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1. Effects of Hospital Early Childcare Intervention in Young Children with Cancer

Author

Zucchetti, G., Ciappina, S., Geuna, T., Nichelli, F., Biondi, A., Camera, F., Ripaldi, M., and Fagioli, F.

Abstract

Purpose: Oncological diseases, especially if experienced in the first 3 years of life, expose young children to conditions of serious physical and emotional stress causing delays in their physical development, cognitive acquisitioning and issues related to their emotional and social functioning. This study examines the effects of the participation in an early childcare intervention to support the children's development, which was proposed to children from 0 to 36 months, at the moment of cancer diagnosis in their hospital setting. Method: A prospective multicenter programme was offered to young children (<3 years) newly diagnosed with cancer. These children were treated in three pediatric oncology centers affiliated to the Italian Association of Pediatric Hematology-Oncology. The children's strengths and weaknesses, in different developmental areas, were examined pre-programme and 6 months post-programme. Results: Fifty-three young children were enrolled in the study. The data showed that at the moment of diagnosis the majority of the children demonstrated low scores across all the abilities in the subscales examined, highlighting a mental age below their chronological age ([delta][subscript T0] = 3.5 months). However, post-programme, the children were shown to have significantly reduced the gap between their chronological and mental age ([delta][subscript T1] = 1 month), demonstrating improvements in all their developmental abilities. Conclusion: The study confirms significant implications for early childcare programmes, delivered in hospitals for young children with cancer, in terms of support for their ongoing physical, cognitive and social development. Therefore, the study demonstrated the necessity, for all pediatric oncology centers, to invest in early childhood education programmes.

Published
2022
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2. Matched unrelated donor transplantation versus haploidentical transplantation with post-transplant cyclophosphamide in children with acute myeloid leukemia: a PDWP-EBMT study

Author

Ruggeri, A, Santoro, N, Galimard, J, Kalwak, K, Algeri, M, Zubarovskaya, L, Czyzewski, K, Skorobogatova, E, Sedlacek, P, Besley, C, Balduzzi, A, Bertrand, Y, Peristeri, J, Fagioli, F, Ifversen, M, Gozdzik, J, Peters, C, Versluijs, B, Biffi, A, Prete, A, Faraci, M, Ghemlas, I, Bodova, I, Aleinikova, O, Dalissier, A, Rocha, V, Corbacioglu, S, Ruggeri A., Santoro N., Galimard J. E., Kalwak K., Algeri M., Zubarovskaya L., Czyzewski K., Skorobogatova E., Sedlacek P., Besley C., Balduzzi A., Bertrand Y., Peristeri J., Fagioli F., Ifversen M., Gozdzik J., Peters C., Versluijs B., Biffi A., Prete A., Faraci M., Ghemlas I., Bodova I., Aleinikova O., Dalissier A., Rocha V., Corbacioglu S., Ruggeri, A, Santoro, N, Galimard, J, Kalwak, K, Algeri, M, Zubarovskaya, L, Czyzewski, K, Skorobogatova, E, Sedlacek, P, Besley, C, Balduzzi, A, Bertrand, Y, Peristeri, J, Fagioli, F, Ifversen, M, Gozdzik, J, Peters, C, Versluijs, B, Biffi, A, Prete, A, Faraci, M, Ghemlas, I, Bodova, I, Aleinikova, O, Dalissier, A, Rocha, V, Corbacioglu, S, Ruggeri A., Santoro N., Galimard J. E., Kalwak K., Algeri M., Zubarovskaya L., Czyzewski K., Skorobogatova E., Sedlacek P., Besley C., Balduzzi A., Bertrand Y., Peristeri J., Fagioli F., Ifversen M., Gozdzik J., Peters C., Versluijs B., Biffi A., Prete A., Faraci M., Ghemlas I., Bodova I., Aleinikova O., Dalissier A., Rocha V., and Corbacioglu S.

Abstract

In children with acute myeloid leukemia (AML) who lack a human leukocyte antigen (HLA) identical sibling, the donor can be replaced with an HLA-matched unrelated donor (MUD) or a haploidentical donor (haplo). We compared outcomes of patients <18 years with AML in first and second complete remission (CR1 & CR2) undergoing a hematopoietic stem cell transplantation (HCT) either with a MUD with anti-thymocyte globulin (ATG) (N=420) or a haplo HCT with post-transplant cyclophosphamide (PT-CY) (N=96) after a myeloablative conditioning regimen (MAC) between 2011 and 2021, reported to the European Society for Blood and Marrow Transplantation. A matched pair analysis was performed to adjust for differences among groups. The final analysis was performed on 253 MUD and 95 haplo-HCT. In the matched cohort, median age at HCT was 11.2 and 10 years and median year of HCT was 2017 and 2018, in MUD and haplo-HCT recipients, respectively. The risk of grade III-IV acute graft-versus-host disease (aGVHD) was significantly higher in the haplo group (hazard ratio [HR]=2.33, 95% confidence interval [CI]: 1.18-4.58; P=0.01). No significant differences were found in 2 years overall survival (OS; 78.4% vs. 71.5%; HR=1.39, 95% CI: 0.84-2.31; P=0.19), leukemia-free survival (LFS; 72.7% vs. 69.5%; HR=1.22, 95% CI: 0.76-1.95; P=0.41), CI of relapse (RI; 19.3% vs. 19.5%; HR=1.14, 95% CI: 0.62-2.08; P=0.68) non-relapse-mortality (NRM; 8% vs. 11%; HR=1.39, 95% CI: 0.66-2.93; P=0.39) and graft-versus-host free relapse-free survival (GRFS; 60.7% vs. 54.5%, HR=1.38, 95% CI: 0.95-2.02; P=0.09) after MUD and haplo-HCT respectively. Our study suggests that haplo-HCT with PT-CY is a suitable option to transplant children with AML lacking a matched related donor.

Published
2024

3. Musculoskeletal manifestations of childhood cancer and differential diagnosis with juvenile idiopathic arthritis (ONCOREUM): a multicentre, cross-sectional study

Author

Amatruda, M, Atzeni, C, Bertolini, P, Bigucci, B, Caniglia, M, Cappella, M, Cattalini, M, Cefalo, MG, Cellini, M, Cortis, E, Davì, S, De Benedetti, F, Di Cataldo, A, Fabbri, E, Fagioli, F, Fontanili, I, Garaventa, A, Gicchino, MF, Ladogana, S, Locatelli, F, Magnolato, A, Marsili, M, Martino, S, Mascarin, M, Messina, C, Micalizzi, C, Porta, F, Rizzari, C, Civino, Adele, Alighieri, Giovanni, Prete, Eleonora, Caroleo, Anna Maria, Magni-Manzoni, Silvia, Vinti, Luciana, Romano, Micol, Santoro, Nicola, Filocamo, Giovanni, Belotti, Tamara, Santarelli, Francesca, Gorio, Chiara, Ricci, Francesca, Colombini, Antonella, Pastore, Serena, Cesaro, Simone, Barone, Patrizia, Verzegnassi, Federico, Olivieri, Alma Nunzia, Ficara, Monica, Miniaci, Angela, Russo, Giovanna, Gallizzi, Romina, Pericoli, Roberta, Breda, Luciana, Mura, Rossella, Podda, Rosa Anna, Onofrillo, Daniela, Lattanzi, Bianca, Tirtei, Elisa, Maggio, Maria Cristina, De Santis, Raffaela, Consolini, Rita, Arlotta, Annalisa, La Torre, Francesco, Mainardi, Chiara, Pelagatti, Maria Antonietta, Coassin, Elisa, Capolsini, Ilaria, Burnelli, Roberta, Tornesello, Assunta, Soscia, Francesca, De Fanti, Alessandro, Rigante, Donato, Pizzato, Cristina, De Fusco, Carmela, Abate, Massimo Eraldo, Roncadori, Andrea, Rossi, Elisa, Stabile, Giulia, Biondi, Andrea, Lepore, Loredana, Conter, Valentino, Rondelli, Roberto, Pession, Andrea, and Ravelli, Angelo

Published
2021
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4. Adolescent gender dysphoria management: position paper from the Italian Academy of Pediatrics, the Italian Society of Pediatrics, the Italian Society for Pediatric Endocrinology and Diabetes, the Italian Society of Adolescent Medicine and the Italian Society of Child and Adolescent Neuropsychiatry

