Prednisolone markedly reduced serum IgG4 levels along with the improvement of pituitary mass and anterior pituitary function in a patient with IgG4-related infundibulo-hypophysitis

Iseda, Izumi; Hida, Kazuyuki; Tone, Atsuhiko; Tenta, Masafumi; Shibata, Yusuke; Matsuo, Kiyoshi; Yamadori, Ichiro; Hashimoto, Kozo

doi:10.1507/endocrj.ej13-0407

Cited by 43 publications

(35 citation statements)

References 31 publications

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“…With respect to clinical manifestations, both patients presented hypogonadotrophic hypogonadism and one had diabetes insipidus, in agreement with findings in the literature, where 25 of 32 cases presented varying degrees of hypopituitarism, 21 had diabetes insipidus and 16 patients presented both conditions (5) (9).…”

Section: Discussionsupporting

confidence: 89%

“…Various disorders such as Riedel's thyroiditis, Mikulicz's disease, retroperitoneal fibrosis, tubulo-interstitial nephritis, and sclerosing cholangitis, among others, are now considered part of this disease spectrum (5).…”

Section: Discussionmentioning

confidence: 99%

“…Moreover, in the first case there was considerable invasion of the sphenoid sinus and, in the second, signs of bleeding were present. Because these images did not initially suggest hypophysitis, both patients were submitted to TSS at which time definitive diagnosis of IgG4-related hypophysitis was established under the first Leporati's criterion, as also occurred in 11 of the 32 cases published to date (5) (9). Baseline IgG4 levels were not assayed since we did not initially suspect IgG4-related disease.…”

Section: Discussionmentioning

confidence: 99%

“…To date, 32 cases of IgG4-related hypophysitis have been reported in the literature, 11 of which included confirmatory tissue biopsy (5) and the majority affecting multiple organs. The aim of this report is to present two new cases of biopsy-proven IgG4-related hypophysitis occurring in two young female patients, with no evidence of involvement of other organs at the time of diagnosis.…”

Section: Introductionmentioning

confidence: 99%

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Histologically confirmed isolated IgG4-related hypophysitis: two case reports in young women

Sosa¹,

Bell²,

Christiansen³

et al. 2014

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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SummaryIgG4-related hypophysitis is a recently described entity belonging to the group of IgG4-related diseases. Many other organs can also be affected, and it is more common in older men. To date, 32 cases of IgG4-related hypophysitis have been reported in the literature, 11 of which included confirmatory tissue biopsy and the majority affecting multiple organs. The aim of this report is to present two cases of biopsy-proven IgG4-related hypophysitis occurring in two young female patients with no evidence of involvement of other organs at the time of diagnosis.Learning points IgG4-related hypophysitis belongs to the group of IgG4-related diseases, and is a fibro-inflammatory condition characterized by dense lymphoplasmacytic infiltrates rich in IgG4-positive plasma cells and storiform fibrosis.It is more common in older men, but young women may also present this type of hypophysitis.Although involvement of other organs is frequent, isolated pituitary disease is possible.Frequent clinical manifestations include anterior hypopituitarism and/or diabetes insipidus.The diagnosis may be confirmed with any of the following criteria: a pituitary biopsy with lymphoplasmacytic infiltrates, with more than ten IgG4-positive cells; a sellar mass and/or thickened pituitary stalk and a biopsy-proven involvement of another organ; a sellar mass and/or thickened pituitary stalk and IgG4 serum levels >140 mg/dl and sellar mass reduction and symptom improvement after corticosteroid treatment.Glucocorticoids are recommended as first-line therapy.

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Section: Discussionsupporting

confidence: 89%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Histologically confirmed isolated IgG4-related hypophysitis: two case reports in young women

Sosa¹,

Bell²,

Christiansen³

et al. 2014

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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“…Glucocorticoids have been found to be beneficial in reducing circulating levels of IgG4 and size of pituitary, and improving pituitary function in a patient with IgG4-related infundibulo-hypophysitis [30]. However, as yet the use of glucocorticoids in the management of IgG4RTD has not been evaluated and established in terms of actual hastening the recovery and reducing the need for long-term levothyroxine supplementation, a sequel of resulting primary hypothyroidism.…”

