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3. Hypogonadism and sexual function in men affected by adrenocortical carcinoma under mitotane therapy

Author

Canu, Letizia, primary, Sparano, Clotilde, additional, Naletto, Lara, additional, De Filpo, Giuseppina, additional, Cantini, Giulia, additional, Rapizzi, Elena, additional, Martinelli, Serena, additional, Ercolino, Tonino, additional, Cioppi, Francesca, additional, Fantoni, Alessandro, additional, Zanatta, Lorenzo, additional, Terreni, Alessandro, additional, Mannelli, Massimo, additional, Luconi, Michaela, additional, Maggi, Mario, additional, and Lotti, Francesco, additional

Published
2024
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4. Novel Germline PHD2 Variant in a Metastatic Pheochromocytoma and Chronic Myeloid Leukemia, but in the Absence of Polycythemia

Author

Provenzano, Aldesia, primary, Chetta, Massimiliano, additional, De Filpo, Giuseppina, additional, Cantini, Giulia, additional, La Barbera, Andrea, additional, Nesi, Gabriella, additional, Santi, Raffaella, additional, Martinelli, Serena, additional, Rapizzi, Elena, additional, Luconi, Michaela, additional, Maggi, Mario, additional, Mannelli, Massimo, additional, Ercolino, Tonino, additional, and Canu, Letizia, additional

Published
2022
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5. A Multicenter Epidemiological Study on Second Malignancy in Non-Syndromic Pheochromocytoma/Paraganglioma Patients in Italy

Author

Canu, Letizia, primary, Puglisi, Soraya, additional, Berchialla, Paola, additional, De Filpo, Giuseppina, additional, Brignardello, Francesca, additional, Schiavi, Francesca, additional, Ferrara, Alfonso Massimiliano, additional, Zovato, Stefania, additional, Luconi, Michaela, additional, Pia, Anna, additional, Appetecchia, Marialuisa, additional, Arvat, Emanuela, additional, Letizia, Claudio, additional, Maccario, Mauro, additional, Parasiliti-Caprino, Mirko, additional, Altieri, Barbara, additional, Faggiano, Antongiulio, additional, Modica, Roberta, additional, Morelli, Valentina, additional, Arosio, Maura, additional, Verga, Uberta, additional, Pellegrino, Micaela, additional, Petramala, Luigi, additional, Concistrè, Antonio, additional, Razzore, Paola, additional, Ercolino, Tonino, additional, Rapizzi, Elena, additional, Maggi, Mario, additional, Stigliano, Antonio, additional, Burrello, Jacopo, additional, Terzolo, Massimo, additional, Opocher, Giuseppe, additional, Mannelli, Massimo, additional, and Reimondo, Giuseppe, additional

Published
2021
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6. Stimulated Expression of CXCL12 in Adrenocortical Carcinoma by the PPARgamma Ligand Rosiglitazone Impairs Cancer Progression

Author

Cantini, Giulia, primary, Fei, Laura, additional, Canu, Letizia, additional, Lazzeri, Elena, additional, Sottili, Mariangela, additional, Francalanci, Michela, additional, Angelotti, Maria Lucia, additional, De Filpo, Giuseppina, additional, Ercolino, Tonino, additional, Gelmini, Stefania, additional, Mangoni, Monica, additional, Nesi, Gabriella, additional, Hantel, Constanze, additional, Mannelli, Massimo, additional, Maggi, Mario, additional, and Luconi, Michaela, additional

Published
2021
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9. Prognostic and Monitoring Value of Circulating Tumor Cells in Adrenocortical Carcinoma: A Preliminary Monocentric Study

Author

Cantini, Giulia, primary, Canu, Letizia, additional, Armignacco, Roberta, additional, Salvianti, Francesca, additional, De Filpo, Giuseppina, additional, Ercolino, Tonino, additional, Nesi, Gabriella, additional, Maggi, Mario, additional, Mannelli, Massimo, additional, Pinzani, Pamela, additional, and Luconi, Michaela, additional

Published
2020
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10. SDHx and Non-Chromaffin Tumors: A Mediastinal Germ Cell Tumor Occurring in a Young Man with Germline SDHB Mutation

Author

De Filpo, Giuseppina, primary, Cilotti, Antonio, additional, Rolli, Luigi, additional, Pastorino, Ugo, additional, Sonzogni, Angelica, additional, Pradella, Silvia, additional, Cantini, Giulia, additional, Ercolino, Tonino, additional, Nesi, Gabriella, additional, Mannelli, Massimo, additional, Maggi, Mario, additional, and Canu, Letizia, additional

