A Case of Lymphocytic Infundibuloneurohypophysitis Showing Diabetes Insipidus Followed by Anterior Hypopituitarism Associated with Thrombasthenia.

Takao, Toshihiro; Asaba, Koichi; Tanaka, Hiroaki; Matsumoto, Reiko; Nanamiya, Wakako; Hashimoto, Kozo

doi:10.1507/endocrj.47.285

Cited by 10 publications

(5 citation statements)

References 34 publications

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“…Access to the pituitary gland is not easy, nor is antipituitary antibody detection [52][53][54][55]. This would support a diagnosis of present or past hypophysitis.…”

Section: Discussionmentioning

confidence: 99%

Empty sella and primary autoimmune hypothyroidism

et al. 2009

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In order to assess the association between empty sella (ES) and primary autoimmune hypothyroidism, and the possibility of a common pathogenesis. We retrospectively studied all patients with presumed ES diagnosed in the last 20 years, most of whom were treated by our Endocrinology Department. Subjects with a known etiology were excluded. Incomplete records or those with a doubtful diagnosis were also excluded. A total of 56 subjects were included in the study. ES was diagnosed by pituitary MRI. The measurement of free T4, TSH, and antithyroid antibodies (TPOAb and TgAb) was assayed using commercial kits. The cases of hypothyroidism obtained were compared with those in another group of similar patients, diagnosed with diabetes mellitus type 2, through chi2 test. A total of 15 (26.78%) patients of 56 with ES had autoimmune thyroid disease (subclinical or clinical hypothyroidism). Primary hypothyroidism with negative antithyroid autoantibodies was found in a further 13 patients (23.21%). The 46.42% of ES had primary hypothyroidism; this result had obtained a statistically significant difference when compared to the ratio obtained in the group of diabetes mellitus type 2 (P < 0.0029). There is an important association between ES and autoimmune thyroid disease, which reached 26.78% in our series. We suggest the possibility of a common pathogenesis for certain cases of ES and autoimmune thyroid disease, with the end point of ES in the pituitary, and atrophy in the thyroid gland.

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“…Access to the pituitary gland is not easy, nor is antipituitary antibody detection [52][53][54][55]. This would support a diagnosis of present or past hypophysitis.…”

Section: Discussionmentioning

confidence: 99%

Empty sella and primary autoimmune hypothyroidism

et al. 2009

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“…Although lymphocytic hypophysitis can have similar symptoms and radiologic findings, it usually regresses spontaneously. The features of the MRI indicative of lymphocytic hypophysitis include loss of the hyperintense “bright spot” signal of the posterior pituitary on T1-weighted images, enlargement of the posterior gland, or an isodense homogeneous mass [9]. None of these features were noted in our patient, which led us to exclude the possibility of lymphocytic hypophysitis.…”

Section: Discussionmentioning

confidence: 71%

A Case of Possible Neurosarcoidosis Presenting as Intractable Headache and Panhypopituitarism

Hwang

Cho²,

Park³

et al. 2013

Case Reports in Endocrinology

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Sarcoidosis is a chronic multisystemic inflammatory disease of unknown etiology, which is characterized by noncaseating granulomatous inflammation of the involved organs. It is known that neurosarcoidosis involving the nervous system occurs in about 5% of patients with sarcoidosis. However, neurosarcoidosis without systemic involvement is extremely rare. We present a case of suspicious neurosarcoidosis affecting the pituitary gland, which was manifested as chronic uncontrolled headache, panhypopituitarism, central diabetes insipidus, and hypercalcemia. Though the biopsy at the pituitary lesion was not performed due to the high risk of surgical complication, treatment was needed urgently and we started steroid therapy. After steroid therapy, we observed the immediate symptom relief with improved hypercalcemia. According to the follow-up examination, no recurrent symptom was seen, and resolution of the pituitary lesion with improving panhypopituitarism was noted.

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“…We observed that overall clinical response and especially visual field recovery was better with high and very high dose vs medium dose glucocorticoids. A pathophysiological explanation for improved clinical response to very high doses (>100 mg/day) is probably related to the non-specific non-genomic actions, where higher supra-physiological doses (>100 mg/day) have anti-inflammatory effects whereas lower supra-physiological doses (<30 mg/day) are just immunomodulatory (5,19). It has been demonstrated that the non-genomic action of glucocorticoids has a log-linear relationship with dose after ≥30 mg/day, below which it is negligible ( 5).…”

Section: Discussionmentioning

confidence: 99%

Glucocorticoid therapy as first-line treatment in primary hypophysitis: a systematic review and individual patient data meta-analysis

Krishnappa

Shah

Memon

et al. 2023

Endocrine Connections

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Objectives: High-dose glucocorticoids are associated with improved recovery of deficits in primary autoimmune hypophysitis (PAH), but optimal dosing, route, and duration are unclear. Design: We reviewed literature for first-line glucocorticoid treatment in PAH until December 2021 and performed an individual-patient data meta-analysis to analyze clinical, hormonal, and radiological outcomes with respect to route, dose, and duration (<6.5 vs. 6.5-12 vs. >12 weeks) of glucocorticoid treatment according to disease severity. Results: 153 PAH patients from 83 publications were included. The median age at presentation was 41(32.5-48) years with a female preponderance (70.3%). Visual field recovery was significantly better with intravenous (91.7%) as compared to oral (54.5%) route, high dose (100%) and very-high dose (90.9%) as compared to medium dose (20%) of glucocorticoids. Corticotroph axis recovery was greater in intravenous (54.8% vs. 28.1% oral, p=0.033) route and increasing glucocorticoid dose group (0% vs. 38.1% vs. 57.1%), attaining statistical significance (p=0.012) with very high-dose. A longer duration of treatment (>6.5 weeks) was associated with better corticotroph and thyrotroph recovery. Need for rescue therapy with lower with intravenous route (38% vs 17.5%, p=0.012) and with increasing glucocorticoid doses (53.3% vs. 34.3% vs. 17.3%, p=0.016). In severe disease, visual field and corticotroph axis recovery was significantly higher with intravenous route and very-high dose steroids. The adverse effects of glucocorticoids were independent of dose and duration of treatment. Conclusions: Very high-dose glucocorticoids by intravenous route and cumulative longer duration (>6.5 weeks) lead to better outcomes and could be considered as first-line treatment of severe PAH cases.

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A Case of Lymphocytic Infundibuloneurohypophysitis Showing Diabetes Insipidus Followed by Anterior Hypopituitarism Associated with Thrombasthenia.

Cited by 10 publications

References 34 publications

Empty sella and primary autoimmune hypothyroidism

Empty sella and primary autoimmune hypothyroidism

A Case of Possible Neurosarcoidosis Presenting as Intractable Headache and Panhypopituitarism

Glucocorticoid therapy as first-line treatment in primary hypophysitis: a systematic review and individual patient data meta-analysis

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