Author

Calcaterra, V, Tornese, G, Zuccotti, G, Staiano, A, Cherubini, V, Gaudino, R, Fazzi, E, Barbi, E, Chiarelli, F, Corsello, G, Esposito, S, Ferrara, P, Iughetti, L, Laforgia, N, Maghnie, M, Marseglia, G, Perilongo, G, Pettoello-Mantovani, M, Ruggieri, M, Russo, G, Salerno, M, Striano, P, Valerio, G, Wasniewska, M, Agosti, M, Agostoni, C, Aiuti, A, Azzari, C, Badolato, R, Balduzzi, A, Baraldi, E, Canani, R, Biffi, A, Biondi, A, Bisogno, G, Pierri, N, Carnielli, V, Cianfarani, S, Cogo, P, Corvaglia, L, Dani, C, Di Salvo, G, Fagioli, F, Fanos, V, Ferrero, G, Francavilla, R, Galli, L, Gazzolo, D, Giaquinto, C, Giordano, P, Gitto, E, Grosso, S, Guarino, A, Indrio, F, Lanari, M, Lionetti, P, Locatelli, F, Lombardo, F, Maffeis, C, Marino, B, Midulla, F, Del Giudice, E, Del Giudice, M, Montini, G, Parenti, G, Parisi, P, Peroni, D, Perrotta, S, Piacentini, G, Pietrobelli, A, Raimondi, F, Ramenghi, U, Ravelli, A, Romano, C, Rossi, F, Rossi, P, Damiano, V, Spalice, A, Suppiej, A, Troncone, R, Verrotti, A, Null, N, Calcaterra, Valeria, Tornese, Gianluca, Zuccotti, Gianvincenzo, Staiano, Annamaria, Cherubini, Valentino, Gaudino, Rossella, Fazzi, Elisa Maria, Barbi, Egidio, Chiarelli, Francesco, Corsello, Giovanni, Esposito, Susanna Maria Roberta, Ferrara, Pietro, Iughetti, Lorenzo, Laforgia, Nicola, Maghnie, Mohamad, Marseglia, Gianluigi, Perilongo, Giorgio, Pettoello-Mantovani, Massimo, Ruggieri, Martino, Russo, Giovanna, Salerno, Mariacarolina, Striano, Pasquale, Valerio, Giuliana, Wasniewska, Malgorzata, Agosti, Massimo, Agostoni, Carlo Virginio, Aiuti, Alessandro, Azzari, Chiara, Badolato, Raffaele, Balduzzi, Adriana, Baraldi, Eugenio, Canani, Roberto Berni, Biffi, Alessandra, Biondi, Andrea, Bisogno, Gianni, Pierri, Nicola Brunetti, Carnielli, Virginio, Cianfarani, Stefano, Cogo, Paola, Corvaglia, Luigi, Dani, Carlo, Di Salvo, Giovanni, Fagioli, Franca, Fanos, Vassilios, Ferrero, Giovanni Battista, Francavilla, Ruggiero, Galli, Luisa, Gazzolo, Diego, Giaquinto, Carlo, Giordano, Paola, Gitto, Eloisa, Grosso, Salvatore, Guarino, Alfredo, Indrio, Flavia, Lanari, Marcello, Lionetti, Paolo, Locatelli, Franco, Lombardo, Fortunato, Maffeis, Claudio, Marino, Bruno, Midulla, Fabio, Del Giudice, Emanuele Miraglia, Del Giudice, Michele Miraglia, Montini, Giovanni, Parenti, Giancarlo, Parisi, Pasquale, Peroni, Diego, Perrotta, Silverio, Piacentini, Giorgio, Pietrobelli, Angelo, Raimondi, Francesco, Ramenghi, Ugo, Ravelli, Angelo, Romano, Claudio, Rossi, Francesca, Rossi, Paolo, Damiano, Vincenzo Salpietro, Spalice, Alberto, Suppiej, Agnese, Troncone, Riccardo, Verrotti, Alberto, null, null, Calcaterra, V, Tornese, G, Zuccotti, G, Staiano, A, Cherubini, V, Gaudino, R, Fazzi, E, Barbi, E, Chiarelli, F, Corsello, G, Esposito, S, Ferrara, P, Iughetti, L, Laforgia, N, Maghnie, M, Marseglia, G, Perilongo, G, Pettoello-Mantovani, M, Ruggieri, M, Russo, G, Salerno, M, Striano, P, Valerio, G, Wasniewska, M, Agosti, M, Agostoni, C, Aiuti, A, Azzari, C, Badolato, R, Balduzzi, A, Baraldi, E, Canani, R, Biffi, A, Biondi, A, Bisogno, G, Pierri, N, Carnielli, V, Cianfarani, S, Cogo, P, Corvaglia, L, Dani, C, Di Salvo, G, Fagioli, F, Fanos, V, Ferrero, G, Francavilla, R, Galli, L, Gazzolo, D, Giaquinto, C, Giordano, P, Gitto, E, Grosso, S, Guarino, A, Indrio, F, Lanari, M, Lionetti, P, Locatelli, F, Lombardo, F, Maffeis, C, Marino, B, Midulla, F, Del Giudice, E, Del Giudice, M, Montini, G, Parenti, G, Parisi, P, Peroni, D, Perrotta, S, Piacentini, G, Pietrobelli, A, Raimondi, F, Ramenghi, U, Ravelli, A, Romano, C, Rossi, F, Rossi, P, Damiano, V, Spalice, A, Suppiej, A, Troncone, R, Verrotti, A, Null, N, Calcaterra, Valeria, Tornese, Gianluca, Zuccotti, Gianvincenzo, Staiano, Annamaria, Cherubini, Valentino, Gaudino, Rossella, Fazzi, Elisa Maria, Barbi, Egidio, Chiarelli, Francesco, Corsello, Giovanni, Esposito, Susanna Maria Roberta, Ferrara, Pietro, Iughetti, Lorenzo, Laforgia, Nicola, Maghnie, Mohamad, Marseglia, Gianluigi, Perilongo, Giorgio, Pettoello-Mantovani, Massimo, Ruggieri, Martino, Russo, Giovanna, Salerno, Mariacarolina, Striano, Pasquale, Valerio, Giuliana, Wasniewska, Malgorzata, Agosti, Massimo, Agostoni, Carlo Virginio, Aiuti, Alessandro, Azzari, Chiara, Badolato, Raffaele, Balduzzi, Adriana, Baraldi, Eugenio, Canani, Roberto Berni, Biffi, Alessandra, Biondi, Andrea, Bisogno, Gianni, Pierri, Nicola Brunetti, Carnielli, Virginio, Cianfarani, Stefano, Cogo, Paola, Corvaglia, Luigi, Dani, Carlo, Di Salvo, Giovanni, Fagioli, Franca, Fanos, Vassilios, Ferrero, Giovanni Battista, Francavilla, Ruggiero, Galli, Luisa, Gazzolo, Diego, Giaquinto, Carlo, Giordano, Paola, Gitto, Eloisa, Grosso, Salvatore, Guarino, Alfredo, Indrio, Flavia, Lanari, Marcello, Lionetti, Paolo, Locatelli, Franco, Lombardo, Fortunato, Maffeis, Claudio, Marino, Bruno, Midulla, Fabio, Del Giudice, Emanuele Miraglia, Del Giudice, Michele Miraglia, Montini, Giovanni, Parenti, Giancarlo, Parisi, Pasquale, Peroni, Diego, Perrotta, Silverio, Piacentini, Giorgio, Pietrobelli, Angelo, Raimondi, Francesco, Ramenghi, Ugo, Ravelli, Angelo, Romano, Claudio, Rossi, Francesca, Rossi, Paolo, Damiano, Vincenzo Salpietro, Spalice, Alberto, Suppiej, Agnese, Troncone, Riccardo, Verrotti, Alberto, and null, null

Abstract

Background In response to the imperative need for standardized support for adolescent Gender Dysphoria (GD), the Italian Academy of Pediatrics, in collaboration with the Italian Society of Pediatrics, the Italian Society for Pediatric Endocrinology and Diabetes, Italian Society of Adolescent Medicine and Italian Society of Child and Adolescent Neuropsychiatry is drafting a position paper. The purpose of this paper is to convey the author's opinion on the topic, offering foundational information on potential aspects of gender-affirming care and emphasizing the care and protection of children and adolescents with GD.Main body Recognizing that adolescents may choose interventions based on their unique needs and goals and understanding that every individual within this group has a distinct trajectory, it is crucial to ensure that each one is welcomed and supported. The approach to managing individuals with GD is a multi-stage process involving a multidisciplinary team throughout all phases. Decisions regarding treatment should be reached collaboratively by healthcare professionals and the family, while considering the unique needs and circumstances of the individual and be guided by scientific evidence rather than biases or ideologies. Politicians and high court judges should address discrimination based on gender identity in legislation and support service development that aligns with the needs of young people. It is essential to establish accredited multidisciplinary centers equipped with the requisite skills and experience to effectively manage adolescents with GD, thereby ensuring the delivery of high-quality care.Conclusion Maintaining an evidence-based approach is essential to safeguard the well-being of transgender and gender diverse adolescents.

Published
2024

5. Long-term complications after allogeneic hematopoietic stem cell transplantation for pediatric patients with acute leukemia or myelodysplastic syndrome given either a Treosulfan- or a Busulfan-based conditioning regimen: results of an AIEOP (Associazione Italiana Ematologia Oncologia Pediatrica) retrospective study

Author

Saglio, F, Pagliara, D, Zecca, M, Balduzzi, A, Cattoni, A, Prete, A, Tambaro, F, Faraci, M, Calore, E, Locatelli, F, Fagioli, F, Saglio, Francesco, Pagliara, Daria, Zecca, Marco, Balduzzi, Adriana, Cattoni, Alessandro, Prete, Arcangelo, Tambaro, Francesco Paolo, Faraci, Maura, Calore, Elisabetta, Locatelli, Franco, Fagioli, Franca, Saglio, F, Pagliara, D, Zecca, M, Balduzzi, A, Cattoni, A, Prete, A, Tambaro, F, Faraci, M, Calore, E, Locatelli, F, Fagioli, F, Saglio, Francesco, Pagliara, Daria, Zecca, Marco, Balduzzi, Adriana, Cattoni, Alessandro, Prete, Arcangelo, Tambaro, Francesco Paolo, Faraci, Maura, Calore, Elisabetta, Locatelli, Franco, and Fagioli, Franca

Abstract

Patients undergoing hematopoietic stem cell transplantation (HSCT) for hematologic malignancies during childhood have an increased risk of developing long-term sequelae that are in part attributable to the conditioning regimen. The present study aimed to assess the occurrence of long-term toxicities in a population of children who underwent HSCT for hematologic malignancies using either treosulfan or busulfan in the conditioning regimen. The cumulative incidences of growth impairment, altered gonadal function, altered thyroid function, cataracts, secondary malignant neoplasia, and altered pulmonary function were evaluated retrospectively by univariable and multivariable analyses in a population of 521 pediatric patients with acute leukemias or myelodysplastic syndromes treated in 20 Italian transplant centers affiliated with the Associazione Italiana Ematologia ed Oncologia Pediatrica (AIEOP). The median duration of follow-up for the entire study population was 7.1 years (range, 1 to 16 years). Overall, a larger proportion of patients given busulfan developed long-term toxicities compared to patients treated with treosulfan (34% versus 20%; P = .01). In univariable analysis, gonadal toxicity developed in 10% of patients who received treosulfan (95% confidence interval [CI], 3% to 15%), compared with 38% (95% CI, 24% to 39%) of busulfan-treated patients (P = .02), and this finding was confirmed by multivariable analysis (relative risk, .51; 95% CI, .34 to .76; P = .0009). We did not find any statistically significant associations between the occurrence of other long-term toxicities and the use of either busulfan or treosulfan. This study provides evidence that the use of treosulfan is correlated with a reduced incidence of gonadal toxicity in children undergoing HSCT for hematologic malignancies.

Published
2024

10. Bone sarcomas: ESMO–PaedCan–EURACAN Clinical Practice Guidelines for diagnosis, treatment and follow-up

Author

Casali, P.G., Bielack, S., Abecassis, N., Aro, H.T., Bauer, S., Biagini, R., Bonvalot, S., Boukovinas, I., Bovee, J V M G, Brennan, B., Brodowicz, T., Broto, J.M., Brugières, L., Buonadonna, A., De Álava, E., Dei Tos, A.P., Del Muro, X.G., Dileo, P., Dhooge, C., Eriksson, M., Fagioli, F., Fedenko, A., Ferraresi, V., Ferrari, A., Ferrari, S., Frezza, A.M., Gaspar, N., Gasperoni, S., Gelderblom, H., Gil, T., Grignani, G., Gronchi, A., Haas, R.L., Hassan, B., Hecker-Nolting, S., Hohenberger, P., Issels, R., Joensuu, H., Jones, R.L., Judson, I., Jutte, P., Kaal, S., Kager, L., Kasper, B., Kopeckova, K., Krákorová, D.A., Ladenstein, R., Le Cesne, A., Lugowska, I., Merimsky, O., Montemurro, M., Morland, B., Pantaleo, M.A., Piana, R., Picci, P., Piperno-Neumann, S., Pousa, A.L., Reichardt, P., Robinson, M.H., Rutkowski, P., Safwat, A.A., Schöffski, P., Sleijfer, S., Stacchiotti, S., Strauss, S.J., Sundby Hall, K., Unk, M., Van Coevorden, F., van der Graaf, W.T.A., Whelan, J., Wardelmann, E., Zaikova, O., and Blay, J.Y.