Section: Treatmentmentioning

confidence: 99%

Zaburzenia tarczycy związane z immunoglobuliną G4 — wyzwanie diagnostyczne i wyniki kliniczne

Dutta

Ahuja

Selvan

2016

Endokrynologia Polska

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Ig-G4 related disease (IgG4RD) is a heterogeneous disorder with multi-organ involvement recognised as a separate entity at the start of this century only. It is currently one of the hottest areas of clinical and translational research across specialties. Thyroid involvement in IgG4RD is rare, believed to occur in less than 4% of cases, may be isolated or may be associated with other organ involvement. As of today Riedel's thyroiditis, fibrosing variant of Hashimoto's thyroiditis, and few patients of Graves' orbitopathy represent the types of IgG4-related thyroid disease (IgG4RTD). This disorder is frequently confused with malignancy due to the intense sclerosis of thyroid resulting in hard texture on palpation compounded by often-associated compressive symptoms. Diagnosis involves establishing high circulating levels of IgG4 > 135 mg/dL, increased serum IgG4 to IgG ratio of > 8%, immunohistochemistry showing dense lymphoplasmacellular inflammatory infiltrate consisting of IgG4-positive plasma cells with storiform fibrosis and obliterative phlebitis, and increased IgG4 positive plasma cell > 10 cells per high-power field when at least three fields are evaluated. Glucocorticoids are the primary form of therapy in IgG4RD. However, their role in IgG4RTD needs to be evaluated. As of today levothyroxine supplementation for resulting hypothyroidism, appropriate management of Graves' disease, and surgical excision of thyroid in case of compressive symptoms remain the primary treatment options. StreszczenieChoroba związana z Ig-G4 (IgG4RD) jest heterogennym zaburzeniem z zajęciem wielu narządów. Jako osobna jednostka chorobowa została wyodrębniona dopiero na początku tego stulecia. Choroba ta stanowi aktualnie najgorętszy obszar badań klinicznych i translacyjnych w różnych dziedzinach medycyny. Zajęcie tarczycy w IgG4RD jest rzadkie, szacuje się, że występuje w około 4% przypadków, może być izolowane lub związane zajęciem innych narządów. Na chwilę obecną zapalenie tarczycy związane z IgG4 (IgG4RTD) reprezentują: zapalenie tarczycy Riedela, które jest włókniejącą odmianą zapalenia tarczycy w chorobie Hashimoto oraz niektórzy pacjenci z orbitopatią Gravesa. Zapalenie tarczycy związane z IgG4 jest często mylone z chorobą nowotworową ze względu na nasilone stwardnienie tarczycy, które powoduje powstanie twardej struktury wyczuwalnej w badaniu palpacyjnym z towarzyszącymi często objawami uciskowymi. Rozpoznanie choroby obejmuje: stwierdzenie wysokich stężeń krążącej IgG4 > 135 mg/dl, zwiększony stosunek w surowicy IgG4 do IgG > 8%, w badaniu immunohistochemicznym stwierdzenie gęstych nacieków zapalnych złożonych z limfocytów i IgG4 dodatnich plazmocytów , z włóknieniem i zarostowym zapaleniem żył oraz zwiększeniem liczby IgG4 dodatnich plazmocytów > 10 w polu widzenia, jeżeli oceniono minimum 3 pola. Glikokortykosteroidy stanowią leczenie pierwszego wyboru w IgG4RTD. Niemniej ich rola w IgG4RTD wymaga dalszej oceny. Na chwilę obecną podstawowe opcje terapeutyczne obejmują: suplementację lewotyroksyny we wtórnej nied...

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Infiltrative and Inflammatory Disorders of the Hypothalamus and Pituitary

Fernandes

Varlamov

2022

Pituitary Disorders Throughout the Life Cycle

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Prednisolone markedly reduced serum IgG4 levels along with the improvement of pituitary mass and anterior pituitary function in a patient with IgG4-related infundibulo-hypophysitis

Cited by 43 publications

References 31 publications

Histologically confirmed isolated IgG4-related hypophysitis: two case reports in young women

Histologically confirmed isolated IgG4-related hypophysitis: two case reports in young women

Zaburzenia tarczycy związane z immunoglobuliną G4 — wyzwanie diagnostyczne i wyniki kliniczne

Infiltrative and Inflammatory Disorders of the Hypothalamus and Pituitary

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