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Patrizio Caturegli - One of the best experts on this subject based on the ideXlab platform.
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diabetes insipidus is an unfavorable prognostic factor for response to glucocorticoids in patients with Autoimmune Hypophysitis
European Journal of Endocrinology, 2017Co-Authors: Isabella Lupi, Patrizio Caturegli, Enio Martino, Luca Manetti, Mirco Cosottini, Claudio Urbani, Daniele Cappellani, Ilaria Scattina, Claudio Marcocci, Fausto BogazziAbstract:Introduction Autoimmune Hypophysitis (AH) has a variable clinical presentation and natural history; likewise, its response to glucocorticoid therapy is often unpredictable. Objective To identify clinical and radiological findings associated with response to glucocorticoids. Design and methods 12 consecutive patients with AH, evaluated from 2008 to 2016. AH was the exclusion diagnosis after ruling out other pituitary masses and secondary causes of Hypophysitis. Mean follow-up time was 30 ± 27 months (range 12-96 months). Results MRI identified two main patterns of presentation: global enlargement of the pituitary gland or panHypophysitis (n = 4, PH), and pituitary stalk abnormality only, or infundibulo-neuro-Hypophysitis (n = 8, INH). Multiple tropin defects were more common in PH (100%) than those in INH (28% P = 0.014), whereas diabetes insipidus was more common in INH (100%) than that in PH (50%; P = 0.028). All 4 PH and 4 out of 8 INH were treated with glucocorticoids. Pituitary volume significantly reduced in all PH patients (P = 0.012), defective anterior pituitary function recovered only in the two patients without diabetes insipidus (50%) and panhypopituitarism persisted, along with diabetes insipidus, in the remaining 2 (50%). In all INH patients, either treated or untreated, pituitary stalk diameter reduced (P = 0.008) but diabetes insipidus persisted in all. Conclusions Glucocorticoid therapy may improve anterior pituitary function in a subset of patients but has no effect on restoring posterior pituitary function. Diabetes insipidus appears as a negative prognostic factor for response to glucocorticoids.
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in situ activation of pituitary infiltrating t lymphocytes in Autoimmune Hypophysitis
Scientific Reports, 2017Co-Authors: Han Huei Lin, Angelika Gutenberg, Patrizio Caturegli, Tzu Yu Chen, Nu Man Tsai, Chiajung Lee, Yu Che Cheng, Wen Hui Cheng, Ywhmin Tzou, Shey Cherng TzouAbstract:Autoimmune Hypophysitis (AH) is a chronic inflammatory disease characterized by infiltration of T and B lymphocytes in the pituitary gland. The mechanisms through which infiltrating lymphocytes cause disease remain unknown. Using a mouse model of AH we assessed whether T lymphocytes undergo activation in the pituitary gland. Infiltrating T cells co-localized with dendritic cells in the pituitary and produced increased levels of interferon-γ and interleukin-17 upon stimulation in vitro. Assessing proliferation of CD3- and B220-postive lymphocytes by double immunohistochemistry (PCNA-staining) and flow cytometry (BrdU incorporation) revealed that a discrete proportion of infiltrating T cells and B cells underwent proliferation within the pituitary parenchyma. This proliferation persisted into the late disease stage (day 56 post-immunization), indicating the presence of a continuous generation of autoreactive T and B cells within the pituitary gland. T cell proliferation in the pituitary was confirmed in patients affected by Autoimmune Hypophysitis. In conclusion, we show that pituitary-infiltrating lymphocytes proliferate in situ during AH, providing a previously unknown pathogenic mechanism and new avenues for treatment.
