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6. Characteristics and outcomes of Italian patients from the observational, multicentre, hypopituitary control and complications study (HypoCCS) according to tertiles of growth hormone peak concentration following stimulation testing at study entry

Author

Losa, Marco, Beck-Peccoz, Paolo, Aimaretti, Gianluca, Di Somma, Carolina, Ambrosio, Maria Rosaria, Ferone, Diego, Giampietro, Antonella, Corsello, Salvatore M., Poggi, Maurizio, Scaroni, Carla, Jia, Nan, Mossetto, Gilberto, Cannavò, Salvatore, and Rochira, Vincenzo

Published
2015
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9. Secondary adrenal insufficiency: From the physiopathology to the possible role of modified-release hydrocortisone treatment

Author

Paragliola, Rosa M., Corsello, Salvatore M., Paragliola, Rosa M. (ORCID:0000-0002-5070-7771), Corsello, Salvatore M. (ORCID:0000-0002-4544-7274), Paragliola, Rosa M., Corsello, Salvatore M., Paragliola, Rosa M. (ORCID:0000-0002-5070-7771), and Corsello, Salvatore M. (ORCID:0000-0002-4544-7274)

Abstract

due to an impairment production of adrenocorticotropic hormone (ACTH). In particular, secondary AI results from pituitary disease that impedes the release of ACTH, while tertiary AI is caused from an impaired synthesis of corticotropinreleasing hormone. Central AI has an estimated prevalence of 150-280 per million, resulting more common than primary AI. Prompt diagnosis and management of this condition is crucial, but the diagnostic investigation can often be challenging, in particular in cases of recent onset of secondary adrenal insufficiency. Moreover, different formulations of steroid replacement therapy are available for both primary and central adrenal insufficiency, but the therapy of choice for the treatment of secondary hypoadrenalism is still debated. In particular, several data confirm the advantages of dual-release hydrocortisone formulation in primary hypoadrenalism, while data for secondary AI are still lacking. However, in spite of few clinical data, the use of dual release hydrocortisone can be extremely favorable in ACTH deficiency, which is associated with an increase of overall and cardiovascular mortality, also due to the risk of overtreatment. In this condition, the use of a modified-release glucocorticoid formulation, which has been demonstrated to improve metabolic profile, can be extremely attractive.

Published
2018

10. Current management and outcome of pregnancies in women with adrenal insufficiency: experience from a multi-center survey

Author

Bothou, Christina, Anand, Gurpreet, Li, Dingfeng, Kienitz, Tina, Seejore, Khyatisha, Simeoli, Chiara, Ebbehoj, Andreas, Ward, Emma G, Paragliola, Rosa Maria, Ferrigno, Rosario, Badenhoop, Klaus, Bensing, Sophie, Oksnes, Marianne, Esposito, Daniela, Bergthorsdottir, Ragnhildur, Drake, William, Wahlberg, Jeanette, Reisch, Nicole, Hahner, Stefanie, Pearce, Simon, Trainer, Peter, Etzrodt-Walter, Gwendolin, Thalmann, Sébastien P, Sævik, Åse B, Husebye, Eystein, Isidori, Andrea M, Falhammar, Henrik, Meyer, Gesine, Corsello, Salvatore M, Pivonello, Rosario, et al, and University of Zurich

Subjects
2712 Endocrinology, Diabetes and Metabolism, 1303 Biochemistry, 10265 Clinic for Endocrinology and Diabetology, 610 Medicine & health, 1308 Clinical Biochemistry, 2704 Biochemistry (medical), 1310 Endocrinology
Published
2020
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11. Current Management and Outcome of Pregnancies in Women With Adrenal Insufficiency : Experience from a Multicenter Survey