Published
2018
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11. Bone sarcomas: ESMO–EURACAN–GENTURIS–ERN PaedCan Clinical Practice Guideline for diagnosis, treatment and follow-up

Author

Strauss, S.J., Frezza, A.M., Abecassis, N., Bajpai, J., Bauer, S., Biagini, R., Bielack, S., Blay, J.Y., Bolle, S., Bonvalot, S., Boukovinas, I., Bovee, J.V.M.G., Boye, K., Brennan, B., Brodowicz, T., Buonadonna, A., Alava, E. de, Tos, A.P. dei, Muro, X.G. del, Dufresne, A., Eriksson, M., Fagioli, F., Fedenko, A., Ferraresi, V., Ferrari, A., Gaspar, N., Gasperoni, S., Gelderblom, H., Gouin, F., Grignani, G., Gronchi, A., Haas, R., Hassan, A.B., Hecker-Nolting, S., Hindi, N., Hohenberger, P., Joensuu, H., Jones, R.L., Jungels, C., Jutte, P., Kager, L., Kasper, B., Kawai, A., Kopeckova, K., Krakorova, D.A., Cesne, A. le, Grange, F. le, Legius, E., Leithner, A., Pousa, A.L., Martin-Broto, J., Merimsky, O., Messiou, C., Miah, A.B., Mir, O., Montemurro, M., Morland, B., Morosi, C., Palmerini, E., Pantaleo, M.A., Piana, R., Piperno-Neumann, S., Reichardt, P., Rutkowski, P., Safwat, A.A., Sangalli, C., Sbaraglia, M., Scheipl, S., Schoffski, P., Sleijfer, S., Strauss, D., Hall, K.S., Trama, A., Unk, M., Sande, M.A.J. van de, Graaf, W.T.A. van der, Houdt, W.J. van, Frebourg, T., Ladenstein, R., Casali, P.G., Stacchiotti, S., ESMO Guidelines Comm, EURACAN, GENTURIS, ERN PaedCan, European Society for Medical Oncology, Strauss S.J., Frezza A.M., Abecassis N., Bajpai J., Bauer S., Biagini R., Bielack S., Blay J.Y., Bolle S., Bonvalot S., Boukovinas I., Bovee J.V.M.G., Boye K., Brennan B., Brodowicz T., Buonadonna A., de Alava E., Dei Tos A.P., Garcia del Muro X., Dufresne A., Eriksson M., Fagioli F., Fedenko A., Ferraresi V., Ferrari A., Gaspar N., Gasperoni S., Gelderblom H., Gouin F., Grignani G., Gronchi A., Haas R., Hassan A.B., Hecker-Nolting S., Hindi N., Hohenberger P., Joensuu H., Jones R.L., Jungels C., Jutte P., Kager L., Kasper B., Kawai A., Kopeckova K., Krakorova D.A., Le Cesne A., Le Grange F., Legius E., Leithner A., Lopez Pousa A., Martin-Broto J., Merimsky O., Messiou C., Miah A.B., Mir O., Montemurro M., Morland B., Morosi C., Palmerini E., Pantaleo M.A., Piana R., Piperno-Neumann S., Reichardt P., Rutkowski P., Safwat A.A., Sangalli C., Sbaraglia M., Scheipl S., Schoffski P., Sleijfer S., Strauss D., Sundby Hall K., Trama A., Unk M., van de Sande M.A.J., van der Graaf W.T.A., van Houdt W.J., Frebourg T., Ladenstein R., Casali P.G., Stacchiotti S., ​Basic and Translational Research and Imaging Methodology Development in Groningen (BRIDGE), Public Health Research (PHR), and Man, Biomaterials and Microbes (MBM)

Subjects
medicine.medical_specialty, diagnosis, Medizin, bone sarcoma, Bone Neoplasm, Bone Sarcoma, Follow-Up Studie, 03 medical and health sciences, 0302 clinical medicine, follow-up, medicine, 030304 developmental biology, Osteosarcoma, 0303 health sciences, treatment, business.industry, Sarcoma, Hematology, Guideline, clinical practice guideline, management, 3. Good health, Clinical Practice, diagnosi, Oncology, Diagnosis treatment, 030220 oncology & carcinogenesis, Radiology, business, Human
Abstract

A. Kawai43, K. Kopeckova44, D. A. Krakorova45, A. Le Cesne46, F. Le Grange1, E. Legius47, A. Leithner48, A. Lopez Pousa49, J. Martin-Broto36, O. Merimsky50, C. Messiou51, A. B. Miah52, O. Mir53, M. Montemurro54, B. Morland55, C. Morosi56, E. Palmerini57, M. A. Pantaleo58, R. Piana59, S. Piperno-Neumann60, P. Reichardt61, P. Rutkowski62, A. A. Safwat63, C. Sangalli64, M. Sbaraglia19, S. Scheipl48, P. Schoffski65, S. Sleijfer66, D. Strauss67, K. Sundby Hall13, A. Trama68, M. Unk69, M. A. J. van de Sande70, W. T. A. van der Graaf66,71, W. J. van Houdt72, T. Frebourg73x, R. Ladenstein41z, P. G. Casali2,74z &

Published
2021

13. Analysis of a direct access testing system for the detection of SARS-CoV-2 in the paediatric population attending school.

Author

Bert, F., Lo Moro, G., Barbaro, S., Barbero, S., Boietti, E., Minutiello, E., Sinigaglia, T., Fagioli, F., and Siliquini, R.

Subjects
COVID-19 pandemic, PEDIATRICS, DAY hospitals, MEDICAL emergencies, PUBLIC health
Abstract

Copyright of Annali di Igiene, Medicina Preventiva e di Comunità is the property of Societa Editrice Universo s.r.l. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use. This abstract may be abridged. No warranty is given about the accuracy of the copy. Users should refer to the original published version of the material for the full abstract. (Copyright applies to all Abstracts.)

Published
2023
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15. GAU-PED study for early diagnosis of Gaucher disease in children with splenomegaly and cytopenia

Author

Pession, A., Di Rocco, M., Venturelli, F., Tappino, B., Morello, W., Santoro, N., Giordano, P., Filippini, B., Rinieri, S., Russo, G., Girardi, K., Ruggiero, A., Galea, E., Antonucci, R., Tovaglieri, N., Porta, F., Tartaglione, I., Giona, F., Fagioli, F., Burlina, A., Mura, R., Russo, B., Tornesello, A., Menna, G., Russo, D., Caniglia, M., Schettini, S., Onofrillo, D., Ladogana, S., and Civino, A.

Subjects
Cytopenia, Splenomegaly, Lysosomal storage disease, Gaucher disease, Thrombocytopenia
Published
2023

18. PHASE 2 KEYNOTE-667 STUDY OF PEMBROLIZUMAB (PEMBRO) IN CHILDREN AND YOUNG ADULTS WITH NEWLY DIAGNOSED CLASSICAL HODGKIN LYMPHOMA (Chl) AND SLOW EARLY RESPONSE (SER) TO FRONTLINE CHEMOTHERAPY (CHEMO)

Author

Vinti, L., primary, Daw, Stephen, additional, Alvarez, C., additional, Fagioli, F., additional, Beishuizen, A., additional, Michel, G., additional, Moleti, M., additional, Cepelova, M., additional, Thorwarth, A., additional, Rigaud, C., additional, de Sabando, D., additional, Parker, J., additional, Zhu, Y., additional, Pillai, P., additional, Nahar, A., additional, and Mauz-Koerholz, C., additional

Published
2022
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21. Pregnancy and pregnancy outcomes after hematopoietic stem cell transplantation in childhood: A cross-sectional survey of the EBMT Pediatric Diseases Working Party

Author

Diesch-Furlanetto, T, Rovo, A, Galimard, J, Szinnai, G, Dalissier, A, Sedlacek, P, Bodova, I, Roussou, V, Gibson, B, Poire, X, Fagioli, F, Pichler, H, Faraci, M, Gumy-Pause, F, Dalle, J, Balduzzi, A, Bader, P, Corbacioglu, S, Diesch-Furlanetto T., Rovo A., Galimard J. E., Szinnai G., Dalissier A., Sedlacek P., Bodova I., Roussou V. K., Gibson B. E., Poire X., Fagioli F., Pichler H., Faraci M., Gumy-Pause F. G., Dalle J. H., Balduzzi A., Bader P., Corbacioglu S., Diesch-Furlanetto, T, Rovo, A, Galimard, J, Szinnai, G, Dalissier, A, Sedlacek, P, Bodova, I, Roussou, V, Gibson, B, Poire, X, Fagioli, F, Pichler, H, Faraci, M, Gumy-Pause, F, Dalle, J, Balduzzi, A, Bader, P, Corbacioglu, S, Diesch-Furlanetto T., Rovo A., Galimard J. E., Szinnai G., Dalissier A., Sedlacek P., Bodova I., Roussou V. K., Gibson B. E., Poire X., Fagioli F., Pichler H., Faraci M., Gumy-Pause F. G., Dalle J. H., Balduzzi A., Bader P., and Corbacioglu S.

Abstract

STUDY QUESTION: What are the characteristics of patients with conceptions transplanted in childhood and adolescence? SUMMARY ANSWER: Insemination and conception after hematopoietic stem cell transplantation (HCT) in childhood or adolescence was possible, even after myeloablative conditioning regimes, although some patients required reproductive medicine support. WHAT IS KNOWN ALREADY: Preparative regimens of HCT are highly gonadotoxic, which leads to gonadal failure and pubertal development disorders. There are few population-based studies assessing the risk of future infertility in children after HCT. STUDY DESIGN, SIZE, DURATION: We conducted a retrospective study to investigate natural or assisted conceptions and their outcomes in patients <18 years old before their first transplantation who received HCT between 1995 and 2016 and were in the European Society for Blood and Marrow Transplantation (EBMT) registry. Adoptions were excluded from the analysis. PARTICIPANTS/MATERIALS, SETTING, METHODS: Detailed information concerning pregnancy occurrences and outcomes were obtained by a separate questionnaire. Quantitative variables were presented as medians with their interquartile range (IQR) or range, and categorical variables were presented as frequencies and percentages. MAIN RESULTS AND THE ROLE OF CHANCE: In total, 62 988 pediatric patients received a first HCT in EBMT centers between 1995 and 2016. Pregnancy was reported in 406 patients in the database. The median age at transplantation was 15.7 (range: 0.7–18) years, and the median age at declared conception was 25.0 (range: 16.3–38.8) years. Details concerning the first pregnancy and pregnancy outcome were obtained from 99 patients (24%) from the returned questionnaires. The median age at delivery or pregnancy interruption of the females was 23.0 (IQR: 20.8–27) years, with a median time after transplant of 10.7 (IQR: 6.6–15.4) years. Compared with the mean age of healthy women at their first child’s birth (2

Published
2021

22. CD56, HLA-DR, and CD45 recognize a subtype of childhood AML harboring CBFA2T3-GLIS2 fusion transcript

Author

Zangrando, A, Cavagnero, F, Scarparo, P, Varotto, E, Francescato, S, Tregnago, C, Cuccurullo, R, Fagioli, F, Nigro, L, Masetti, R, Putti, M, Rizzari, C, Santoro, N, Pession, A, Pigazzi, M, Locatelli, F, Basso, G, Buldini, B, Zangrando A., Cavagnero F., Scarparo P., Varotto E., Francescato S., Tregnago C., Cuccurullo R., Fagioli F., Nigro L. L., Masetti R., Putti M. C., Rizzari C., Santoro N., Pession A., Pigazzi M., Locatelli F., Basso G., Buldini B., Zangrando, A, Cavagnero, F, Scarparo, P, Varotto, E, Francescato, S, Tregnago, C, Cuccurullo, R, Fagioli, F, Nigro, L, Masetti, R, Putti, M, Rizzari, C, Santoro, N, Pession, A, Pigazzi, M, Locatelli, F, Basso, G, Buldini, B, Zangrando A., Cavagnero F., Scarparo P., Varotto E., Francescato S., Tregnago C., Cuccurullo R., Fagioli F., Nigro L. L., Masetti R., Putti M. C., Rizzari C., Santoro N., Pession A., Pigazzi M., Locatelli F., Basso G., and Buldini B.

