A Case of IgG4-Related Hypophysitis Without Pituitary Insufficiency

Hattori, Yujiro; Tahara, Shigeyuki; Ishii, Yudo; Kitamura, Takayuki; Inomoto, Chie; Osamura, R. Yoshiyuki; Teramoto, Akira; Morita, Akio

doi:10.1210/jc.2013-1088

Cited by 49 publications

(29 citation statements)

References 22 publications

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“…Almost all cases of IgG4-related hypophysitis showed hypopituitarism and/or diabetes insipidus, although a rare case of IgG4-related hypophysitis without pituitary insufficiency was recently reported by Hattori et al [27]. Glucocorticoid, mainly PSL administration, markedly improved the enlargement of pituitary gland and stalk.…”

Section: Case Reportmentioning

confidence: 95%

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

et al. 2014

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Section: Case Reportmentioning

confidence: 95%

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

et al. 2014

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“…Leporati et al (7), the first to report IgG4-related hypophysitis in Caucasian patients, reviewed the published literature and proposed diagnostic criteria on the basis of findings of 11 cases that were analyzed. Although the first IgG4-related hypophysitis case was described in 2004 (3), only six histologically proven cases have been reported (3,4,7,8,9,10,11,12,13,14,15,16,17,18,19,20,21).…”

Section: Introductionmentioning

confidence: 99%

The prevalence of IgG4-related hypophysitis in 170 consecutive patients with hypopituitarism and/or central diabetes insipidus and review of the literature

Bando

Iguchi

Fukuoka

et al. 2014

European Journal of Endocrinology

115

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Objective: The prevalence and clinical characteristics of IgG4-related hypophysitis remain unclear due to the limited number of case reports. Therefore, in this study, we screened consecutive outpatients with hypopituitarism and/or diabetes insipidus (DI) to estimate its prevalence. Methods: A total of 170 consecutive outpatients with hypopituitarism and/or central DI were screened at Kobe University Hospital for detecting IgG4-related hypophysitis by pituitary magnetic resonance imaging, measuring serum IgG4 concentrations, assessing the involvement of other organs, and carrying out an immunohistochemical analysis to detect IgG4-positive cell infiltration. Results: Among the screened cases, 116 cases were excluded due to diagnosis of other causes such as tumors and congenital abnormalities. Additionally, 22 cases with isolated ACTH deficiency were analyzed and were found not to meet the criteria of IgG4-related hypophysitis. The remaining 32 cases were screened and seven were diagnosed with IgG4-related hypophysitis, of which three cases were diagnosed by analyzing pituitary specimens. IgG4-related hypophysitis was detected in 30% (seven of 23 patients) of hypophysitis cases and 4% of all hypopituitarism/DI cases. The mean age at the onset of IgG4-related hypophysitis was 61.8G8.8 years, and the serum IgG4 concentration was 191.1G78.3 mg/dl (normal values 5-105 mg/dl and values in IgG4-related disease (RD) R135 mg/dl). Pituitary gland and/or stalk swelling was observed in six patients, and an empty sella was observed in one patient. Multiple co-existing organ involvement was observed in four of the seven patients prior to the onset of IgG4-related hypophysitis. Conclusion: These data suggest that the prevalence of IgG4-related hypophysitis has been underestimated. We should also consider the possibility of the development of hypopituitarism/DI caused by IgG4-related hypophysitis during the clinical course of other IgG4-RDs.

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“…Cranial imaging reveals sellar mass and/or thickened pituitary stalk. 32,33 Our cases with elevated IgG4 levels were not evaluated thoroughly regarding these conditions; however, absence of relevant symptoms or laboratory findings (e.g. central hypothyroidism) militates against pancreatic or pituitary involvement.…”

Section: Discussionmentioning

confidence: 99%

Autoimmune thyroiditis in children and adolescents with type 1 diabetes mellitus is associated with elevated IgG4 but not with low vitamin D

Demir

Keskın

Kör

et al. 2014

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ObJEcTIVE: To assess levels of vitamin D and of immunoglobulin G subclasses in children and adolescents with type 1 Diabetes Mellitus with or without autoimmune thyroiditis. DE-SIGN: Among 213 patients with type 1 diabetes, the cases with thyroid-specific autoantibodies formed Group 1 [n=19, M/F: 7/12, median age 13 years (10.1-14.7)]. Nineteen age-, gender-, and diabetes duration-matched cases with type 1 diabetes without any other systemic disease were designated as controls [Group 2, M/F: 7/12, median age 12.9 years (10.5-14.9)]. rESULTS: Levels of thyroid hormones, vitamin D, total IgG and IgG subclasses, as well as IgG subclasses/ total IgG ratios were similar between the groups. Five cases (26%) in Group 1 had IgG4 levels > + 2 SDS, whereas there were no such cases in Group 2 (p=0.046). These five patients had similar clinical features but higher median IgG4 levels and IgG4/Total IgG ratios compared to the subjects with IgG4 levels < + 2 SDS in Group 1 and Group 2. cONcLUSIONS: There was no difference of vitamin D levels between the groups. Only a small percentage of patients with type 1 diabetes also having autoimmune thyroiditis had elevated serum IgG4 levels, revealing the heterogeneity of autoimmune thyroiditis and existence of IgG4 thyroiditis in the pediatric age group. Total IgG, the other IgG subclasses, and vitamin D levels did not differ in patients with autoimmune thyroiditis and type 1 diabetes compared to those suffering only from type 1 diabetes.

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A Case of IgG4-Related Hypophysitis Without Pituitary Insufficiency

Abstract: We described the first case of IgG4-related hypophysitis without pituitary insufficiency. However, further case collection is needed to characterize the pathophysiology of IgG4-related hypophysitis.

Cited by 49 publications

References 22 publications

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

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

The prevalence of IgG4-related hypophysitis in 170 consecutive patients with hypopituitarism and/or central diabetes insipidus and review of the literature

Autoimmune thyroiditis in children and adolescents with type 1 diabetes mellitus is associated with elevated IgG4 but not with low vitamin D

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