Author

Bothou, Christina, Anand, Gurpreet, Li, Dingfeng, Kienitz, Tina, Seejore, Khyatisha, Simeoli, Chiara, Ebbehoj, Andreas, Ward, Emma G., Paragliola, Rosa Maria, Ferrigno, Rosario, Badenhoop, Klaus, Bensing, Sophie, Oksnes, Marianne, Esposito, Daniela, Bergthorsdottir, Ragnhildur, Drake, William, Wahlberg, Jeanette, Reisch, Nicole, Hahner, Stefanie, Pearce, Simon, Trainer, Peter, Etzrodt-Walter, Gwendolin, Thalmann, Sébastien P., Sævik, Åse B., Husebye, Eystein, Isidori, Andrea M., Falhammar, Henrik, Meyer, Gesine, Corsello, Salvatore M., Pivonello, Rosario, Murray, Robert, Bancos, Irina, Quinkler, Marcus, Beuschlein, Felix, Bothou, Christina, Anand, Gurpreet, Li, Dingfeng, Kienitz, Tina, Seejore, Khyatisha, Simeoli, Chiara, Ebbehoj, Andreas, Ward, Emma G., Paragliola, Rosa Maria, Ferrigno, Rosario, Badenhoop, Klaus, Bensing, Sophie, Oksnes, Marianne, Esposito, Daniela, Bergthorsdottir, Ragnhildur, Drake, William, Wahlberg, Jeanette, Reisch, Nicole, Hahner, Stefanie, Pearce, Simon, Trainer, Peter, Etzrodt-Walter, Gwendolin, Thalmann, Sébastien P., Sævik, Åse B., Husebye, Eystein, Isidori, Andrea M., Falhammar, Henrik, Meyer, Gesine, Corsello, Salvatore M., Pivonello, Rosario, Murray, Robert, Bancos, Irina, Quinkler, Marcus, and Beuschlein, Felix

Abstract

Context: Appropriate management of adrenal insufficiency (AI) in pregnancy can be challenging due to the rarity of the disease and lack of evidence-based recommendations to guide glucocorticoid and mineralocorticoid dosage adjustment. Objective: Multicenter survey on current clinical approaches in managing AI during pregnancy. Design: Retrospective anonymized data collection from 19 international centers from 2013 to 2019. Setting and patients: 128 pregnancies in 113 women with different causes of AI: Addison disease (44%), secondary AI (25%), congenital adrenal hyperplasia (25%), and acquired AI due to bilateral adrenalectomy (6%). Results: Hydrocortisone (HC) was the most commonly used glucocorticoid in 83% (97/117) of pregnancies. Glucocorticoid dosage was increased at any time during pregnancy in 73/128 (57%) of cases. In these cases, the difference in the daily dose of HC equivalent between baseline and the third trimester was 8.6 ± 5.4 (range 1-30) mg. Fludrocortisone dosage was increased in fewer cases (7/54 during the first trimester, 9/64 during the second trimester, and 9/62 cases during the third trimester). Overall, an adrenal crisis was reported in 9/128 (7%) pregnancies. Cesarean section was the most frequent mode of delivery at 58% (69/118). Fetal complications were reported in 3/120 (3%) and minor maternal complications in 15/120 (13%) pregnancies without fatal outcomes. Conclusions: This survey confirms good maternal and fetal outcome in women with AI managed in specialized endocrine centers. An emphasis on careful endocrine follow-up and repeated patient education is likely to have reduced the risk of adrenal crisis and resulted in positive outcomes., Funding Agencies:James A. Ruppe Career Development Award in EndocrinologyCatalyst Award for Advancing in Academics from Mayo Clinic

Published
2020
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12. Subclinical Cushing Syndrome: How To Predict Post-Surgical Hypoadrenalism

Author

Ricciato, Maria Pia, primary, Di Donna, Vincenzo, additional, Ianni, Francesca, additional, De Rosa, Annapina, additional, Gallo, Francesca, additional, Senes, Paola, additional, Castellino, Laura, additional, Raffaelli, Marco, additional, Lombardi, Celestino Pio, additional, Bellantone, Rocco, additional, Pontecorvi, Alfredo, additional, and Corsello, Salvatore M, additional

Published
2011
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14. An Old Tool for an Effective Diagnosis of Thyroid Autonomy

Author

Ianni, Francesca, primary, Perotti, Germano, additional, Paragliola, Rosa Maria, additional, Ricciato, Maria Pia, additional, Castellino, Laura, additional, Prete, Alessandro, additional, Luotto, Valentina, additional, Pontecorvi, Alfredo, additional, and Corsello, Salvatore M, additional