Abstract

The presence of CBFA2T3-GLIS2 fusion gene has been identified in childhood Acute Myeloid Leukemia (AML). In view of the genomic studies indicating a distinct gene expression profile, we evaluated the role of immunophenotyping in characterizing a rare subtype of AML-CBFA2T3-GLIS2 rearranged. Immunophenotypic data were obtained by studying a cohort of 20 pediatric CBFA2T3-GLIS2-AML and 77 AML patients not carrying the fusion transcript. Enrolled cases were included in the Associazione Italiana di Ematologia Oncologia Pediatrica (AIEOP) AML trials and immunophenotypes were compared using different statistical approaches. By multiple computational procedures, we identified two main core antigens responsible for the identification of the CBFA2T3-GLIS2-AML. CD56 showed the highest performance in single marker evaluation (AUC = 0.89) and granted the most accurate prediction when used in combination with HLA-DR (AUC = 0.97) displaying a 93% sensitivity and 99% specificity. We also observed a weak-to-negative CD45 expression, being exceptional in AML. We here provide evidence that the combination of HLA-DR negativity and intense bright CD56 expression detects a rare and aggressive pediatric AML genetic lesion improving the diagnosis performance.

Published
2021

23. Genetic and epigenetic characterization of a discordant kmt2a/aff1-rearranged infant monozygotic twin pair

Author

Russo, A, Viberti, C, Mareschi, K, Casalone, E, Guarrera, S, Birolo, G, Cazzaniga, G, Corral, L, Trentin, L, Basso, G, Fagioli, F, Matullo, G, Russo A., Viberti C., Mareschi K., Casalone E., Guarrera S., Birolo G., Cazzaniga G., Corral L., Trentin L., Basso G., Fagioli F., Matullo G., Russo, A, Viberti, C, Mareschi, K, Casalone, E, Guarrera, S, Birolo, G, Cazzaniga, G, Corral, L, Trentin, L, Basso, G, Fagioli, F, Matullo, G, Russo A., Viberti C., Mareschi K., Casalone E., Guarrera S., Birolo G., Cazzaniga G., Corral L., Trentin L., Basso G., Fagioli F., and Matullo G.

Abstract

The KMT2A/AFF1 rearrangement is associated with an unfavorable prognosis in infant acute lymphocytic leukemia (ALL). Discordant ALL in monozygotic twins is uncommon and represents an attractive resource to evaluate intrauterine environment–genetic interplay in ALL. Mutational and epigenetic profiles were characterized for a discordant KMT2A/AFF1-rearranged infant monozy-gotic twin pair and their parents, and they were compared to three independent KMT2A/AFF1-positive ALL infants, in which the DNA methylation and gene expression profiles were investigated. A de novo Q61H NRAS mutation was detected in the affected twin at diagnosis and backtracked in both twins at birth. The KMT2A/AFF1 rearrangement was absent at birth in both twins. Genetic analyses conducted at birth gave more insights into the timing of the mutation hit. We identified correlations between DNA methylation and gene expression changes for 32 genes in the three independent affected versus remitted patients. The strongest correlations were observed for the RAB32, PDK4, CXCL3, RANBP17, and MACROD2 genes. This epigenetic signature could be a putative target for the development of novel epigenetic-based therapies and could help in explaining the molecular mechanisms characterizing ALL infants with KMT2A/AFF1 fusions.

Published
2021

24. Musculoskeletal manifestations of childhood cancer and differential diagnosis with juvenile idiopathic arthritis (ONCOREUM): a multicentre, cross-sectional study

Author

Civino, A, Alighieri, G, Prete, E, Caroleo, A, Magni-Manzoni, S, Vinti, L, Romano, M, Santoro, N, Filocamo, G, Belotti, T, Santarelli, F, Gorio, C, Ricci, F, Colombini, A, Pastore, S, Cesaro, S, Barone, P, Verzegnassi, F, Olivieri, A, Ficara, M, Miniaci, A, Russo, G, Gallizzi, R, Pericoli, R, Breda, L, Mura, R, Podda, R, Onofrillo, D, Lattanzi, B, Tirtei, E, Maggio, M, De Santis, R, Consolini, R, Arlotta, A, La Torre, F, Mainardi, C, Pelagatti, M, Coassin, E, Capolsini, I, Burnelli, R, Tornesello, A, Soscia, F, De Fanti, A, Rigante, D, Pizzato, C, De Fusco, C, Abate, M, Roncadori, A, Rossi, E, Stabile, G, Biondi, A, Lepore, L, Conter, V, Rondelli, R, Pession, A, Ravelli, A, Amatruda, M, Atzeni, C, Bertolini, P, Bigucci, B, Caniglia, M, Cappella, M, Cattalini, M, Cefalo, M, Cellini, M, Cortis, E, Davi, S, De Benedetti, F, Di Cataldo, A, Fabbri, E, Fagioli, F, Fontanili, I, Garaventa, A, Gicchino, M, Ladogana, S, Locatelli, F, Magnolato, A, Marsili, M, Martino, S, Mascarin, M, Messina, C, Micalizzi, C, Porta, F, Rizzari, C, Civino A., Alighieri G., Prete E., Caroleo A. M., Magni-Manzoni S., Vinti L., Romano M., Santoro N., Filocamo G., Belotti T., Santarelli F., Gorio C., Ricci F., Colombini A., Pastore S., Cesaro S., Barone P., Verzegnassi F., Olivieri A. N., Ficara M., Miniaci A., Russo G., Gallizzi R., Pericoli R., Breda L., Mura R., Podda R. A., Onofrillo D., Lattanzi B., Tirtei E., Maggio M. C., De Santis R., Consolini R., Arlotta A., La Torre F., Mainardi C., Pelagatti M. A., Coassin E., Capolsini I., Burnelli R., Tornesello A., Soscia F., De Fanti A., Rigante D., Pizzato C., De Fusco C., Abate M. E., Roncadori A., Rossi E., Stabile G., Biondi A., Lepore L., Conter V., Rondelli R., Pession A., Ravelli A., Amatruda M., Atzeni C., Bertolini P., Bigucci B., Caniglia M., Cappella M., Cattalini M., Cefalo M. G., Cellini M., Cortis E., Davi S., De Benedetti F., Di Cataldo A., Fabbri E., Fagioli F., Fontanili I., Garaventa A., Gicchino M. F., Ladogana S., Locatelli F., Magnolato A., Marsili M., Martino S., Mascarin M., Messina C., Micalizzi C., Porta F., Rizzari C., Civino, A, Alighieri, G, Prete, E, Caroleo, A, Magni-Manzoni, S, Vinti, L, Romano, M, Santoro, N, Filocamo, G, Belotti, T, Santarelli, F, Gorio, C, Ricci, F, Colombini, A, Pastore, S, Cesaro, S, Barone, P, Verzegnassi, F, Olivieri, A, Ficara, M, Miniaci, A, Russo, G, Gallizzi, R, Pericoli, R, Breda, L, Mura, R, Podda, R, Onofrillo, D, Lattanzi, B, Tirtei, E, Maggio, M, De Santis, R, Consolini, R, Arlotta, A, La Torre, F, Mainardi, C, Pelagatti, M, Coassin, E, Capolsini, I, Burnelli, R, Tornesello, A, Soscia, F, De Fanti, A, Rigante, D, Pizzato, C, De Fusco, C, Abate, M, Roncadori, A, Rossi, E, Stabile, G, Biondi, A, Lepore, L, Conter, V, Rondelli, R, Pession, A, Ravelli, A, Amatruda, M, Atzeni, C, Bertolini, P, Bigucci, B, Caniglia, M, Cappella, M, Cattalini, M, Cefalo, M, Cellini, M, Cortis, E, Davi, S, De Benedetti, F, Di Cataldo, A, Fabbri, E, Fagioli, F, Fontanili, I, Garaventa, A, Gicchino, M, Ladogana, S, Locatelli, F, Magnolato, A, Marsili, M, Martino, S, Mascarin, M, Messina, C, Micalizzi, C, Porta, F, Rizzari, C, Civino A., Alighieri G., Prete E., Caroleo A. M., Magni-Manzoni S., Vinti L., Romano M., Santoro N., Filocamo G., Belotti T., Santarelli F., Gorio C., Ricci F., Colombini A., Pastore S., Cesaro S., Barone P., Verzegnassi F., Olivieri A. N., Ficara M., Miniaci A., Russo G., Gallizzi R., Pericoli R., Breda L., Mura R., Podda R. A., Onofrillo D., Lattanzi B., Tirtei E., Maggio M. C., De Santis R., Consolini R., Arlotta A., La Torre F., Mainardi C., Pelagatti M. A., Coassin E., Capolsini I., Burnelli R., Tornesello A., Soscia F., De Fanti A., Rigante D., Pizzato C., De Fusco C., Abate M. E., Roncadori A., Rossi E., Stabile G., Biondi A., Lepore L., Conter V., Rondelli R., Pession A., Ravelli A., Amatruda M., Atzeni C., Bertolini P., Bigucci B., Caniglia M., Cappella M., Cattalini M., Cefalo M. G., Cellini M., Cortis E., Davi S., De Benedetti F., Di Cataldo A., Fabbri E., Fagioli F., Fontanili I., Garaventa A., Gicchino M. F., Ladogana S., Locatelli F., Magnolato A., Marsili M., Martino S., Mascarin M., Messina C., Micalizzi C., Porta F., and Rizzari C.

