Hypophysitis Outcome and Factors Predicting Responsiveness to Glucocorticoid Therapy: A Prospective and Double-Arm Study

Chiloiro, Sabrina; Tartaglione, Tommaso; Capoluongo, Ettore; Angelini, Flavia; Arena, Vincenzo; Giampietro, Antonella; Bianchi, Antonio; Zoli, Angelo; Pontecorvi, Alfredo; Colosimo, Cesare; Marinis, Laura De

doi:10.1210/jc.2018-01021

Cited by 34 publications

(34 citation statements)

References 34 publications

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“…Diabetes insipidus in LYH appears to be resistant to treatment 49 with only a small percentage of patients, approximately 10%, exhibiting recovery 36 . The presence of DI in patients with LYH is considered to be a negative indicator of response to treatment with glucocorticoids 52 …”

Section: Discussionmentioning

confidence: 99%

“…36 The presence of DI in patients with LYH is considered to be a negative indicator of response to treatment with glucocorticoids. 52 Involvement of two or more organs at diagnosis of LCH is the most important risk factor for DI, with a 4.7-fold increased risk as opposed to patients with single organ involvement. Importantly, lesions in the craniofacial area, especially in the region of the ears, eyes and oral cavity, carry a significantly increased risk for DI; LCHrelated DI is usually permanent.…”

Section: Re Sultsmentioning

confidence: 99%

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Diabetes insipidus secondary to sellar/parasellar lesions

Angelousi

Mytareli

Xekouki

et al. 2021

J Neuroendocrinology

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Diabetes insipidus (DI) is a well‐recognised transient or permanent complication following transsphenoidal surgery for pituitary adenomas or other sellar/parasellar lesions. However, data regarding the prevalence of pre‐operative DI in sellar/parasellar lesions other than pituitary adenomas are scarce. We systematically reviewed the existing data for defining the prevalence of DI before any treatment in adult patients with sellar/parasellar lesions, excluding pituitary adenomas and metastatic lesions. In total, 646 patients with sellar/parasellar lesions presenting with DI at diagnosis were identified. The most common pathologies of sellar/parasellar lesions presenting with DI at diagnosis were lymphocytic hypophysitis (26.5%), craniopharyngiomas (23.4%), Langerhans's cell histiocytosis (18.9%) and Rathke's cleft cyst (12.7%), accounting for the vast majority (more than 80%) of these lesions. Overall, DI at diagnosis was found in 23.4% of all patients with sellar/parasellar lesions, albeit with a wide range from 10.6% to 76.7%, depending on the nature of the pathology. The highest prevalence of DI was found in less commonly encountered lesions namely germ‐cell tumours (76.7%), abscesses (55.4%) and neurosarcoidosis (54.5%), each accounting for less than 3% of all sellar/parasellar lesions. Most DI cases (68.8%) were associated with anterior pituitary hormonal deficiencies, in contrast to pituitary adenomas that rarely present with DI. The enlargement and enhancement of the pituitary stalk were the most common findings on magnetic resonance imaging besides the loss of the high signal of the posterior pituitary on T1‐weighted images. Resolution of DI spontaneously or following systemic and surgical management occurred in 22.4% of cases. Post‐operative DI, not evident before surgery, was found in 27.8% of non‐adenomatous sellar/parasellar lesions, and was transient in 11.6% of them. Besides distinctive imaging features and symptoms, early recognition of DI in such lesions is important because it directs the diagnosis towards a non‐adenomatous sellar/parasellar tumour and the early initiation of appropriate treatment.

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Section: Discussionmentioning

confidence: 99%

Section: Re Sultsmentioning

confidence: 99%

Diabetes insipidus secondary to sellar/parasellar lesions

Angelousi

Mytareli

Xekouki

et al. 2021

J Neuroendocrinology

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“…Diagnosis of PAH was conducted, according to clinical criteria in most cases, as described in our previous studies [15][16][17]. All the following criteria have to be satisfied:…”

Section: Methodsmentioning

confidence: 99%

“…Diagnosis of PAH was conducted, according to clinical criteria in most cases, as described in our previous studies [ 15 – 17 ]. All the following criteria have to be satisfied: Occurrence of hypopituitarism and/or hyperprolactinemia and/or diabetes insipidus and/or visual field deficit and/or headache Identification of the typical hypophysitis findings [ 18 ] through a pituitary magnetic resonance (MR), as pituitary enlargement, pituitary stalk swelling and absence of the posterior pituitary “bright spot” on T1-weighed (T1-w) images; Exclusion of focal hypothalamic-pituitary lesions/masses; Exclusion of secondary causes as granulomatous vasculitis, sarcoidosis, Langerhans cell histiocytosis and tuberculosis [ 19 ].…”

Section: Methodsmentioning

confidence: 99%

Markers of humoral and cell-mediated immune response in primary autoimmune hypophysitis: a pilot study

et al. 2021

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Introduction Primary autoimmune hypophysitis (PAHs) is a rare inflammatory disease of the pituitary gland. Although largely investigated, the pathogenesis of PAH is not completely clarified. We aimed to investigate the immune response in PAHs. Material and methods Serum anti-pituitary and anti-hypothalamus antibodies (respectively APAs and AHAs) were investigated though an indirect immunofluorescence on monkey hypophysis and hypothalamus slides, serum cytokines though an array membrane and cell-mediated immunity though the white blood cells count. Results Nineteen PAH cases entered the study. APA or AHA were identified in all cases. APA were detected in 13 patients (68.4%) and AHA in 13 patients (68.4%). Ten patients (52.6%) were simultaneously positive for both APA and AHA. The prevalence of APAs and AHAs was higher as compared to those observed in 50 health controls (respectively 14% p < 0.001 and 24% p = 0.004) and in 100 not-secreting pituitary adenoma (NFPAs) (respectively 22% p = 0.002 and 8% p < 0.001). Similarly, the prevalence of simultaneous positivity for APA and AHA (52.9%) was higher as compared to the those detected in patients affected by NFPAs (0%; p < 0.001) and in health controls (16% p = 0.002). No differences were identified between PAHs and controls at qualitative and quantitative analysis of serum cytokines and white blood cells count. Conclusions This study suggest that APA and AHA may be detected in an high percentage of PAH cases and that their simultaneous identification may be useful for the differential diagnosis between PAH and NFPAs, in an appropriate clinical context.

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“…Presence of central DI was considered an unfavorable prognostic factor for response to glucocorticoids [76]. However, a recent study showed that presence of anti-pituitary antibody, central DI, absence of “bright spot” on MRI, and pituitary stalk >3.9 mm correlated with a better responsiveness to glucocorticoids in LH [77].…”

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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Hypophysitis Outcome and Factors Predicting Responsiveness to Glucocorticoid Therapy: A Prospective and Double-Arm Study

Abstract: Glucocorticoid treatment of hypophysitis improves pituitary secretion and should be encouraged in accordance with the evaluation of endocrine-, immunological-, and morphological-predictive markers.

Cited by 34 publications

References 34 publications

Diabetes insipidus secondary to sellar/parasellar lesions

Diabetes insipidus secondary to sellar/parasellar lesions

Markers of humoral and cell-mediated immune response in primary autoimmune hypophysitis: a pilot study

Hypophysitis (Including IgG4 and Immunotherapy)

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