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ORIGINAL RESEARCH A Radiologic Score to Distinguish Autoimmune Hypophysitis from Nonsecreting Pituitary
2015Co-Authors: Adenoma Preoperatively, Angelika Gutenberg, J Larsen, Veit Rohde, I. Lupi, Patrizio CaturegliAbstract:BACKGROUND AND PURPOSE: Autoimmune Hypophysitis (AH) mimics the more common nonsecret-ing pituitary adenomas and can be diagnosed with certainty only histologically. Approximately 40 % of patients with AH are still misdiagnosed as having pituitary macroadenoma and undergo unnecessary surgery. MR imaging is currently the best noninvasive diagnostic tool to differentiate AH from nonsecreting adenomas, though no single radiologic sign is diagnostically accurate. The purpose of this study was to develop a scoring system that summarizes numerous MR imaging signs to increase the probability of diagnosing AH before surgery. MATERIALS AND METHODS: This was a case-control study of 402 patients, which compared the presurgical pituitary MR imaging features of patients with nonsecreting pituitary adenoma and controls with AH. MR images were compared on the basis of 16 morphologic features besides sex, age, and relation to pregnancy. RESULTS: Only 2 of the 19 proposed features tested lacked prognostic value. When the other 17 predictors were analyzed jointly in a multiple logistic regression model, 8 (relation to pregnancy, pituitary mass volume and symmetry, signal intensity and signal intensity homogeneity after gadolin-ium administration, posterior pituitary bright spot presence, stalk size, and mucosal swelling) remaine
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ORIGINAL RESEARCH A Radiologic Score to Distinguish Autoimmune Hypophysitis from Nonsecreting Pituitary
2015Co-Authors: Adenoma Preoperatively, Angelika Gutenberg, J Larsen, Veit Rohde, I. Lupi, Patrizio CaturegliAbstract:BACKGROUND AND PURPOSE: Autoimmune Hypophysitis (AH) mimics the more common nonsecret-ing pituitary adenomas and can be diagnosed with certainty only histologically. Approximately 40 % of patients with AH are still misdiagnosed as having pituitary macroadenoma and undergo unnecessary surgery. MR imaging is supposedly the best noninvasive diagnostic tool to differentiate AH from nonsecreting adenomas, but no comparative study has been performed to date. MATERIALS AND METHODS: This was a case-control study of 402 patients, which compared the presurgical pituitary MR imaging features of patients with nonsecreting pituitary adenoma and controls with AH. MR images were compared on the basis of 16 morphologic features besides sex, age, and relation to pregnancy. RESULTS: Only 2 of the 19 proposed features tested lacked prognostic value. When the other 17 predictors were analyzed jointly in a multiple logistic regression model, 8 (relation to pregnancy, pituitary mass volume and symmetry, signal intensity and signal intensity homogeneity after gadolin-ium administration, posterior pituitary bright spot presence, stalk size, and mucosal swelling) remained significant predictors of a correct classification. The diagnostic score had a global performance of 0.9917 and correctly classified 97 % of the patients, with a sensitivity of 92%, a specificity of 99%,
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ORIGINAL ARTICLE Blackwell Publishing Ltd
2014Co-Authors: Patrizio CaturegliAbstract:Background Pituitary autoantibodies are found in Autoimmune Hypophysitis and other conditions. They are a marker of pituitary autoimmunity but currently have limited clinical value. The methods used for their detection lack adequate sensitivity and specificity, mainly because the pathogenic pituitary autoantigen(s) are not known and therefore antigen-based immunoassays have not been developed. Objectives This study aimed to identify novel pituitary autoantigens using sera as probes in proteomic assays. We also compared immunoblotting and immunofluorescence methods for their accuracy in diagnosing Autoimmune Hypophysitis. Study design and subjects Twenty-eight sera from Autoimmune Hypophysitis cases (14 histologically proven and 14 clinically suspected) were compared to 98 sera from controls, which included 14 patients with pituitary adenomas, 48 with Autoimmune thyroiditi
Isabella Lupi - One of the best experts on this subject based on the ideXlab platform.
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diabetes insipidus is an unfavorable prognostic factor for response to glucocorticoids in patients with Autoimmune Hypophysitis
European Journal of Endocrinology, 2017Co-Authors: Isabella Lupi, Patrizio Caturegli, Enio Martino, Luca Manetti, Mirco Cosottini, Claudio Urbani, Daniele Cappellani, Ilaria Scattina, Claudio Marcocci, Fausto BogazziAbstract:Introduction Autoimmune Hypophysitis (AH) has a variable clinical presentation and natural history; likewise, its response to glucocorticoid therapy is often unpredictable. Objective To identify clinical and radiological findings associated with response to glucocorticoids. Design and methods 12 consecutive patients with AH, evaluated from 2008 to 2016. AH was the exclusion diagnosis after ruling out other pituitary masses and secondary causes of Hypophysitis. Mean follow-up time was 30 ± 27 months (range 12-96 months). Results MRI identified two main patterns of presentation: global enlargement of the pituitary gland or panHypophysitis (n = 4, PH), and pituitary stalk abnormality only, or infundibulo-neuro-Hypophysitis (n = 8, INH). Multiple tropin defects were more common in PH (100%) than those in INH (28% P = 0.014), whereas diabetes insipidus was more common in INH (100%) than that in PH (50%; P = 0.028). All 4 PH and 4 out of 8 INH were treated with glucocorticoids. Pituitary volume significantly reduced in all PH patients (P = 0.012), defective anterior pituitary function recovered only in the two patients without diabetes insipidus (50%) and panhypopituitarism persisted, along with diabetes insipidus, in the remaining 2 (50%). In all INH patients, either treated or untreated, pituitary stalk diameter reduced (P = 0.008) but diabetes insipidus persisted in all. Conclusions Glucocorticoid therapy may improve anterior pituitary function in a subset of patients but has no effect on restoring posterior pituitary function. Diabetes insipidus appears as a negative prognostic factor for response to glucocorticoids.