Abstract

Background: Presenting symptoms of childhood cancers might mimic those of rheumatic diseases. However, the evidence available to guide differential diagnosis remains scarce. Preventing wrong or delayed diagnosis is therefore important to avoid incorrect administration of glucocorticoid or immunosuppressive therapy and worsening of prognosis. As such, we aimed to assess the prevalence and characteristics of presenting musculoskeletal manifestations in patients at cancer onset and to identify the factors that differentiate childhood malignancies with arthropathy from juvenile idiopathic arthritis. Methods: We did a multicentre, cross-sectional study at 25 paediatric haemato-oncology centres and 22 paediatric rheumatology centres in Italy. We prospectively recruited patients who were younger than 16 years that were newly diagnosed with cancer or juvenile idiopathic arthritis. We excluded patients with glucocorticoid pre-treatment (>1 mg/kg per day of oral prednisone or equivalent for ≥2 consecutive weeks). We collected data for patients with a new diagnosis of cancer or juvenile idiopathic arthritis using an electronic case report form on a web-based platform powered by the Cineca Interuniversity Consortium. The primary outcome was to describe the frequency and characteristics of musculoskeletal manifestations at cancer onset; and the secondary outcome was to identify factors that could discriminate malignancies presenting with arthropathy, with or without other musculoskeletal symptoms, from juvenile idiopathic arthritis using multivariable logistic regression analysis. Findings: Between May 1, 2015, and May 31, 2018, 1957 patients were eligible, of which 1277 (65%) had cancer and 680 (35%) had juvenile idiopathic arthritis. Musculoskeletal symptoms occurred in 324 (25% [95% CI 23·0–27·8]) of 1277 patients with cancer, of whom 207 had arthropathy. Patients with malignant bone tumours had the highest frequency of musculoskeletal symptoms (53 [80%] of 66), followed b

Published
2021

25. Changes in Stem Cell Transplant activity and procedures during SARS-CoV2 pandemic in Italy: an Italian Bone Marrow Transplant Group (GITMO) nationwide analysis (TransCOVID-19 Survey)

Author

Russo, D, Polverelli, N, Malagola, M, Farina, M, Leoni, A, Bernardi, S, Mammoliti, S, Sacchi, N, Martino, M, Ciceri, F, Zallio, F, Olivieri, A, Falcioni, S, Storti, G, Michieli, M, Carluccio, P, Grassi, A, Oldani, E, Bonifazi, F, Prete, A, Cavattoni, I, Maffeis, M, Pastore, D, Vacca, A, Caravelli, D, Mirabile, M, Mordini, N, Nozzoli, C, Faraci, M, Federico, V, Ronconi, S, Skert, C, Onida, F, Marcatti, M, Piemontese, S, Narni, F, Balduzzi, A, De Simone, G, Picardi, A, De Gobbi, M, Calore, E, Tringali, S, Zecca, M, Secondino, S, Guiducci, B, Pelosini, M, Zuffa, E, Facchini, L, Imola, M, Iori, A, Proia, A, Sica, S, Armiento, D, Carella, A, Dellacasa, C, Fagioli, F, Rabusin, M, Ferrario, A, Elice, F, Russo D., Polverelli N., Malagola M., Farina M., Leoni A., Bernardi S., Mammoliti S., Sacchi N., Martino M., Ciceri F., Zallio F., Olivieri A., Falcioni S., Storti G., Michieli M., Carluccio P., Grassi A., Oldani E., Bonifazi F., Prete A., Cavattoni I. M., Maffeis M., Pastore D., Vacca A., Caravelli D., Mirabile M., Mordini N., Nozzoli C., Faraci M., Federico V., Ronconi S., Skert C., Onida F., Marcatti M., Piemontese S., Narni F., Balduzzi A., De Simone G., Picardi A., De Gobbi M., Calore E., Tringali S., Zecca M., Secondino S., Guiducci B., Pelosini M., Zuffa E., Facchini L., Imola M., Iori A. P., Proia A., Sica S., Armiento D., Carella A. M., Dellacasa C. M., Fagioli F., Rabusin M., Ferrario A., Elice F., Russo, D, Polverelli, N, Malagola, M, Farina, M, Leoni, A, Bernardi, S, Mammoliti, S, Sacchi, N, Martino, M, Ciceri, F, Zallio, F, Olivieri, A, Falcioni, S, Storti, G, Michieli, M, Carluccio, P, Grassi, A, Oldani, E, Bonifazi, F, Prete, A, Cavattoni, I, Maffeis, M, Pastore, D, Vacca, A, Caravelli, D, Mirabile, M, Mordini, N, Nozzoli, C, Faraci, M, Federico, V, Ronconi, S, Skert, C, Onida, F, Marcatti, M, Piemontese, S, Narni, F, Balduzzi, A, De Simone, G, Picardi, A, De Gobbi, M, Calore, E, Tringali, S, Zecca, M, Secondino, S, Guiducci, B, Pelosini, M, Zuffa, E, Facchini, L, Imola, M, Iori, A, Proia, A, Sica, S, Armiento, D, Carella, A, Dellacasa, C, Fagioli, F, Rabusin, M, Ferrario, A, Elice, F, Russo D., Polverelli N., Malagola M., Farina M., Leoni A., Bernardi S., Mammoliti S., Sacchi N., Martino M., Ciceri F., Zallio F., Olivieri A., Falcioni S., Storti G., Michieli M., Carluccio P., Grassi A., Oldani E., Bonifazi F., Prete A., Cavattoni I. M., Maffeis M., Pastore D., Vacca A., Caravelli D., Mirabile M., Mordini N., Nozzoli C., Faraci M., Federico V., Ronconi S., Skert C., Onida F., Marcatti M., Piemontese S., Narni F., Balduzzi A., De Simone G., Picardi A., De Gobbi M., Calore E., Tringali S., Zecca M., Secondino S., Guiducci B., Pelosini M., Zuffa E., Facchini L., Imola M., Iori A. P., Proia A., Sica S., Armiento D., Carella A. M., Dellacasa C. M., Fagioli F., Rabusin M., Ferrario A., and Elice F.

Abstract

The Transplant Centers belonging to Gruppo Italiano per il Trapianto di Midollo Osseo (GITMO) conducted a survey with the aim of evaluating the effect of SARS-CoV2 pandemic on the allogeneic transplant activity in Italy. The pandemic period from 1/3/2020 to 31/7/2020 was compared with the same period in 2019. Overall, in 2020 there was a 2.4% reduction in the number of allo-HCT cases compared to 2019. Interestingly, this deflection did not affect the acute leukemia cases (+5.7% in 2020). The use of peripheral blood-derived stem cells (+10.7%) and cryopreservation (97.4% of the centers) was highly adopted in 2020. Despite the sanitary emergency, almost all of the surveyed centers declared no impact of SARS-CoV2 pandemic on the transplant timing and outcomes, and the sanitary policy was positively evaluated by the majority of centers. The emergency measures ensured that only a minority of the allo-HCT patients had been infected by SARS-CoV2; however, a mortality of 42.1% among the allo-HCT patients hospitalized for COVID-19 was recorded. This survey gives us the information that the GITMO Group reacted positively to the pandemic. Thanks to the emergency strategies, the Italian allo-HCT activity continued safely, showing only a minor deflection and offering the same probability of cure to the transplanted patients.

Published
2021

26. Correction: Myeloablative conditioning for allo-HSCT in pediatric ALL: FTBI or chemotherapy?—A multicenter EBMT-PDWP study (Bone Marrow Transplantation, (2020), 55, 8, (1540-1551), 10.1038/s41409-020-0854-0)

Author

Willasch A. M., Willasch, A, Peters, C, Sedlacek, P, Dalle, J, Kitra-Roussou, V, Yesilipek, A, Wachowiak, J, Lankester, A, Prete, A, Hamidieh, A, Ifversen, M, Buechner, J, Krivan, G, Hamladji, R, Diaz-de-Heredia, C, Skorobogatova, E, Michel, G, Locatelli, F, Bertaina, A, Veys, P, Dupont, S, Or, R, Gungor, T, Aleinikova, O, Sufliarska, S, Sundin, M, Rascon, J, Kaare, A, Nemet, D, Fagioli, F, Klingebiel, T, Styczynski, J, Bierings, M, Nagy, K, Abecasis, M, Afanasyev, B, Ansari, M, Vettenranta, K, Alseraihy, A, Chybicka, A, Robinson, S, Bertrand, Y, Kupesiz, A, Ghavamzadeh, A, Campos, A, Pichler, H, Dalissier, A, Labopin, M, Corbacioglu, S, Balduzzi, A, Galimard, J, Bader, P, Willasch A. M., Peters C., Sedlacek P., Dalle J. -H., Kitra-Roussou V., Yesilipek A., Wachowiak J., Lankester A., Prete A., Hamidieh A. A., Ifversen M., Buechner J., Krivan G., Hamladji R. -M., Diaz-de-Heredia C., Skorobogatova E., Michel G., Locatelli F., Bertaina A., Veys P., Dupont S., Or R., Gungor T., Aleinikova O., Sufliarska S., Sundin M., Rascon J., Kaare A., Nemet D., Fagioli F., Klingebiel T. E., Styczynski J., Bierings M., Nagy K., Abecasis M., Afanasyev B., Ansari M., Vettenranta K., Alseraihy A., Chybicka A., Robinson S., Bertrand Y., Kupesiz A., Ghavamzadeh A., Campos A., Pichler H., Dalissier A., Labopin M., Corbacioglu S., Balduzzi A., Galimard J. -E., Bader P., Willasch A. M., Willasch, A, Peters, C, Sedlacek, P, Dalle, J, Kitra-Roussou, V, Yesilipek, A, Wachowiak, J, Lankester, A, Prete, A, Hamidieh, A, Ifversen, M, Buechner, J, Krivan, G, Hamladji, R, Diaz-de-Heredia, C, Skorobogatova, E, Michel, G, Locatelli, F, Bertaina, A, Veys, P, Dupont, S, Or, R, Gungor, T, Aleinikova, O, Sufliarska, S, Sundin, M, Rascon, J, Kaare, A, Nemet, D, Fagioli, F, Klingebiel, T, Styczynski, J, Bierings, M, Nagy, K, Abecasis, M, Afanasyev, B, Ansari, M, Vettenranta, K, Alseraihy, A, Chybicka, A, Robinson, S, Bertrand, Y, Kupesiz, A, Ghavamzadeh, A, Campos, A, Pichler, H, Dalissier, A, Labopin, M, Corbacioglu, S, Balduzzi, A, Galimard, J, Bader, P, Willasch A. M., Peters C., Sedlacek P., Dalle J. -H., Kitra-Roussou V., Yesilipek A., Wachowiak J., Lankester A., Prete A., Hamidieh A. A., Ifversen M., Buechner J., Krivan G., Hamladji R. -M., Diaz-de-Heredia C., Skorobogatova E., Michel G., Locatelli F., Bertaina A., Veys P., Dupont S., Or R., Gungor T., Aleinikova O., Sufliarska S., Sundin M., Rascon J., Kaare A., Nemet D., Fagioli F., Klingebiel T. E., Styczynski J., Bierings M., Nagy K., Abecasis M., Afanasyev B., Ansari M., Vettenranta K., Alseraihy A., Chybicka A., Robinson S., Bertrand Y., Kupesiz A., Ghavamzadeh A., Campos A., Pichler H., Dalissier A., Labopin M., Corbacioglu S., Balduzzi A., Galimard J. -E., and Bader P.