Published
2011
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16. Current Management and Outcome of Pregnancies in Women With Adrenal Insufficiency: Experience from a Multicenter Survey

Author

Bothou, Christina, primary, Anand, Gurpreet, additional, Li, Dingfeng, additional, Kienitz, Tina, additional, Seejore, Khyatisha, additional, Simeoli, Chiara, additional, Ebbehoj, Andreas, additional, Ward, Emma G, additional, Paragliola, Rosa Maria, additional, Ferrigno, Rosario, additional, Badenhoop, Klaus, additional, Bensing, Sophie, additional, Oksnes, Marianne, additional, Esposito, Daniela, additional, Bergthorsdottir, Ragnhildur, additional, Drake, William, additional, Wahlberg, Jeanette, additional, Reisch, Nicole, additional, Hahner, Stefanie, additional, Pearce, Simon, additional, Trainer, Peter, additional, Etzrodt-Walter, Gwendolin, additional, Thalmann, Sébastien P, additional, Sævik, Åse B, additional, Husebye, Eystein, additional, Isidori, Andrea M, additional, Falhammar, Henrik, additional, Meyer, Gesine, additional, Corsello, Salvatore M, additional, Pivonello, Rosario, additional, Murray, Robert, additional, Bancos, Irina, additional, Quinkler, Marcus, additional, and Beuschlein, Felix, additional

Published
2020
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17. Management and outcome of pregnancies in women with adrenal insufficiency: experience from a retrospective European study

Author

Bothou, Christina, primary, Anand, Gurpreet, additional, Kienitz, Tina, additional, Seejore, Khyatisha, additional, Simeoli, Chiara, additional, Ward, Emma G, additional, Paragliola, Rosa Maria, additional, Ferrigno, Rosario, additional, Badenhoop, Klaus, additional, Bensing, Sophie, additional, Oksnes, Marianne, additional, Drake, William, additional, Wahlberg, Jeanette, additional, Reisch, Nicole, additional, Hahner, Stefanie, additional, Pearce, Simon, additional, Trainer, Peter, additional, Etzrodt-Walter, Gwendolin, additional, Thalmann, Sebastien P, additional, Saevik, AEse B, additional, Husebye, Eystein, additional, Isidori, Andrea M, additional, Falhammar, Henrik, additional, Meyer, Gesine, additional, Corsello, Salvatore M, additional, Pivonello, Rosario, additional, Murray, Robert, additional, Quinkler, Marcus, additional, and Beuschlein, Felix, additional

Published
2019
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18. Medullary thyroid carcinoma with exon 2 p. L56M RET variant: Clinical particular features in two patients

Author

Paragliola, Rosa Maria, Lovicu, Rosa M., Papi, Giampaolo, Capoluongo, Ettore Domenico, Minucci, Angelo, Canu, Giulia, Pontecorvi, Alfredo, Corsello, Salvatore Maria, Paragliola, Rosa M. (ORCID:0000-0002-5070-7771), Capoluongo, Ettore (ORCID:0000-0001-9872-0572), Pontecorvi, Alfredo (ORCID:0000-0003-0570-6865), Corsello, Salvatore M. (ORCID:0000-0002-4544-7274), Paragliola, Rosa Maria, Lovicu, Rosa M., Papi, Giampaolo, Capoluongo, Ettore Domenico, Minucci, Angelo, Canu, Giulia, Pontecorvi, Alfredo, Corsello, Salvatore Maria, Paragliola, Rosa M. (ORCID:0000-0002-5070-7771), Capoluongo, Ettore (ORCID:0000-0001-9872-0572), Pontecorvi, Alfredo (ORCID:0000-0003-0570-6865), and Corsello, Salvatore M. (ORCID:0000-0002-4544-7274)