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Prevalence and Functional Significance of Antipituitary Antibodies in Patients with Autoimmune and Non- Autoimmune Thyroid
2015Co-Authors: Luca Manetti, Isabella Lupi, Mirco Cosottini, Stefano Mariotti, Giovanni Pinna, Lisa L. Morselli, Sonia Albertini, Lucia Grasso, Maura Genovesi, Fausto BogazziAbstract:Background: Circulating antipituitary antibodies (APA) are mark-ers of Autoimmune Hypophysitis, which may cause deficient pituitary function. The prevalence of APA in Autoimmune thyroid disorders (AITD) is uncertain. Objectives: The aims of this study were 1) to evaluate APA preva-lence in a large series of patients with AITD and non-AITD and 2) to investigate the functional significance of APA by assessing pituitary function in APA-positive patients. Design and Setting: We conducted a health survey on consecutive AITD and non-AITD patients at a tertiary referral center (Depart-ment of Endocrinology, Pisa). Patients: Subjects, including 1290 consecutive patients with thyroid disorders (961 AITD and 329 non-AITD) and 135 controls, were en-rolled in the study. Methods: APA (indirect immunofluorescence), free T4, free T3, TSH
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pituitary and systemic autoimmunity in a case of intrasellar germinoma
Pituitary, 2011Co-Authors: Angelika Gutenberg, Shey Cherng Tzou, Isabella Lupi, Roberto Salvatori, Hiroaki Kimura, Melissa A Landeksalgado, Jennifer J Bell, Lefkothea Karaviti, Patrizio CaturegliAbstract:Germinomas arising in the sella turcica are difficult to differentiate from Autoimmune Hypophysitis because of similar clinical and pathological features. This differentiation, nevertheless, is critical for patient care due to different treatments of the two diseases. We report the case of an 11-year-old girl who presented with diabetes insipidus and growth retardation, and was found to have an intra- and supra-sellar mass. Initial examination of the pituitary biopsy showed diffuse lymphocytic infiltration of the adenohypophysis and absent placental alkaline phosphatase expression, leading to a diagnosis of Hypophysitis and glucocorticoid treatment. Because of the lack of clinical and radiological response, the pituitary specimen was re-examined, revealing this time the presence of scattered c-kit and Oct4 positive germinoma cells. The revised diagnosis prompted the initiation of radiotherapy, which induced disappearance of the pituitary mass. Immunological studies showed that the patient’s serum recognized antigens expressed by the patient’s own germinoma cells, as well as pituitary antigens like growth hormone and systemic antigens like the Sjogren syndrome antigen B and alpha-enolase. The study first reports the presence of pituitary and systemic antibodies in a patient with intrasellar germinoma, and reminds us that diffuse lymphocytic infiltration of the pituitary gland and pituitary antibodies does not always indicate a diagnosis of Autoimmune Hypophysitis.
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from pituitary expansion to empty sella disease progression in a mouse model of Autoimmune Hypophysitis
Endocrinology, 2011Co-Authors: Isabella Lupi, Shey Cherng Tzou, Angelika Gutenberg, Melissa A Landeksalgado, Jiangyang Zhang, Susumu Mori, Patrizio CaturegliAbstract:Lymphocytic Hypophysitis has a variable clinical course, where a swelling of the pituitary gland at presentation is thought to be followed by pituitary atrophy and empty sella. Data in patients, however, are scanty and contradictory. To better define the course of Hypophysitis, we used an experimental model based on the injection of pituitary proteins into SJL mice. A cohort of 33 mice was divided into three groups: 18 cases were immunized with pituitary proteins emulsified in complete Freund's adjuvant; six controls were injected with adjuvant only; and nine controls were left untreated. Mice were followed by cranial magnetic resonance imaging (MRI) for up to 300 d, for a total of 106 MRI scans, and killed at different time points to correlate radiological and pathological findings. Empty sella was defined as a reduction in pituitary volume greater than 2 sd below the mean volume. All immunized mice showed by MRI a significant expansion of pituitary volume during the early phases of the disease. The volu...