Published
2020
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11. Germline Mutation in KIF1Bβ Gene Associated with Loss of Heterozygosity: Usefulness of Next-Generation Sequencing in the Genetic Screening of Patients with Pheochromocytoma

Author

De Filpo, Giuseppina, primary, Contini, Elisa, additional, Serio, Viola, additional, Valeri, Andrea, additional, Chetta, Massimiliano, additional, Guasti, Daniele, additional, Bani, Daniele, additional, Mannelli, Massimo, additional, Rapizzi, Elena, additional, Luconi, Michaela, additional, Maggi, Mario, additional, Ercolino, Tonino, additional, and Canu, Letizia, additional

Published
2020
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12. Mitotane Concentrations Influence the Risk of Recurrence in Adrenocortical Carcinoma Patients on Adjuvant Treatment

Author

Puglisi, Soraya, primary, Calabrese, Anna, additional, Basile, Vittoria, additional, Ceccato, Filippo, additional, Scaroni, Carla, additional, Simeoli, Chiara, additional, Torlontano, Massimo, additional, Cannavò, Salvatore, additional, Arnaldi, Giorgio, additional, Stigliano, Antonio, additional, Malandrino, Pasqualino, additional, Saba, Laura, additional, Altieri, Barbara, additional, Della Casa, Silvia, additional, Perotti, Paola, additional, Berchialla, Paola, additional, De Filpo, Giuseppina, additional, Canu, Letizia, additional, Loli, Paola, additional, Reimondo, Giuseppe, additional, and Terzolo, Massimo, additional

Published
2019
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15. A multicenter epidemiological study on second malignancy in non-syndromic pheochromocytoma/paraganglioma patients in Italy

Author

Luigi Petramala, Tonino Ercolino, Valentina Morelli, Marialuisa Appetecchia, Antongiulio Faggiano, Francesca Schiavi, P. Razzore, Uberta Verga, Anna Pia, Barbara Altieri, Francesca Brignardello, Massimo Terzolo, Antonio Concistrè, Antonio Stigliano, Soraya Puglisi, Micaela Pellegrino, Mario Maggi, Michaela Luconi, Giuseppe Opocher, Paola Berchialla, Massimo Mannelli, Emanuela Arvat, Mauro Maccario, Mirko Parasiliti-Caprino, Giuseppina De Filpo, Jacopo Burrello, Roberta Modica, Letizia Canu, Stefania Zovato, Alfonso Massimiliano Ferrara, Maura Arosio, Giuseppe Reimondo, Claudio Letizia, Elena Rapizzi, Canu, Letizia, Puglisi, Soraya, Berchialla, Paola, DE FILPO, Giuseppina, Brignardello, Francesca, Schiavi, Francesca, Ferrara, ALFONSO MASSIMILIANO, Zovato, Stefania, Luconi, Michaela, Anna, Pia, Appetecchia, Marialuisa, Arvat, Emanuela, Letizia, Claudio, Maccario, Mauro, Parasiliti-Caprino, Mirko, Altieri, Barbara, Faggiano, Antongiulio, Modica, Roberta, Morelli, Valentina, Arosio, Maura, Verga, Uberta, Pellegrino, Micaela, Petramala, Luigi, Concistrè, Antonio, Razzore, Paola, Ercolino, Tonino, Rapizzi, Elena, Maggi, Mario, Stigliano, Antonio, Burrello, Jacopo, Terzolo, Massimo, Opocher, Giuseppe, Mannelli, Massimo, and Matteo REIMONDO, Giuseppe

Subjects
Oncology, Cancer Research, medicine.medical_specialty, Population, genetic analysis, Malignancy, Mortality surveillance, Article, Pheochromocytoma, paraganglioma, Breast cancer, Paraganglioma, Internal medicine, medicine, ddc:610, education, Thyroid cancer, RC254-282, epidemiology, mortality surveillance, pheochromocytoma, education.field_of_study, business.industry, Incidence (epidemiology), Neoplasms. Tumors. Oncology. Including cancer and carcinogens, medicine.disease, mortality, Standardized mortality ratio, surveillance, business
Abstract