Abstract

The article “Myeloablative conditioning for allo-HSCT in pediatric ALL: FTBI or chemotherapy?—A multicenter EBMT-PDWP study,” written by Andre Manfred Willasch, Christina Peters, Petr Sedlácek, Jean-Hugues Dalle, Vassiliki Kitra-Roussou, Akif Yesilipek, Jacek Wachowiak, Arjan Lankester, Arcangelo Prete, Amir Ali Hamidieh, Marianne Ifversen, Jochen Buechner, Gergely Kriván, Rose-Marie Hamladji, Cristina Diaz-de-Heredia, Elena Skorobogatova, Gérard Michel, Franco Locatelli, Alice Bertaina, Paul Veys, Sophie Dupont, Reuven Or, Tayfun Güngör, Olga Aleinikova, Sabina Sufliarska, Mikael Sundin, Jelena Rascon, Ain Kaare, Damir Nemet, Franca Fagioli, Thomas Erich Klingebiel, Jan Styczynski, Marc Bierings, Kálmán Nagy, Manuel Abecasis, Boris Afanasyev, Marc Ansari, Kim Vettenranta, Amal Alseraihy, Alicja Chybicka, Stephen Robinson, Yves Bertrand, Alphan Kupesiz, Ardeshir Ghavamzadeh, Antonio Campos, Herbert Pichler, Arnaud Dalissier, Myriam Labopin, Selim Corbacioglu, Adriana Balduzzi, Jacques-Emmanuel Galimard, Peter Bader, on behalf of the EBMT Paediatric Diseases Working Party, was originally published online first without Open Access. After publication in volume 55, issue 8, page 1540–1551, the author decided to opt for Open Choice and to make the article an Open Access publication. Therefore, the copyright of the article has been changed to © The Author(s) 2020 and the article is forthwith distributed under the terms of the Creative Commons Attribution 4.0 InternationalS License, which permits use, sharing, adaptation, distribution, and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The images or other third-party material in this article are included in the article’s Creative Commons license, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Comm

Published
2021

27. Using accounting dataset for agricultural sustainability assessment through a multi‐criteria approach: an Italian case study.

Author

Boggia, A., Fagioli, F. F., Paolotti, L., Ruiz, F., Cabello, J.M., and Rocchi, L.

Subjects
AGRICULTURE, SUSTAINABLE agriculture, AGRICULTURAL extension work, SUSTAINABILITY, FARM income, AGRICULTURAL laborers
Abstract

Sustainability has become a key issue for agricultural systems. In this context, sustainability assessment implies considering different aspects of agricultural activities, including environmental, economic and social issues, and it is usually based on practice‐based indicators. However, proposed methods and applications are still rare due to the difficulty to build a strong general framework and to find appropriate data. The present work develops a performance indicator framework using farm accounting data from the Italian Agricultural Accounting Information Network and proposes a multi‐criteria approach based on the multiple reference point weak‐strong composite indicators (MRP‐WSCI) method to assess the degree of sustainability at the farm level. This method, widely applied to sustainability assessment, calculates the relative rankings including the weak and strong approaches to sustainability and facilitates an understanding of the different indices used, but it is still unused in the agricultural sector. The proposed evaluation framework shows that MRP‐WSCI can handle a holistic set of indicators and rank the farms according to their level of sustainability, making it suitable for the agricultural farm sustainability assessment. Our results show that none of the farms in the case study sample can be classified as "good," according to some of the indices produced, that is, the strong–strong and strong–weak index, while just two of them are good for the weak–weak index. Although the size of the sample used is small, the methodological approach proposed can be straightforwardly extended to larger samples. This methodology can provide information regarding the sustainability of farms to regional and/or national agricultural offices, agricultural extension workers, and farmers themselves, helping in understanding issues in specific dimensions. [ABSTRACT FROM AUTHOR]

Published
2023
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28. Long-term effect of anticancer therapy on dentition of Italian children in remission from malignant disease: A cross-sectional study

Author

Guagnano, R., Romano, F., Berger, M., Fagioli, F., Vallone, V., Bello, L., Vitale, M. C., and Defabianis, P.

Subjects
Adverse effects, DMF Index, Tooth Abnormalities, Anomalies, Dental Caries, Cross-Sectional Studies, Caries, Dental, Malignancies, Child, Dentition, Humans, Prevalence, Anodontia
Abstract

To investigate the effects of anticancer therapy on dental development and caries formation in Italian childhood cancer survivors compared to healthy controls.A total of 52 children treated with chemotherapy and/or radiotherapy when younger than 10 years and in remission from at least 2 years, and 52 healthy age- and gender-matched children were consecutively enrolled in this cross-sectional study. All participants were examined for dental caries and enamel defects according to the decayed-missing-filled teeth (dmft/DMFT) index and the Aine rating scale. Panoramic radiographs were taken to estimate dental age and to assess dental abnormalities using the Höltta Defect Index.These children are at high risk for tooth developmental abnormalities and poor dental health and should be closely monitored by a specialist dentist.

Published
2022

30. Changes in Stem Cell Transplant activity and procedures during SARS-CoV2 pandemic in Italy: an Italian Bone Marrow Transplant Group (GITMO) nationwide analysis (TransCOVID-19 Survey)

Author

Russo D., Polverelli N., Malagola M., Farina M., Leoni A., Bernardi S., Mammoliti S., Sacchi N., Martino M., Ciceri F., Zallio F., Olivieri A., Falcioni S., Storti G., Michieli M., Carluccio P., Grassi A., Oldani E., Bonifazi F., Prete A., Cavattoni I. M., Maffeis M., Pastore D., Vacca A., Caravelli D., Mirabile M., Mordini N., Nozzoli C., Faraci M., Federico V., Ronconi S., Skert C., Onida F., Marcatti M., Piemontese S., Narni F., Balduzzi A., De Simone G., Picardi A., De Gobbi M., Calore E., Tringali S., Zecca M., Secondino S., Guiducci B., Pelosini M., Zuffa E., Facchini L., Imola M., Iori A. P., Proia A., Sica S., Armiento D., Carella A. M., Dellacasa C. M., Fagioli F., Rabusin M., Ferrario A., Elice F., Russo, D, Polverelli, N, Malagola, M, Farina, M, Leoni, A, Bernardi, S, Mammoliti, S, Sacchi, N, Martino, M, Ciceri, F, Zallio, F, Olivieri, A, Falcioni, S, Storti, G, Michieli, M, Carluccio, P, Grassi, A, Oldani, E, Bonifazi, F, Prete, A, Cavattoni, I, Maffeis, M, Pastore, D, Vacca, A, Caravelli, D, Mirabile, M, Mordini, N, Nozzoli, C, Faraci, M, Federico, V, Ronconi, S, Skert, C, Onida, F, Marcatti, M, Piemontese, S, Narni, F, Balduzzi, A, De Simone, G, Picardi, A, De Gobbi, M, Calore, E, Tringali, S, Zecca, M, Secondino, S, Guiducci, B, Pelosini, M, Zuffa, E, Facchini, L, Imola, M, Iori, A, Proia, A, Sica, S, Armiento, D, Carella, A, Dellacasa, C, Fagioli, F, Rabusin, M, Ferrario, A, Elice, F, Russo, D., Polverelli, N., Malagola, M., Farina, M., Leoni, A., Bernardi, S., Mammoliti, S., Sacchi, N., Martino, M., Ciceri, F., Zallio, F., Olivieri, A., Falcioni, S., Storti, G., Michieli, M., Carluccio, P., Grassi, A., Oldani, E., Bonifazi, F., Prete, A., Cavattoni, I. M., Maffeis, M., Pastore, D., Vacca, A., Caravelli, D., Mirabile, M., Mordini, N., Nozzoli, C., Faraci, M., Federico, V., Ronconi, S., Skert, C., Onida, F., Marcatti, M., Piemontese, S., Narni, F., Balduzzi, A., De Simone, G., Picardi, A., De Gobbi, M., Calore, E., Tringali, S., Zecca, M., Secondino, S., Guiducci, B., Pelosini, M., Zuffa, E., Facchini, L., Imola, M., Iori, A. P., Proia, A., Sica, S., Armiento, D., Carella, A. M., Dellacasa, C. M., Fagioli, F., Rabusin, M., Ferrario, A., and Elice, F.

Subjects
Homologous, Myeloid, medicine.medical_specialty, 2019-20 coronavirus outbreak, Bone marrow transplant, Coronavirus disease 2019 (COVID-19), Epidemiology, medicine.medical_treatment, Severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2), Hematopoietic stem cell transplantation, Acute, Humans, Italy, Pandemics, RNA, Viral, SARS-CoV-2, Stem Cell Transplantation, Transplantation, Homologous, COVID-19, Hematopoietic Stem Cell Transplantation, Leukemia, Myeloid, Acute, Article, hematopoietic stem cell transplantation, covid-19, pandemic, Internal medicine, Pandemic, medicine, Viral, Acute leukemia, Transplantation, Leukemia, business.industry, Hematology, Stem-cell research, RNA, Stem cell, business
Abstract

The Transplant Centers belonging to Gruppo Italiano per il Trapianto di Midollo Osseo (GITMO) conducted a survey with the aim of evaluating the effect of SARS-CoV2 pandemic on the allogeneic transplant activity in Italy. The pandemic period from 1/3/2020 to 31/7/2020 was compared with the same period in 2019. Overall, in 2020 there was a 2.4% reduction in the number of allo-HCT cases compared to 2019. Interestingly, this deflection did not affect the acute leukemia cases (+5.7% in 2020). The use of peripheral blood-derived stem cells (+10.7%) and cryopreservation (97.4% of the centers) was highly adopted in 2020. Despite the sanitary emergency, almost all of the surveyed centers declared no impact of SARS-CoV2 pandemic on the transplant timing and outcomes, and the sanitary policy was positively evaluated by the majority of centers. The emergency measures ensured that only a minority of the allo-HCT patients had been infected by SARS-CoV2; however, a mortality of 42.1% among the allo-HCT patients hospitalized for COVID-19 was recorded. This survey gives us the information that the GITMO Group reacted positively to the pandemic. Thanks to the emergency strategies, the Italian allo-HCT activity continued safely, showing only a minor deflection and offering the same probability of cure to the transplanted patients.

Published
2021

31. CD56, HLA-DR, and CD45 recognize a subtype of childhood AML harboring CBFA2T3-GLIS2 fusion transcript

Author

Zangrando, A., Cavagnero, F., Scarparo, P., Varotto, E., Francescato, S., Tregnago, C., Cuccurullo, R., Fagioli, F., Nigro, L. L., Masetti, R., Putti, M. C., Rizzari, C., Santoro, N., Pession, A., Pigazzi, M., Locatelli, F., Basso, G., Buldini, B., Zangrando, A, Cavagnero, F, Scarparo, P, Varotto, E, Francescato, S, Tregnago, C, Cuccurullo, R, Fagioli, F, Nigro, L, Masetti, R, Putti, M, Rizzari, C, Santoro, N, Pession, A, Pigazzi, M, Locatelli, F, Basso, G, Buldini, B, Zangrando A., Cavagnero F., Scarparo P., Varotto E., Francescato S., Tregnago C., Cuccurullo R., Fagioli F., Nigro L.L., Masetti R., Putti M.C., Rizzari C., Santoro N., Pession A., Pigazzi M., Locatelli F., Basso G., and Buldini B.