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from pituitary expansion to empty sella disease progression in a mouse model of Autoimmune Hypophysitis
Endocrinology, 2011Co-Authors: Isabella Lupi, Shey Cherng Tzou, Angelika Gutenberg, Melissa A Landeksalgado, Jiangyang Zhang, Susumu Mori, Patrizio CaturegliAbstract:Lymphocytic Hypophysitis has a variable clinical course, where a swelling of the pituitary gland at presentation is thought to be followed by pituitary atrophy and empty sella. Data in patients, however, are scanty and contradictory. To better define the course of Hypophysitis, we used an experimental model based on the injection of pituitary proteins into SJL mice. A cohort of 33 mice was divided into three groups: 18 cases were immunized with pituitary proteins emulsified in complete Freund's adjuvant; six controls were injected with adjuvant only; and nine controls were left untreated. Mice were followed by cranial magnetic resonance imaging (MRI) for up to 300 d, for a total of 106 MRI scans, and killed at different time points to correlate radiological and pathological findings. Empty sella was defined as a reduction in pituitary volume greater than 2 sd below the mean volume. All immunized mice showed by MRI a significant expansion of pituitary volume during the early phases of the disease. The volume then decreased gradually in the majority of cases (14 of 18, 78%), reaching empty sella values by d 300 after immunization. In a minority of cases (four of 18, 22%), the decrease was so rapid and marked to induce a central area of necrosis accompanied by hemorrhages, mimicking the condition known in patients as pituitary apoplexy. No radiological or pathological changes were observed in controls. Overall, these findings indicate that the evolution of Hypophysitis is complex but can lead, through different routes, to the development of empty sella.
Angelika Gutenberg - One of the best experts on this subject based on the ideXlab platform.
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in situ activation of pituitary infiltrating t lymphocytes in Autoimmune Hypophysitis
Scientific Reports, 2017Co-Authors: Han Huei Lin, Angelika Gutenberg, Patrizio Caturegli, Tzu Yu Chen, Nu Man Tsai, Chiajung Lee, Yu Che Cheng, Wen Hui Cheng, Ywhmin Tzou, Shey Cherng TzouAbstract:Autoimmune Hypophysitis (AH) is a chronic inflammatory disease characterized by infiltration of T and B lymphocytes in the pituitary gland. The mechanisms through which infiltrating lymphocytes cause disease remain unknown. Using a mouse model of AH we assessed whether T lymphocytes undergo activation in the pituitary gland. Infiltrating T cells co-localized with dendritic cells in the pituitary and produced increased levels of interferon-γ and interleukin-17 upon stimulation in vitro. Assessing proliferation of CD3- and B220-postive lymphocytes by double immunohistochemistry (PCNA-staining) and flow cytometry (BrdU incorporation) revealed that a discrete proportion of infiltrating T cells and B cells underwent proliferation within the pituitary parenchyma. This proliferation persisted into the late disease stage (day 56 post-immunization), indicating the presence of a continuous generation of autoreactive T and B cells within the pituitary gland. T cell proliferation in the pituitary was confirmed in patients affected by Autoimmune Hypophysitis. In conclusion, we show that pituitary-infiltrating lymphocytes proliferate in situ during AH, providing a previously unknown pathogenic mechanism and new avenues for treatment.
