed to review current concepts and recent developments in the pathogenesis, diagnosis, and management of hypophysitis.

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Anti-pituitary antibodies against corticotrophs in IgG4-related hypophysitis

Abstract: Our study suggests that autoimmunity is involved in the pathogenesis of IgG4-RH and that corticotrophs are the main antigenic target, highlighting a possible new diagnostic marker for this condition.

Cited by 21 publications

References 52 publications

Clinical, Endocrine and Imaging Characteristics of Patients with Primary Hypophysitis

Clinical, Endocrine and Imaging Characteristics of Patients with Primary Hypophysitis

Diagnosis and treatment of autoimmune and IgG4-related hypophysitis: clinical guidelines of the Japan Endocrine Society

Hypophysitis (Including IgG4 and Immunotherapy)

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