Sabrina Chiloiro1, Tommaso Tartaglione2,3, Ettore Domenico Capoluongo4,5, Flavia Angelini6, Vincenzo Arena7, Antonella Giampietro1, Antonio Bianchi1, Angelo Zoli8, Alfredo Pontecorvi1, Cesare Colosimo7, Laura De Marinis1. 1. Pituitary Unit, Department of Endocrinology, Catholic University of the Sacred Heart, School of Medicine, Rome, Italy. 2. Department of Radiology, Istituto Dermopatico dell'Immacolata, Rome, Italy. 3. Catholic University of the Sacred Heart, School of Medicine, Rome, Italy. 4. Department of Laboratory Medicine, Genetic and Toxicology, Istituto Dermopatico dell'Immacolata, Rome, Italy. 5. Institute of Biochemistry and Clinical Biochemistry, Catholic University of the Sacred Heart, School of Medicine, Rome, Italy. 6. Laboratory of Vascular Biology and Genetics, Department of Medicine, Catholic University of the Sacred Heart, School of Medicine, Rome, Italy. 7. Department of Pathology, Catholic University of the Sacred Heart, School of Medicine, Rome, Italy. 8. Institute of Rheumatology and Affine Sciences, Division of Rheumatology, Catholic University of the Sacred Heart, School of Medicine, Rome, Italy.
Abstract
Context: Primary autoimmune hypophysitis (PAH) evolves in most untreated cases in irreversible hypopituitarism. PAH outcome, instead, after immunosuppressive treatment has not been completely clarified. Objective: To evaluate hypophysitis and pituitary function outcomes. Design: A prospective, double-arm study with a 2-year follow-up. Setting: Referral center for pituitary disease. Patients: Twenty PAH cases. Interventions: Oral prednisone 50 mg/d or conservative strategy by observation. Main Outcome Measures: Primary endpoint was the improvement/stabilization/worsening of PAH from baseline to a 2-year visit. Secondary endpoint was the improvement/stabilization/worsening of pituitary function from baseline to a 2-year visit. Results: Twelve patients (57.1%) were treated with a glucocorticoid-immunosuppressive therapy, and eight patients (42.9%) were observed. At the 2-year visit, PAH improvement/recovery occurred in eight immunosuppressive-treated (66.7%) patients and in two untreated patients (25%). PAH worsened in three untreated patients (37.5%) and was considered stable in four immunosuppressive-treated (33.3%) and three untreated patients (37.5%). Improvement/recovery of pituitary function occurred more frequently in immunosuppressive-treated patients (58.3%) compared with untreated ones (25%; P = 0.04). Responsiveness to immunosuppressive treatment is correlated with antipituitary antibody presence (P = 0.01), occurrence of diabetes insipidus at PAH diagnosis (P = 0.01), absence of the physiological neuropituitary "bright spot" on T1-weighed images (P = 0.01), and pituitary stalk at optical chiasm larger than 3.9 mm (area under the curve: 0.97, sensibility: 100%, specificity: 100%; P = 0.04). On the other hand, we failed to identify factors predicting the outcome, among untreated patients. Conclusions: Glucocorticoid treatment of hypophysitis improves pituitary secretion and should be encouraged in accordance with the evaluation of endocrine-, immunological-, and morphological-predictive markers.
Context:Primary autoimmune hypophysitis (PAH) evolves in most untreated cases in irreversible hypopituitarism. PAH outcome, instead, after immunosuppressive treatment has not been completely clarified. Objective: To evaluate hypophysitis and pituitary function outcomes. Design: A prospective, double-arm study with a 2-year follow-up. Setting: Referral center for pituitary disease. Patients: Twenty PAH cases. Interventions: Oral prednisone 50 mg/d or conservative strategy by observation. Main Outcome Measures: Primary endpoint was the improvement/stabilization/worsening of PAH from baseline to a 2-year visit. Secondary endpoint was the improvement/stabilization/worsening of pituitary function from baseline to a 2-year visit. Results: Twelve patients (57.1%) were treated with a glucocorticoid-immunosuppressive therapy, and eight patients (42.9%) were observed. At the 2-year visit, PAH improvement/recovery occurred in eight immunosuppressive-treated (66.7%) patients and in two untreated patients (25%). PAH worsened in three untreated patients (37.5%) and was considered stable in four immunosuppressive-treated (33.3%) and three untreated patients (37.5%). Improvement/recovery of pituitary function occurred more frequently in immunosuppressive-treated patients (58.3%) compared with untreated ones (25%; P = 0.04). Responsiveness to immunosuppressive treatment is correlated with antipituitary antibody presence (P = 0.01), occurrence of diabetes insipidus at PAH diagnosis (P = 0.01), absence of the physiological neuropituitary "bright spot" on T1-weighed images (P = 0.01), and pituitary stalk at optical chiasm larger than 3.9 mm (area under the curve: 0.97, sensibility: 100%, specificity: 100%; P = 0.04). On the other hand, we failed to identify factors predicting the outcome, among untreated patients. Conclusions: Glucocorticoid treatment of hypophysitis improves pituitary secretion and should be encouraged in accordance with the evaluation of endocrine-, immunological-, and morphological-predictive markers.
Authors: Sriram Gubbi; Fady Hannah-Shmouni; Joseph G Verbalis; Christian A Koch Journal: Best Pract Res Clin Endocrinol Metab Date: 2019-12-12 Impact factor: 4.690