From Pituitary Expansion to Empty Sella: Disease Progression in a Mouse Model of Autoimmune Hypophysitis

Lupi, Isabella; Zhang, Jiangyang; Gutenberg, Angelika; Landek-Salgado, Melissa A.; Tzou, Shey Cherng; Mori, Susumu; Caturegli, Patrizio

doi:10.1210/en.2011-1004

Cited by 48 publications

(26 citation statements)

References 30 publications

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“…Although the final condition of IgG4-related hypophysitis has not been reported, Gao et al (37) reported that an empty sella is detected after a long period of treatment with glucocorticoids in cases of autoimmune hypophysitis. Furthermore, development of the pituitary from the expanded state to an empty sella has been reported in a mouse model of autoimmune hypophysitis (38). These data suggest that at least in some cases, IgG4-related hypophysitis results in an empty sella after a stage of swelling.…”

Section: European Journal Of Endocrinologymentioning

confidence: 68%

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

117

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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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Section: European Journal Of Endocrinologymentioning

confidence: 68%

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

117

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“…In addition, antipituitary antibodies (APA) and antihypothalamus (AHA) are often seen in patients with different pituitary pathologies including hypopituitarism, central hypothyroidism, acromegaly, adenomas, CDI, and in patients with Prader-Willi syndrome associated with hypothalamic dysfunction (13)(14)(15)(16)(17)(18)(19)(20)(21). Likewise, a mouse model of autoimmune hypophysitis showed endocrine dysfunction and disease progression associated with pituitary expansion followed by pituitary atrophy, thus suggesting the existence of a relationship between autoimmunity and endocrinopathy (22).…”

Section: Introductionmentioning

confidence: 99%

Antibodies Against Hypothalamus and Pituitary Gland in Childhood-Onset Brain Tumors and Pituitary Dysfunction

et al. 2020

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Purpose: To detect the presence of antipituitary (APA) and antihypothalamus antibodies (AHA) in subjects treated for brain cancers, and to evaluate their potential association with pituitary dysfunction. Methods:We evaluated 63 patients with craniopharyngioma, glioma, and germinoma treated with surgery and/or radiotherapy and/or chemotherapy at a median age of 13 years. Forty-one had multiple pituitary hormone deficiencies (MPHD), six had a single pituitary defect. GH was the most common defect (65.1%), followed by AVP (61.9%), TSH (57.1%), ACTH (49.2%), and gonadotropin (38.1%). APA and AHA were evaluated by simple indirect immunofluorescence method indirect immunofluorescence in patients and in 50 healthy controls.Results: Circulating APA and/or AHA were found in 31 subjects (49.2%) and in none of the healthy controls. In particular, 25 subjects out of 31 were APA (80.6%), 26 were AHA (83.90%), and 20 were both APA and AHA (64.5%). Nine patients APA and/or AHA have craniopharyngioma (29%), seven (22.6%) have glioma, and 15 (48.4%) have germinoma. Patients with craniopharyngioma were positive for at least one antibody in 39.1% compared to 33.3% of patients with glioma and to 78.9% of those with germinoma with an analogous distribution for APA and AHA between the three tumors. The presence of APA or AHA and of both APA and AHA was significantly increased in patients with germinoma. The presence of APA (P = 0.001) and their titers (P = 0.001) was significantly associated with the type of tumor in the following order: germinomas, craniopharyngiomas, and gliomas; an analogous distribution was observed for the presence of AHA (P = 0.002) and their titers (P = 0.012). In addition, we found a significant association between radiotherapy and APA (P = 0.03). Patti et al. Autoimmunity, Brain Tumors, Pituitary Dysfunction Conclusions: Brain tumors especially germinoma are associated with the development of hypothalamic-pituitary antibodies and pituitary defects. The correct interpretation of APA/AHA antibodies is essential to avoid a misdiagnosis of an autoimmune infundibulo-neurohypophysitis or pituitary hypophysitis in patients with germinoma.

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“…3,7 Cases of AH with secondary ES have also been reported. 14,15 In our series, we suggested that secondary ES (2/5, 40%) could be the consequence of AHT.…”

Section: Discussionmentioning

confidence: 60%

Clinical aspects of autoimmune hypothalamitis, a variant of autoimmune hypophysitis: Experience from one center

et al. 2019

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Objective Autoimmune hypothalamitis (AHT) is a rare inflammatory disorder that involves the hypothalamus. It remains unclear whether autoimmune hypophysitis (AH) and AHT represent different diseases or different aspects of the same disease. Thus, further investigation of AHT is required. Methods A retrospective review of medical and pathological records of AHT patients from the Chinese PLA General Hospital were examined from January 1, 2005 to May 1, 2017. Clinical data, treatments, and outcomes were investigated. Results Five female patients were identified (median age, 42.6 years). Symptoms included central diabetes insipidus, hypopituitarism, hyperprolactinemia, headache, and hypothalamic syndrome. The following hormonal deficits were noted: follicle-stimulating hormone, luteinizing hormone, adrenocorticotropic hormone, thyroid stimulating hormone, and growth hormone. One patient underwent high-dose methylprednisolone pulse treatment (HDMPT) and azathioprine plus intensity modulated radiation therapy (IMRT), and two patients underwent HDMPT and two rounds of replacement therapy. During follow-up, one patient died because of non-compliance with therapy and the others were in remission or they recovered. Conclusions AHT had similar MRI results, pathology, and treatment compared with AH. Thus, it may be a subtype of AH, and AHT patients may also show hypothalamic syndrome.

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From Pituitary Expansion to Empty Sella: Disease Progression in a Mouse Model of Autoimmune Hypophysitis

Cited by 48 publications

References 30 publications

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

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

Antibodies Against Hypothalamus and Pituitary Gland in Childhood-Onset Brain Tumors and Pituitary Dysfunction

Clinical aspects of autoimmune hypothalamitis, a variant of autoimmune hypophysitis: Experience from one center

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