Subjects
Oncogene Proteins, Fusion, Brief Report, HLA-DR Antigens, acute myeloid leukemia, Repressor Proteins, CBFA2T3‐GLIS2, Leukemia, Myeloid, Acute, computational analysi, Settore MED/38 - PEDIATRIA GENERALE E SPECIALISTICA, immunophenotyping, computational analysis, hemic and lymphatic diseases, Humans, CBFA2T3-GLIS2, Child, Transcriptome
Abstract

The presence of CBFA2T3‐GLIS2 fusion gene has been identified in childhood Acute Myeloid Leukemia (AML). In view of the genomic studies indicating a distinct gene expression profile, we evaluated the role of immunophenotyping in characterizing a rare subtype of AML‐CBFA2T3‐GLIS2 rearranged. Immunophenotypic data were obtained by studying a cohort of 20 pediatric CBFA2T3‐GLIS2‐AML and 77 AML patients not carrying the fusion transcript. Enrolled cases were included in the Associazione Italiana di Ematologia Oncologia Pediatrica (AIEOP) AML trials and immunophenotypes were compared using different statistical approaches. By multiple computational procedures, we identified two main core antigens responsible for the identification of the CBFA2T3‐GLIS2‐AML. CD56 showed the highest performance in single marker evaluation (AUC = 0.89) and granted the most accurate prediction when used in combination with HLA‐DR (AUC = 0.97) displaying a 93% sensitivity and 99% specificity. We also observed a weak‐to‐negative CD45 expression, being exceptional in AML. We here provide evidence that the combination of HLA‐DR negativity and intense bright CD56 expression detects a rare and aggressive pediatric AML genetic lesion improving the diagnosis performance.

Published
2021

32. Soft tissue and visceral sarcomas

Author

Gronchi, A., Miah, A.B., DeiTos, A.P., Abecassis, N., Bajpai, J., Bauer, S., Biagini, R., Bielack, S., Blay, J.Y., Bolle, S., Bonvalot, S., Boukovinas, I., Bovee, J.V.M.G., Boye, K., Brennan, B., Brodowicz, T., Buonadonna, A., DeAlava, E., Muro, X.G. del, Dufresne, A., Eriksson, M., Fagioli, F., Fedenko, A., Ferraresi, V., Ferrari, A., Frezza, A.M., Gasperoni, S., Gelderblom, H., Gouin, F., Grignani, G., Haas, R., Hassan, A.B., Hecker-Nolting, S., Hindi, N., Hohenberger, P., Joensuu, H., Jones, R.L., Jungels, C., Jutte, P., Kager, L., Kasper, B., Kawai, A., Kopeckova, K., Krakorova, D.A., Cesne, A. le, LeGrange, F., Legius, E., Leithner, A., Lopez-Pousa, A., Martin-Broto, J., Merimsky, O., Messiou, C., Mir, O., Montemurro, M., Morland, B., Morosi, C., Palmerini, E., Pantaleo, M.A., Piana, R., Piperno-Neumann, S., Reichardt, P., Rutkowski, P., Safwat, A.A., Sangalli, C., Sbaraglia, M., Scheipl, S., Schoffski, P., Sleijfer, S., Strauss, D., Strauss, S., SundbyHall, K., Trama, A., Unk, M., VandeSande, M.A.J., VanderGraaf, W.T.A., Houdt, W.J. van, Frebourg, T., Casali, P.G., Stacchiotti, S., ESMO Guidelines Comm, EURACAN, GENTURIS, ​Basic and Translational Research and Imaging Methodology Development in Groningen (BRIDGE), Public Health Research (PHR), Man, Biomaterials and Microbes (MBM), Gronchi A., Miah A.B., Dei Tos A.P., Abecassis N., Bajpai J., Bauer S., Biagini R., Bielack S., Blay J.Y., Bolle S., Bonvalot S., Boukovinas I., Bovee J.V.M.G., Boye K., Brennan B., Brodowicz T., Buonadonna A., De Alava E., Del Muro X.G., Dufresne A., Eriksson M., Fagioli F., Fedenko A., Ferraresi V., Ferrari A., Frezza A.M., Gasperoni S., Gelderblom H., Gouin F., Grignani G., Haas R., Hassan A.B., Hecker-Nolting S., Hindi N., Hohenberger P., Joensuu H., Jones R.L., Jungels C., Jutte P., Kager L., Kasper B., Kawai A., Kopeckova K., Krakorova D.A., Le Cesne A., Le Grange F., Legius E., Leithner A., Lopez-Pousa A., Martin-Broto J., Merimsky O., Messiou C., Mir O., Montemurro M., Morland B., Morosi C., Palmerini E., Pantaleo M.A., Piana R., Piperno-Neumann S., Reichardt P., Rutkowski P., Safwat A.A., Sangalli C., Sbaraglia M., Scheipl S., Schoffski P., Sleijfer S., Strauss D., Strauss S., Sundby Hall K., Trama A., Unk M., van de Sande M.A.J., van der Graaf W.T.A., van Houdt W.J., Frebourg T., Casali P.G., and Stacchiotti S.

Subjects
sarcoma, uterine sarcoma, treatment, diagnosis, desmoid, Medizin, Sarcoma, Soft Tissue Neoplasms, Hematology, Clinical Practice Guideline, Soft tissue sarcomas, retroperitoneal sarcoma, Retroperitoneal Sarcoma, soft tissue sarcomas, Follow-Up Studie, Desmoid, Uterine Sarcoma, follow-up, management, Oncology, soft tissue sarcoma, Human
Abstract

ispartof: ANNALS OF ONCOLOGY vol:32 issue:11 pages:1348-1365 ispartof: location:England status: published

Published
2021

33. S204: PEMBROLIZUMAB IN CHILDREN AND YOUNG ADULTS WITH NEWLY DIAGNOSED CLASSICAL HODGKIN LYMPHOMA WITH SLOW EARLY RESPONSE TO FRONTLINE CHEMOTHERAPY: THE PHASE 2, OPEN-LABEL, KEYNOTE-667 STUDY

Author

Vinti, L., primary, Daw, S., additional, Sabado Alvarez, C., additional, Fagioli, F., additional, Beishuizen, A., additional, Michel, G., additional, Moleti, M. L., additional, Cepelova, M., additional, Thorwarth, A., additional, Rigaud, C., additional, Plaza Lopez de Sabando, D., additional, Landman Parker, J., additional, Zhu, Y., additional, Pillai, P., additional, Nahar, A., additional, and Mauz-Koerholz, C., additional

Published
2022
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34. P1088: UPDATED DATA FROM A PHASE 1/2 STUDY OF BRENTUXIMAB VEDOTIN COMBINED WITH CHEMOTHERAPY IN PEDIATRIC PATIENTS WITH ADVANCED STAGE CLASSICAL HODGKIN LYMPHOMA

Author

Locatelli, F., primary, Luisi, F., additional, Pianovski, M., additional, Salvino, M. A., additional, Fagioli, F., additional, Epelman, S., additional, Britto De Abreu Lima, L., additional, Norris, R., additional, Odone Filho, V., additional, Zecca, M., additional, Favre, C., additional, Kobayashi, R., additional, Koga, Y., additional, Sidi, Y., additional, Zhou, X., additional, Bai, X., additional, Campana, F., additional, Leonard, E. J., additional, and Franklin, A. R., additional

Published
2022
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35. Children and coronavirus: The necessary search for a balance between the alleged risks and documented collateral damage [Bambini e coronavirus: La doverosa ricerca di un equilibrio tra i presunti rischi e i documentati danni collaterali]

Author

Tamburlini, G, Marchetti, F, Bertino, E, Bestetti, G, Biasucci, G, Biondi, A, Bonati, M, Brunelli, A, Corsello, G, Esposito, S, Fagioli, F, Farina, D, Gagliardi, L, Gangemi, M, Greco, L, Lanari, M, Lazzerini, M, Maggiore, G, Martelossi, S, Ramenghi, U, Piga, A, Selicorni, A, Spada, M, Ventura, A, Vicari, S, Zampino, G, Zanetto, F, Tamburlini G., Marchetti F., Bertino E., Bestetti G., Biasucci G., Biondi A., Bonati M., Brunelli A., Corsello G., Esposito S., Fagioli F., Farina D., Gagliardi L., Gangemi M., Greco L., Lanari M., Lazzerini M., Maggiore G., Martelossi S., Ramenghi U., Piga A., Selicorni A., Spada M., Ventura A., Vicari S., Zampino G., Zanetto F., Tamburlini, G, Marchetti, F, Bertino, E, Bestetti, G, Biasucci, G, Biondi, A, Bonati, M, Brunelli, A, Corsello, G, Esposito, S, Fagioli, F, Farina, D, Gagliardi, L, Gangemi, M, Greco, L, Lanari, M, Lazzerini, M, Maggiore, G, Martelossi, S, Ramenghi, U, Piga, A, Selicorni, A, Spada, M, Ventura, A, Vicari, S, Zampino, G, Zanetto, F, Tamburlini G., Marchetti F., Bertino E., Bestetti G., Biasucci G., Biondi A., Bonati M., Brunelli A., Corsello G., Esposito S., Fagioli F., Farina D., Gagliardi L., Gangemi M., Greco L., Lanari M., Lazzerini M., Maggiore G., Martelossi S., Ramenghi U., Piga A., Selicorni A., Spada M., Ventura A., Vicari S., Zampino G., and Zanetto F.

Published
2020

36. PACSIN2 rs2413739 influence on thiopurine pharmacokinetics: validation studies in pediatric patients

Author

Franca, R, Stocco, G, Favretto, D, Giurici, N, del Rizzo, I, Locatelli, F, Vinti, L, Biondi, A, Colombini, A, Fagioli, F, Barisone, E, Pelin, M, Martellossi, S, Ventura, A, Decorti, G, Rabusin, M, Franca R., Stocco G., Favretto D., Giurici N., del Rizzo I., Locatelli F., Vinti L., Biondi A., Colombini A., Fagioli F., Barisone E., Pelin M., Martellossi S., Ventura A., Decorti G., Rabusin M., Franca, R, Stocco, G, Favretto, D, Giurici, N, del Rizzo, I, Locatelli, F, Vinti, L, Biondi, A, Colombini, A, Fagioli, F, Barisone, E, Pelin, M, Martellossi, S, Ventura, A, Decorti, G, Rabusin, M, Franca R., Stocco G., Favretto D., Giurici N., del Rizzo I., Locatelli F., Vinti L., Biondi A., Colombini A., Fagioli F., Barisone E., Pelin M., Martellossi S., Ventura A., Decorti G., and Rabusin M.