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ORIGINAL RESEARCH A Radiologic Score to Distinguish Autoimmune Hypophysitis from Nonsecreting Pituitary
2015Co-Authors: Adenoma Preoperatively, Angelika Gutenberg, J Larsen, Veit Rohde, I. Lupi, Patrizio CaturegliAbstract:BACKGROUND AND PURPOSE: Autoimmune Hypophysitis (AH) mimics the more common nonsecret-ing pituitary adenomas and can be diagnosed with certainty only histologically. Approximately 40 % of patients with AH are still misdiagnosed as having pituitary macroadenoma and undergo unnecessary surgery. MR imaging is currently the best noninvasive diagnostic tool to differentiate AH from nonsecreting adenomas, though no single radiologic sign is diagnostically accurate. The purpose of this study was to develop a scoring system that summarizes numerous MR imaging signs to increase the probability of diagnosing AH before surgery. MATERIALS AND METHODS: This was a case-control study of 402 patients, which compared the presurgical pituitary MR imaging features of patients with nonsecreting pituitary adenoma and controls with AH. MR images were compared on the basis of 16 morphologic features besides sex, age, and relation to pregnancy. RESULTS: Only 2 of the 19 proposed features tested lacked prognostic value. When the other 17 predictors were analyzed jointly in a multiple logistic regression model, 8 (relation to pregnancy, pituitary mass volume and symmetry, signal intensity and signal intensity homogeneity after gadolin-ium administration, posterior pituitary bright spot presence, stalk size, and mucosal swelling) remaine
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ORIGINAL RESEARCH A Radiologic Score to Distinguish Autoimmune Hypophysitis from Nonsecreting Pituitary
2015Co-Authors: Adenoma Preoperatively, Angelika Gutenberg, J Larsen, Veit Rohde, I. Lupi, Patrizio CaturegliAbstract:BACKGROUND AND PURPOSE: Autoimmune Hypophysitis (AH) mimics the more common nonsecret-ing pituitary adenomas and can be diagnosed with certainty only histologically. Approximately 40 % of patients with AH are still misdiagnosed as having pituitary macroadenoma and undergo unnecessary surgery. MR imaging is supposedly the best noninvasive diagnostic tool to differentiate AH from nonsecreting adenomas, but no comparative study has been performed to date. MATERIALS AND METHODS: This was a case-control study of 402 patients, which compared the presurgical pituitary MR imaging features of patients with nonsecreting pituitary adenoma and controls with AH. MR images were compared on the basis of 16 morphologic features besides sex, age, and relation to pregnancy. RESULTS: Only 2 of the 19 proposed features tested lacked prognostic value. When the other 17 predictors were analyzed jointly in a multiple logistic regression model, 8 (relation to pregnancy, pituitary mass volume and symmetry, signal intensity and signal intensity homogeneity after gadolin-ium administration, posterior pituitary bright spot presence, stalk size, and mucosal swelling) remained significant predictors of a correct classification. The diagnostic score had a global performance of 0.9917 and correctly classified 97 % of the patients, with a sensitivity of 92%, a specificity of 99%,
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pituitary and systemic autoimmunity in a case of intrasellar germinoma
Pituitary, 2011Co-Authors: Angelika Gutenberg, Shey Cherng Tzou, Isabella Lupi, Roberto Salvatori, Hiroaki Kimura, Melissa A Landeksalgado, Jennifer J Bell, Lefkothea Karaviti, Patrizio CaturegliAbstract:Germinomas arising in the sella turcica are difficult to differentiate from Autoimmune Hypophysitis because of similar clinical and pathological features. This differentiation, nevertheless, is critical for patient care due to different treatments of the two diseases. We report the case of an 11-year-old girl who presented with diabetes insipidus and growth retardation, and was found to have an intra- and supra-sellar mass. Initial examination of the pituitary biopsy showed diffuse lymphocytic infiltration of the adenohypophysis and absent placental alkaline phosphatase expression, leading to a diagnosis of Hypophysitis and glucocorticoid treatment. Because of the lack of clinical and radiological response, the pituitary specimen was re-examined, revealing this time the presence of scattered c-kit and Oct4 positive germinoma cells. The revised diagnosis prompted the initiation of radiotherapy, which induced disappearance of the pituitary mass. Immunological studies showed that the patient’s serum recognized antigens expressed by the patient’s own germinoma cells, as well as pituitary antigens like growth hormone and systemic antigens like the Sjogren syndrome antigen B and alpha-enolase. The study first reports the presence of pituitary and systemic antibodies in a patient with intrasellar germinoma, and reminds us that diffuse lymphocytic infiltration of the pituitary gland and pituitary antibodies does not always indicate a diagnosis of Autoimmune Hypophysitis.
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from pituitary expansion to empty sella disease progression in a mouse model of Autoimmune Hypophysitis
Endocrinology, 2011Co-Authors: Isabella Lupi, Shey Cherng Tzou, Angelika Gutenberg, Melissa A Landeksalgado, Jiangyang Zhang, Susumu Mori, Patrizio CaturegliAbstract:Lymphocytic Hypophysitis has a variable clinical course, where a swelling of the pituitary gland at presentation is thought to be followed by pituitary atrophy and empty sella. Data in patients, however, are scanty and contradictory. To better define the course of Hypophysitis, we used an experimental model based on the injection of pituitary proteins into SJL mice. A cohort of 33 mice was divided into three groups: 18 cases were immunized with pituitary proteins emulsified in complete Freund's adjuvant; six controls were injected with adjuvant only; and nine controls were left untreated. Mice were followed by cranial magnetic resonance imaging (MRI) for up to 300 d, for a total of 106 MRI scans, and killed at different time points to correlate radiological and pathological findings. Empty sella was defined as a reduction in pituitary volume greater than 2 sd below the mean volume. All immunized mice showed by MRI a significant expansion of pituitary volume during the early phases of the disease. The volu...