No studies have carried out an extensive analysis of the possible association between non-syndromic pheochromocytomas and paragangliomas (PPGLs) and other malignancies. To assess the risk of additional malignancy in PPGL, we retrospectively evaluated 741 patients with PPGLs followed-up in twelve referral centers in Italy. Incidence of second malignant tumors was compared between this cohort and Italian patients with two subsequent malignancies. Among our patients, 95 (12.8%) developed a second malignant tumor, which were mainly prostate, colorectal and lung/bronchial cancers in males, breast cancer, differentiated thyroid cancer and melanoma in females. The standardized incidence ratio was 9.59 (95% CI 5.46–15.71) in males and 13.21 (95% CI 7.52–21.63) in females. At multivariable analysis, the risk of developing a second malignant tumor increased with age at diagnosis (HR 2.50, 95% CI 1.15–5.44, p = 0.021 for 50–59 vs. &lt, 50-year category, HR 3.46, 95% CI 1.67–7.15, p &lt, 0.001 for &gt, 60- vs. &lt, 50-year). In patients with available genetic evaluation, a positive genetic test was inversely associated with the risk of developing a second tumor (HR 0.25, 95% CI 0.10–0.63, p = 0.003). In conclusion, PPGLs patients have higher incidence of additional malignant tumors compared to the general population who had a first malignancy, which could have an impact on the surveillance strategy.

Published
2021

16. Mitotane Concentrations Influence the Risk of Recurrence in Adrenocortical Carcinoma Patients on Adjuvant Treatment

Author

Anna Calabrese, Paola Loli, Massimo Torlontano, Antonio Stigliano, Pasqualino Malandrino, Barbara Altieri, Paola Berchialla, Giuseppina De Filpo, Silvia Della Casa, Laura Saba, Filippo Ceccato, Soraya Puglisi, Giorgio Arnaldi, Paola Perotti, Letizia Canu, Chiara Simeoli, Carla Scaroni, Massimo Terzolo, Vittoria Basile, Giuseppe Reimondo, Salvatore Cannavò, Puglisi, Soraya, Calabrese, Anna, Basile, Vittoria, Ceccato, Filippo, Scaroni, Carla, Simeoli, Chiara, Torlontano, Massimo, Cannavò, Salvatore, Arnaldi, Giorgio, Stigliano, Antonio, Malandrino, Pasqualino, Saba, Laura, Altieri, Barbara, Della Casa, Silvia, Perotti, Paola, Berchialla, Paola, De Filpo, Giuseppina, Canu, Letizia, Loli, Paola, Reimondo, Giuseppe, and Terzolo, Massimo

Subjects
mitotane, medicine.medical_specialty, Multivariate analysis, recurrence, lcsh:Medicine, 030209 endocrinology & metabolism, Lower risk, Gastroenterology, survival, Article, 03 medical and health sciences, 0302 clinical medicine, Interquartile range, Internal medicine, medicine, adrenocortical carcinoma, Adrenocortical carcinoma, Mitotane, adrenocortical carcinoma, mitotane, prognosis, recurrence, survival, Maintenance dose, business.industry, lcsh:R, Hazard ratio, General Medicine, medicine.disease, 030220 oncology & carcinogenesis, prognosis, business, Body mass index, prognosi, medicine.drug
Abstract

Mitotane is used as a post-operative adjuvant treatment for patients with adrenocortical carcinoma. Monitoring of plasma mitotane concentrations is recommended, but we do not know what impact target concentrations have on patient outcome. To answer this question, we retrospectively analyzed patient records in the Lysosafe Online® database (HRA Pharma, France) for patients who were treated for ≥6 months and who had ≥3 measurements of plasma mitotane levels during follow-ups at 11 tertiary centers in Italy from 2005 to 2017. We identified 110 patients treated with adjuvant mitotane for a median of 46 months (IQR, interquartile range, 28−62) with a median maintenance dose of 2.0 g/day (IQR 1.5−2.5). Achievement of target mitotane concentrations (≥14 mg/L) required a median of 8 months (IQR 5−19). Female sex was associated inversely with the dose, while body mass index (BMI) was correlated positively. Multivariate analysis showed that the Ki67 index and time to achieve the target range of plasma mitotane were independent predictors of recurrence-free survival (RFS). In a separate multivariate model, considering only the maintenance phase (month 7 to month 36, M7−M36) of treatment, the time in the target range of plasma mitotane was associated with a significantly lower risk of recurrence (Hazard Ratio, HR = 0.93; 0.88−0.98, p < 0.01). The prognostic implications of the time in target range and the time needed to reach target mitotane concentrations support the use of mitotane monitoring and may inform practice.

Published
2019

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