Abstract

RET (REarranged during Transfection) proto-oncogene variants are essential for the development of familial and sporadic forms of medullary thyroid carcinoma (MTC). The most frequent variants are usually located in exons 10, 11, and 13 through 16 of the RET gene. We report two cases of apparently sporadic MTC associated with the variant in exon 2 of RET gene. Patient 1, a 62-year old man who had undergone adrenalectomy for a 5 cm pheochromocytoma, was screened for type 2 multiple endocrine neoplasia (MEN 2) which showed elevated basal and post-intravenous calcium gluconate calcitonin levels. A fine needle aspiration biopsy (FNAB) confirmed the suspicion of MTC. The patient underwent total thyroidectomy and lymphadenectomy, and the histology showed C-cell hyperplasia with medullary microcarcinoma. Patient 2, a 57 years old woman, underwent total thyroidectomy for toxic multinodular goiter. Pre-operative FNAB had shown benign features, while basal calcitonin levels were only borderline increased. Final histology revealed medullary multifocal microcarcinoma. Genetic testing for RET protoncogene on DNA extracted from peripheral blood was performed in both patients and a missense variant on exon 2 (c.166C > A, p. L56M) was identified. To our knowledge, these are the first time two cases of MTC associated to RET p. L56M variant. Interestingly, one patient had also a pheochromocytoma suggesting a possible pathogenetic role of this variant in the genesis of MEN2A. While the association of this variant with MTC or MEN2A has been never reported, it has been described in association with Hirschsprung's disease.

Published
2018

24. The hypertension of Cushing's syndrome

Author

Isidori, Andrea M., primary, Graziadio, Chiara, additional, Paragliola, Rosa Maria, additional, Cozzolino, Alessia, additional, Ambrogio, Alberto G., additional, Colao, Annamaria, additional, Corsello, Salvatore M., additional, and Pivonello, Rosario, additional

Published
2015
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28. Characteristics and outcomes of Italian patients from the observational, multicentre, hypopituitary control and complications study (Hypo CCS) according to tertiles of growth hormone peak concentration following stimulation testing at study entry.

Author

Losa, Marco, Beck‐Peccoz, Paolo, Aimaretti, Gianluca, Di Somma, Carolina, Ambrosio, Maria Rosaria, Ferone, Diego, Giampietro, Antonella, Corsello, Salvatore M., Poggi, Maurizio, Scaroni, Carla, Jia, Nan, Mossetto, Gilberto, Cannavò, Salvatore, and Rochira, Vincenzo

Subjects
HEALTH outcome assessment, SOMATOTROPIN, HYPERLIPIDEMIA, DISEASE prevalence, DIABETES, TRIGLYCERIDES
Abstract

Objective To determine whether characteristics and outcomes of Italian patients in the observational global Hypopituitary Control and Complication Study (Hypo CCS) differed according to the degree of GH deficiency ( GHD). Design Patients were grouped by tertiles of stimulated GH peak concentration at baseline (Group A lowest tertile, n = 342; Group B middle tertile, n = 345; Group C highest tertile, n = 338). Results Baseline demographics, lipid levels, body mass index categories and mean Framingham cardiovascular risk indexes were similar in the three groups and remained substantially unchanged over time, with no subsequent significant between-group differences (except mean levels of triglycerides increased in the highest tertile group). GHD was adult-onset for >75% of patients in all groups. The percentage of patients with multiple pituitary deficiencies was higher in Group A than in the other groups; isolated GHD was reported with highest frequency in Group C. Patients in Group A received the lowest mean starting dose of GH. Hyperlipidaemia at baseline was reported in 35·1%, 31·1% and 24·7% of patients in groups A, B and C, respectively ( P = 0·029). Mean duration of GH treatment was 7·21, 5·45 and 4·96 years, respectively. The proportion of patients with adverse events did not differ significantly between groups, with a low prevalence over time of diabetes and cancer. Conclusions In Italian patients from Hypo CCS, the level of GH deficit did not influence changes over time in metabolic parameters or adverse event profile, despite differences in GHD severity at baseline and in the starting GH dose. [ABSTRACT FROM AUTHOR]

Published
2015
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29. CT60 Single Nucleotide Polymorphisms of the Cytotoxic T-Lymphocyte–Associated Antigen-4 Gene Region is Associated with Graves' Disease in an Italian Population