Shey Cherng Tzou - One of the best experts on this subject based on the ideXlab platform.
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in situ activation of pituitary infiltrating t lymphocytes in Autoimmune Hypophysitis
Scientific Reports, 2017Co-Authors: Han Huei Lin, Angelika Gutenberg, Patrizio Caturegli, Tzu Yu Chen, Nu Man Tsai, Chiajung Lee, Yu Che Cheng, Wen Hui Cheng, Ywhmin Tzou, Shey Cherng TzouAbstract:Autoimmune Hypophysitis (AH) is a chronic inflammatory disease characterized by infiltration of T and B lymphocytes in the pituitary gland. The mechanisms through which infiltrating lymphocytes cause disease remain unknown. Using a mouse model of AH we assessed whether T lymphocytes undergo activation in the pituitary gland. Infiltrating T cells co-localized with dendritic cells in the pituitary and produced increased levels of interferon-γ and interleukin-17 upon stimulation in vitro. Assessing proliferation of CD3- and B220-postive lymphocytes by double immunohistochemistry (PCNA-staining) and flow cytometry (BrdU incorporation) revealed that a discrete proportion of infiltrating T cells and B cells underwent proliferation within the pituitary parenchyma. This proliferation persisted into the late disease stage (day 56 post-immunization), indicating the presence of a continuous generation of autoreactive T and B cells within the pituitary gland. T cell proliferation in the pituitary was confirmed in patients affected by Autoimmune Hypophysitis. In conclusion, we show that pituitary-infiltrating lymphocytes proliferate in situ during AH, providing a previously unknown pathogenic mechanism and new avenues for treatment.
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pituitary and systemic autoimmunity in a case of intrasellar germinoma
Pituitary, 2011Co-Authors: Angelika Gutenberg, Shey Cherng Tzou, Isabella Lupi, Roberto Salvatori, Hiroaki Kimura, Melissa A Landeksalgado, Jennifer J Bell, Lefkothea Karaviti, Patrizio CaturegliAbstract:Germinomas arising in the sella turcica are difficult to differentiate from Autoimmune Hypophysitis because of similar clinical and pathological features. This differentiation, nevertheless, is critical for patient care due to different treatments of the two diseases. We report the case of an 11-year-old girl who presented with diabetes insipidus and growth retardation, and was found to have an intra- and supra-sellar mass. Initial examination of the pituitary biopsy showed diffuse lymphocytic infiltration of the adenohypophysis and absent placental alkaline phosphatase expression, leading to a diagnosis of Hypophysitis and glucocorticoid treatment. Because of the lack of clinical and radiological response, the pituitary specimen was re-examined, revealing this time the presence of scattered c-kit and Oct4 positive germinoma cells. The revised diagnosis prompted the initiation of radiotherapy, which induced disappearance of the pituitary mass. Immunological studies showed that the patient’s serum recognized antigens expressed by the patient’s own germinoma cells, as well as pituitary antigens like growth hormone and systemic antigens like the Sjogren syndrome antigen B and alpha-enolase. The study first reports the presence of pituitary and systemic antibodies in a patient with intrasellar germinoma, and reminds us that diffuse lymphocytic infiltration of the pituitary gland and pituitary antibodies does not always indicate a diagnosis of Autoimmune Hypophysitis.
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from pituitary expansion to empty sella disease progression in a mouse model of Autoimmune Hypophysitis
Endocrinology, 2011Co-Authors: Isabella Lupi, Shey Cherng Tzou, Angelika Gutenberg, Melissa A Landeksalgado, Jiangyang Zhang, Susumu Mori, Patrizio CaturegliAbstract:Lymphocytic Hypophysitis has a variable clinical course, where a swelling of the pituitary gland at presentation is thought to be followed by pituitary atrophy and empty sella. Data in patients, however, are scanty and contradictory. To better define the course of Hypophysitis, we used an experimental model based on the injection of pituitary proteins into SJL mice. A cohort of 33 mice was divided into three groups: 18 cases were immunized with pituitary proteins emulsified in complete Freund's adjuvant; six controls were injected with adjuvant only; and nine controls were left untreated. Mice were followed by cranial magnetic resonance imaging (MRI) for up to 300 d, for a total of 106 MRI scans, and killed at different time points to correlate radiological and pathological findings. Empty sella was defined as a reduction in pituitary volume greater than 2 sd below the mean volume. All immunized mice showed by MRI a significant expansion of pituitary volume during the early phases of the disease. The volu...
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from pituitary expansion to empty sella disease progression in a mouse model of Autoimmune Hypophysitis
Endocrinology, 2011Co-Authors: Isabella Lupi, Shey Cherng Tzou, Angelika Gutenberg, Melissa A Landeksalgado, Jiangyang Zhang, Susumu Mori, Patrizio CaturegliAbstract:Lymphocytic Hypophysitis has a variable clinical course, where a swelling of the pituitary gland at presentation is thought to be followed by pituitary atrophy and empty sella. Data in patients, however, are scanty and contradictory. To better define the course of Hypophysitis, we used an experimental model based on the injection of pituitary proteins into SJL mice. A cohort of 33 mice was divided into three groups: 18 cases were immunized with pituitary proteins emulsified in complete Freund's adjuvant; six controls were injected with adjuvant only; and nine controls were left untreated. Mice were followed by cranial magnetic resonance imaging (MRI) for up to 300 d, for a total of 106 MRI scans, and killed at different time points to correlate radiological and pathological findings. Empty sella was defined as a reduction in pituitary volume greater than 2 sd below the mean volume. All immunized mice showed by MRI a significant expansion of pituitary volume during the early phases of the disease. The volume then decreased gradually in the majority of cases (14 of 18, 78%), reaching empty sella values by d 300 after immunization. In a minority of cases (four of 18, 22%), the decrease was so rapid and marked to induce a central area of necrosis accompanied by hemorrhages, mimicking the condition known in patients as pituitary apoplexy. No radiological or pathological changes were observed in controls. Overall, these findings indicate that the evolution of Hypophysitis is complex but can lead, through different routes, to the development of empty sella.
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Autoimmune Hypophysitis expanding the differential diagnosis to ctla 4 blockade
Expert Review of Endocrinology & Metabolism, 2009Co-Authors: Angelika Gutenberg, Shey Cherng Tzou, Isabella Lupi, Hiroaki Kimura, Melissa A Landeksalgado, Abby L Geis, Patrizio CaturegliAbstract:Autoimmune Hypophysitis is an increasingly recognized disorder that enters in the differential diagnosis of nonfunctioning pituitary masses. The differential diagnosis of these conditions is challenging because of similar clinical presentations and radiological signs. This review describes the essential features of Hypophysitis and the other nonfunctioning pituitary masses. It also emphasizes a recently described feature of Hypophysitis: its appearance with unexpectedly high frequency in patients receiving treatments that abrogate the function of cytotoxic T lymphocyte antigen 4.
Melissa A Landeksalgado - One of the best experts on this subject based on the ideXlab platform.
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pituitary and systemic autoimmunity in a case of intrasellar germinoma
Pituitary, 2011Co-Authors: Angelika Gutenberg, Shey Cherng Tzou, Isabella Lupi, Roberto Salvatori, Hiroaki Kimura, Melissa A Landeksalgado, Jennifer J Bell, Lefkothea Karaviti, Patrizio CaturegliAbstract:Germinomas arising in the sella turcica are difficult to differentiate from Autoimmune Hypophysitis because of similar clinical and pathological features. This differentiation, nevertheless, is critical for patient care due to different treatments of the two diseases. We report the case of an 11-year-old girl who presented with diabetes insipidus and growth retardation, and was found to have an intra- and supra-sellar mass. Initial examination of the pituitary biopsy showed diffuse lymphocytic infiltration of the adenohypophysis and absent placental alkaline phosphatase expression, leading to a diagnosis of Hypophysitis and glucocorticoid treatment. Because of the lack of clinical and radiological response, the pituitary specimen was re-examined, revealing this time the presence of scattered c-kit and Oct4 positive germinoma cells. The revised diagnosis prompted the initiation of radiotherapy, which induced disappearance of the pituitary mass. Immunological studies showed that the patient’s serum recognized antigens expressed by the patient’s own germinoma cells, as well as pituitary antigens like growth hormone and systemic antigens like the Sjogren syndrome antigen B and alpha-enolase. The study first reports the presence of pituitary and systemic antibodies in a patient with intrasellar germinoma, and reminds us that diffuse lymphocytic infiltration of the pituitary gland and pituitary antibodies does not always indicate a diagnosis of Autoimmune Hypophysitis.