Author

Petrone, Antonio, primary, Giorgi, Gabriele, additional, Galgani, Andrea, additional, Alemanno, Irene, additional, Corsello, Salvatore M., additional, Signore, Alberto, additional, Mario, Umberto Di, additional, Nisticò, Lorenza, additional, Cascino, Isabella, additional, and Buzzetti, Raffaella, additional

Published
2005
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37. Current management and outcome of pregnancies in women with adrenal insufficiency: experience from a multicenter survey

Author

Peter J Trainer, Nicole Reisch, Marianne Øksnes, Khyatisha Seejore, Dingfeng Li, Andreas Ebbehoj, Gurpreet Anand, Emma G Ward, Daniela Esposito, Rosario Ferrigno, Christina Bothou, Ragnhildur Bergthorsdottir, Sebastien P Thalmann, Eystein S. Husebye, Salvatore Maria Corsello, Robert D Murray, Marcus Quinkler, Andrea M. Isidori, Irina Bancos, Jeanette Wahlberg, Felix Beuschlein, Gwendolin Etzrodt-Walter, Henrik Falhammar, Åse Bjorvatn Sævik, Rosa Maria Paragliola, Tina Kienitz, Klaus Badenhoop, Rosario Pivonello, Gesine Meyer, Sophie Bensing, Chiara Simeoli, Simon H. S. Pearce, Stefanie Hahner, William Drake, Bothou, Christina, Anand, Gurpreet, Li, Dingfeng, Kienitz, Tina, Seejore, Khyatisha, Simeoli, Chiara, Ebbehoj, Andrea, Ward, Emma G, Paragliola, Rosa Maria, Ferrigno, Rosario, Badenhoop, Klau, Bensing, Sophie, Oksnes, Marianne, Esposito, Daniela, Bergthorsdottir, Ragnhildur, Drake, William, Wahlberg, Jeanette, Reisch, Nicole, Hahner, Stefanie, Pearce, Simon, Trainer, Peter, Etzrodt-Walter, Gwendolin, Thalmann, Sébastien P, Sævik, Åse B, Husebye, Eystein, Isidori, Andrea M, Falhammar, Henrik, Meyer, Gesine, Corsello, Salvatore M, Pivonello, Rosario, Murray, Robert, Bancos, Irina, Quinkler, Marcu, and Beuschlein, Felix

Subjects
Hydrocortisone, Endocrinology, Diabetes and Metabolism, Clinical Biochemistry, miscarriage, ADDISONS-DISEASE, Biochemistry, Severity of Illness Index, Miscarriage, 0302 clinical medicine, Endocrinology, adrenal crisi, mineralocorticoid, 030212 general & internal medicine, addison disease, RISK, Obstetrics, Pregnancy Outcome, Adrenal crisis, Addison disease, Treatment Outcome, Fludrocortisone, Female, pregnancy, medicine.symptom, medicine.drug, Adult, medicine.medical_specialty, adrenal crisis, Hormone Replacement Therapy, 030209 endocrinology & metabolism, Context (language use), DIAGNOSIS, 03 medical and health sciences, Internal medicine, Mineralocorticoids, medicine, Adrenal insufficiency, FERTILITY, Humans, congenital adrenal hyperplasia, Congenital adrenal hyperplasia, HYPERPLASIA, Online Only Articles, Glucocorticoids, Retrospective Studies, Pregnancy, Pregnancy outcomes, Dose-Response Relationship, Drug, business.industry, Cesarean Section, glucocorticoid, Biochemistry (medical), Settore MED/13 - ENDOCRINOLOGIA, medicine.disease, Pregnancy Complications, business, Adrenal Insufficiency
Abstract