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from pituitary expansion to empty sella disease progression in a mouse model of Autoimmune Hypophysitis
Endocrinology, 2011Co-Authors: Isabella Lupi, Shey Cherng Tzou, Angelika Gutenberg, Melissa A Landeksalgado, Jiangyang Zhang, Susumu Mori, Patrizio CaturegliAbstract:Lymphocytic Hypophysitis has a variable clinical course, where a swelling of the pituitary gland at presentation is thought to be followed by pituitary atrophy and empty sella. Data in patients, however, are scanty and contradictory. To better define the course of Hypophysitis, we used an experimental model based on the injection of pituitary proteins into SJL mice. A cohort of 33 mice was divided into three groups: 18 cases were immunized with pituitary proteins emulsified in complete Freund's adjuvant; six controls were injected with adjuvant only; and nine controls were left untreated. Mice were followed by cranial magnetic resonance imaging (MRI) for up to 300 d, for a total of 106 MRI scans, and killed at different time points to correlate radiological and pathological findings. Empty sella was defined as a reduction in pituitary volume greater than 2 sd below the mean volume. All immunized mice showed by MRI a significant expansion of pituitary volume during the early phases of the disease. The volu...
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from pituitary expansion to empty sella disease progression in a mouse model of Autoimmune Hypophysitis
Endocrinology, 2011Co-Authors: Isabella Lupi, Shey Cherng Tzou, Angelika Gutenberg, Melissa A Landeksalgado, Jiangyang Zhang, Susumu Mori, Patrizio CaturegliAbstract:Lymphocytic Hypophysitis has a variable clinical course, where a swelling of the pituitary gland at presentation is thought to be followed by pituitary atrophy and empty sella. Data in patients, however, are scanty and contradictory. To better define the course of Hypophysitis, we used an experimental model based on the injection of pituitary proteins into SJL mice. A cohort of 33 mice was divided into three groups: 18 cases were immunized with pituitary proteins emulsified in complete Freund's adjuvant; six controls were injected with adjuvant only; and nine controls were left untreated. Mice were followed by cranial magnetic resonance imaging (MRI) for up to 300 d, for a total of 106 MRI scans, and killed at different time points to correlate radiological and pathological findings. Empty sella was defined as a reduction in pituitary volume greater than 2 sd below the mean volume. All immunized mice showed by MRI a significant expansion of pituitary volume during the early phases of the disease. The volume then decreased gradually in the majority of cases (14 of 18, 78%), reaching empty sella values by d 300 after immunization. In a minority of cases (four of 18, 22%), the decrease was so rapid and marked to induce a central area of necrosis accompanied by hemorrhages, mimicking the condition known in patients as pituitary apoplexy. No radiological or pathological changes were observed in controls. Overall, these findings indicate that the evolution of Hypophysitis is complex but can lead, through different routes, to the development of empty sella.
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pregnancy postpartum Autoimmune thyroiditis and Autoimmune Hypophysitis intimate relationships
Autoimmunity Reviews, 2010Co-Authors: Melissa A Landeksalgado, Angelika Gutenberg, Isabella Lupi, Patrizio Caturegli, Hiroaki Kimura, Stefano Mariotti, Noel R RoseAbstract:Autoimmune diseases comprise a group of about 85 heterogeneous conditions that can affect virtually any organ and tissue in the body. Many Autoimmune diseases change significantly during pregnancy: some ameliorate, some worsen, and others are unaffected. Two Autoimmune diseases present prominently in relation to pregnancy: postpartum Autoimmune thyroiditis and Autoimmune Hypophysitis. This article will review the current state of knowledge of the immunological changes that occur during normal pregnancy, and will explore the striking temporal association with pregnancy observed in thyroiditis and Hypophysitis.
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Autoimmune Hypophysitis expanding the differential diagnosis to ctla 4 blockade
Expert Review of Endocrinology & Metabolism, 2009Co-Authors: Angelika Gutenberg, Shey Cherng Tzou, Isabella Lupi, Hiroaki Kimura, Melissa A Landeksalgado, Abby L Geis, Patrizio CaturegliAbstract:Autoimmune Hypophysitis is an increasingly recognized disorder that enters in the differential diagnosis of nonfunctioning pituitary masses. The differential diagnosis of these conditions is challenging because of similar clinical presentations and radiological signs. This review describes the essential features of Hypophysitis and the other nonfunctioning pituitary masses. It also emphasizes a recently described feature of Hypophysitis: its appearance with unexpectedly high frequency in patients receiving treatments that abrogate the function of cytotoxic T lymphocyte antigen 4.