Context Appropriate management of adrenal insufficiency (AI) in pregnancy can be challenging due to the rarity of the disease and lack of evidence-based recommendations to guide glucocorticoid and mineralocorticoid dosage adjustment. Objective Multicenter survey on current clinical approaches in managing AI during pregnancy. Design Retrospective anonymized data collection from 19 international centers from 2013 to 2019. Setting and Patients 128 pregnancies in 113 women with different causes of AI: Addison disease (44%), secondary AI (25%), congenital adrenal hyperplasia (25%), and acquired AI due to bilateral adrenalectomy (6%). Results Hydrocortisone (HC) was the most commonly used glucocorticoid in 83% (97/117) of pregnancies. Glucocorticoid dosage was increased at any time during pregnancy in 73/128 (57%) of cases. In these cases, the difference in the daily dose of HC equivalent between baseline and the third trimester was 8.6 ± 5.4 (range 1-30) mg. Fludrocortisone dosage was increased in fewer cases (7/54 during the first trimester, 9/64 during the second trimester, and 9/62 cases during the third trimester). Overall, an adrenal crisis was reported in 9/128 (7%) pregnancies. Cesarean section was the most frequent mode of delivery at 58% (69/118). Fetal complications were reported in 3/120 (3%) and minor maternal complications in 15/120 (13%) pregnancies without fatal outcomes. Conclusions This survey confirms good maternal and fetal outcome in women with AI managed in specialized endocrine centers. An emphasis on careful endocrine follow-up and repeated patient education is likely to have reduced the risk of adrenal crisis and resulted in positive outcomes.

Published
2020
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38. Characteristics and outcomes of Italian patients from the observational, multicentre, hypopituitary control and complications study (HypoCCS) according to tertiles of growth hormone peak concentration following stimulation testing at study entry

Author

Vincenzo Rochira, Salvatore Cannavò, Nan Jia, Gilberto Mossetto, Diego Ferone, Maria Rosaria Ambrosio, Salvatore Maria Corsello, Antonella Giampietro, Gianluca Aimaretti, Carolina Di Somma, Marco Losa, Maurizio Poggi, Carla Scaroni, Paolo Beck-Peccoz, Losa, Marco, Beck-Peccoz, Paolo, Aimaretti, Gianluca, Di Somma, Carolina, Ambrosio, Maria Rosaria, Ferone, Diego, Giampietro, Antonella, Corsello, Salvatore M., Poggi, Maurizio, Scaroni, Carla, Jia, Nan, Mossetto, Gilberto, Cannavò, Salvatore, and Rochira, Vincenzo

Subjects
Adult, Male, Thyroid Hormones, medicine.medical_specialty, Endocrinology, Diabetes and Metabolism, Hypopituitarism, Group B, NO, Body Mass Index, Endocrinology, Internal medicine, Diabetes mellitus, medicine, Thyroid Hormone, Humans, Multicenter Studies as Topic, Dwarfism, Pituitary, Adverse effect, Framingham Risk Score, Growth Hormone, Acromegaly, Hypopituitarism, Human Growth Hormone, business.industry, Settore MED/13 - ENDOCRINOLOGIA, Female, Growth Hormone, Middle Aged, Observational Studies as Topic, Medicine (all), medicine.disease, hypopituitary, Diabetes and Metabolism, Acromegaly, Observational study, business, Complication, Body mass index, Human
Abstract

Objective To determine whether characteristics and outcomes of Italian patients in the observational global Hypopituitary Control and Complication Study (HypoCCS) differed according to the degree of GH deficiency (GHD). Design Patients were grouped by tertiles of stimulated GH peak concentration at baseline (Group A lowest tertile, n = 342; Group B middle tertile, n = 345; Group C highest tertile, n = 338). Results Baseline demographics, lipid levels, body mass index categories and mean Framingham cardiovascular risk indexes were similar in the three groups and remained substantially unchanged over time, with no subsequent significant between-group differences (except mean levels of triglycerides increased in the highest tertile group). GHD was adult-onset for >75% of patients in all groups. The percentage of patients with multiple pituitary deficiencies was higher in Group A than in the other groups; isolated GHD was reported with highest frequency in Group C. Patients in Group A received the lowest mean starting dose of GH. Hyperlipidaemia at baseline was reported in 35·1%, 31·1% and 24·7% of patients in groups A, B and C, respectively (P = 0·029). Mean duration of GH treatment was 7·21, 5·45 and 4·96 years, respectively. The proportion of patients with adverse events did not differ significantly between groups, with a low prevalence over time of diabetes and cancer. Conclusions In Italian patients from HypoCCS, the level of GH deficit did not influence changes over time in metabolic parameters or adverse event profile, despite differences in GHD severity at baseline and in the starting GH dose.

Published
2015

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