Abstract

The aim of the study was to validate the impact of the single-nucleotide polymorphism rs2413739 (T > C) in the PACSIN2 gene on thiopurines pharmacological parameters and clinical response in an Italian cohort of pediatric patients with acute lymphoblastic leukemia (ALL) and inflammatory bowel disease (IBD). In ALL, PACSIN2 rs2413739 T allele was associated with a significant reduction of TPMT activity in erythrocytes (p = 0.0094, linear mixed-effect model, multivariate analysis considering TPMT genotype) and increased severe gastrointestinal toxicity during consolidation therapy (p = 0.049). A similar trend was present also for severe hematological toxicity during maintenance. In IBD, no significant effect of rs2413739 could be found on TPMT activity, however azathioprine effectiveness was reduced in patients carrying the T allele (linear mixed effect, p = 0.0058). In PBMC from healthy donors, a positive correlation between PACSIN2 and TPMT protein concentration could be detected (linear mixed effect, p = 0.045). These results support the role of PACSIN2 polymorphism on TPMT activity and mercaptopurine adverse effects in patients with ALL. Further evidence on PBMC and pediatric patients with IBD supports an association between PACSIN2 variants, TPMT activity, and thiopurines effects, even if more studies are needed since some of these effects may be tissue specific.

Published
2020

37. Occurrence of long-term effects after hematopoietic stem cell transplantation in children affected by acute leukemia receiving either busulfan or total body irradiation: results of an AIEOP (Associazione Italiana Ematologia Oncologia Pediatrica) retrospective study

Author

Saglio, F, Zecca, M, Pagliara, D, Giorgiani, G, Balduzzi, A, Calore, E, Favre, C, Faraci, M, Prete, A, Tambaro, F, Quarello, P, Locatelli, F, Fagioli, F, Saglio F., Zecca M., Pagliara D., Giorgiani G., Balduzzi A., Calore E., Favre C., Faraci M., Prete A., Tambaro F. P., Quarello P., Locatelli F., Fagioli F., Saglio, F, Zecca, M, Pagliara, D, Giorgiani, G, Balduzzi, A, Calore, E, Favre, C, Faraci, M, Prete, A, Tambaro, F, Quarello, P, Locatelli, F, Fagioli, F, Saglio F., Zecca M., Pagliara D., Giorgiani G., Balduzzi A., Calore E., Favre C., Faraci M., Prete A., Tambaro F. P., Quarello P., Locatelli F., and Fagioli F.

Abstract

Patients given allogeneic hematopoietic stem cell transplantation (alloHSCT) present an increased incidence of long-term toxicities that can be attributed to the preparative regimen. We retrospectively analyzed in a population of 670 children receiving allo-HSCT for acute leukemia the occurrence of different late effects in function of the choice made between total body irradiation (TBI) and busulfan, as part of the preparative regimen. In univariable analysis, we found that patients treated with TBI developed cataract in 24% of the cases compared with 4% in patients treated with BU (p = 0.0001) and that the incidence of secondary malignant neoplasia (SMN) was higher in patients treated with TBI (18%) as compared with those prepared to the allograft with a Bu-based regimen (0%) (p = 0.019). Conditioning regimen did not show a statistically significant correlation with the occurrence of all the other investigated late effects. In multivariable analysis, TBI remained associated with the occurrence of cataracts (Relative Risk: 0.33 p = 0.012) and secondary malignancies (Relative Risk 3.96 × 10e−6 p < 0.001); however, other variables, as GvHD and disease type, were also correlated with these long-term sequels, indicating that in our study population the preparative regimen is not the only factor influencing the incidence of these complications.

Published
2020

38. Hematopoietic stem cell transplantation for adolescents and adults with inborn errors of immunity: an EBMT IEWP study

Author

Albert, M. H., Sirait, T., Eikema, D. -J., Bakunina, K., Wehr, C., Suarez, F., Fox, M. L., Mahlaoui, N., Gennery, A. R., Lankester, A. C., Beier, R., Bernardo, M. E., Bigley, V., Lindemans, C. A., Burns, S. O., Carpenter, B., Dybko, J., Gungor, T., Hauck, F., Lum, S. H., Balashov, D., Meisel, R., Moshous, D., Schulz, A., Speckmann, C., Slatter, M. A., Strahm, B., Uckan-Cetinkaya, D., Meyts, I., Vallee, T. C., Wynn, R., Neven, B., Morris, E. C., Aiuti, A., Maschan, A., Aljurf, M., Gedde-Dahl, T., Gurman, G., Bordon, V., Krivan, G., Locatelli, Franco, Porta, F., Valcarcel, D., Beguin, Y., Faraci, M., Kroger, N., Kulagin, A., Shaw, P. J., Veelken, J. H., Diaz de Heredia, C., Fagioli, F., Felber, M., Gruhn, B., Holter, W., Rossig, C., Sedlacek, P., Apperley, J., Ayas, M., Bodova, I., Choi, G., Cornelissen, J. J., Sirvent, A., Khan, A., Kupesiz, A., Lenhoff, S., Ozdogu, H., von der Weid, N., Rovira, M., Schots, R., Vinh, D. C., Locatelli F. (ORCID:0000-0002-7976-3654), Albert, M. H., Sirait, T., Eikema, D. -J., Bakunina, K., Wehr, C., Suarez, F., Fox, M. L., Mahlaoui, N., Gennery, A. R., Lankester, A. C., Beier, R., Bernardo, M. E., Bigley, V., Lindemans, C. A., Burns, S. O., Carpenter, B., Dybko, J., Gungor, T., Hauck, F., Lum, S. H., Balashov, D., Meisel, R., Moshous, D., Schulz, A., Speckmann, C., Slatter, M. A., Strahm, B., Uckan-Cetinkaya, D., Meyts, I., Vallee, T. C., Wynn, R., Neven, B., Morris, E. C., Aiuti, A., Maschan, A., Aljurf, M., Gedde-Dahl, T., Gurman, G., Bordon, V., Krivan, G., Locatelli, Franco, Porta, F., Valcarcel, D., Beguin, Y., Faraci, M., Kroger, N., Kulagin, A., Shaw, P. J., Veelken, J. H., Diaz de Heredia, C., Fagioli, F., Felber, M., Gruhn, B., Holter, W., Rossig, C., Sedlacek, P., Apperley, J., Ayas, M., Bodova, I., Choi, G., Cornelissen, J. J., Sirvent, A., Khan, A., Kupesiz, A., Lenhoff, S., Ozdogu, H., von der Weid, N., Rovira, M., Schots, R., Vinh, D. C., and Locatelli F. (ORCID:0000-0002-7976-3654)

Abstract

Allogeneic hematopoietic stem cell transplantation (HSCT) is the gold standard curative therapy for infants and children with many inborn errors of immunity (IEI), but adolescents and adults with IEI are rarely referred for transplant. Lack of published HSCT outcome data outside small, single-center studies and perceived high risk of transplant-related mortality have delayed the adoption of HSCT for IEI patients presenting or developing significant organ damage later in life. This large retrospective, multicenter HSCT outcome study reports on 329 IEI patients (age range, 15-62.5 years at HSCT). Patients underwent first HSCT between 2000 and 2019. Primary endpoints were overall survival (OS) and event-free survival (EFS). We also evaluated the influence of IEI-subgroup and IEI-specific risk factors at HSCT, including infections, bronchiectasis, colitis, malignancy, inflammatory lung disease, splenectomy, hepatic dysfunction, and systemic immunosuppression. At a median follow-up of 44.3 months, the estimated OS at 1 and 5 years post-HSCT for all patients was 78% and 71%, and EFS was 65% and 62%, respectively, with low rates of severe acute (8%) or extensive chronic (7%) graft-versus-host disease. On univariate analysis, OS and EFS were inferior in patients with primary antibody deficiency, bronchiectasis, prior splenectomy, hepatic comorbidity, and higher hematopoietic cell transplant comorbidity index scores. On multivariable analysis, EFS was inferior in those with a higher number of IEI-associated complications. Neither age nor donor had a significant effect on OS or EFS. We have identified age-independent risk factors for adverse outcome, providing much needed evidence to identify which patients are most likely to benefit from HSCT.

Published
2022

46. No improvement of survival with reduced- versus high-intensity conditioning for allogeneic stem cell transplants in Ewing tumor patients

Author

Thiel, U., Wawer, A., Wolf, P., Badoglio, M., Santucci, A., Klingebiel, T., Basu, O., Borkhardt, A., Laws, H.-J., Kodera, Y., Yoshimi, A., Peters, C., Ladenstein, R., Pession, A., Prete, A., Urban, E.-C., Schwinger, W., Bordigoni, P., Salmon, A., Diaz, M.A., Afanasyev, B., Lisukov, I., Morozova, E., Toren, A., Bielorai, B., Korsakas, J., Fagioli, F., Caselli, D., Ehninger, G., Gruhn, B., Dirksen, U., Abdel-Rahman, F., Aglietta, M., Mastrodicasa, E., Torrent, M., Corradini, P., Demeocq, F., Dini, G., Dreger, P., Eyrich, M., Gozdzik, J., Guilhot, F., Holler, E., Koscielniak, E., Messina, C., Nachbaur, D., Sabbatini, R., Oldani, E., Ottinger, H., Ozsahin, H., Schots, R., Siena, S., Stein, J., Sufliarska, S., Unal, A., Ussowicz, M., Schneider, P., Woessmann, W., Jürgens, H., Bregni, M., and Burdach, S.

Published
2011
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47. Nonmetastatic Ewing family tumors: high-dose chemotherapy with stem cell rescue in poor responder patients. Results of the Italian Sarcoma Group/Scandinavian Sarcoma Group III protocol

Author

Ferrari, S., Sundby Hall, K., Luksch, R., Tienghi, A., Wiebe, T., Fagioli, F., Alvegard, T.A., Brach del Prever, A., Tamburini, A., Alberghini, M., Gandola, L., Mercuri, M., Capanna, R., Mapelli, S., Prete, A., Carli, M., Picci, P., Barbieri, E., Bacci, G., and Smeland, S.

Published
2011
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48. FDG PET in response evaluation of bulky masses in paediatric Hodgkin’s lymphoma (HL) patients enrolled in the Italian AIEOP-LH2004 trial

Author

Lopci, E, Mascarin, M, Piccardo, A, Castello, A, Elia, C, Guerra, L, Borsatti, E, Sala, A, Todesco, A, Zucchetta, P, Farruggia, P, Cistaro, A, Buffardi, S, Bertolini, P, Bianchi, M, Moleti, M, Bunkheila, F, Indolfi, P, Fagioli, F, Garaventa, A, Burnelli, R, Lopci E., Mascarin M., Piccardo A., Castello A., Elia C., Guerra L., Borsatti E., Sala A., Todesco A., Zucchetta P., Farruggia P., Cistaro A., Buffardi S., Bertolini P., Bianchi M., Moleti M. L., Bunkheila F., Indolfi P., Fagioli F., Garaventa A., Burnelli R., Lopci, E, Mascarin, M, Piccardo, A, Castello, A, Elia, C, Guerra, L, Borsatti, E, Sala, A, Todesco, A, Zucchetta, P, Farruggia, P, Cistaro, A, Buffardi, S, Bertolini, P, Bianchi, M, Moleti, M, Bunkheila, F, Indolfi, P, Fagioli, F, Garaventa, A, Burnelli, R, Lopci E., Mascarin M., Piccardo A., Castello A., Elia C., Guerra L., Borsatti E., Sala A., Todesco A., Zucchetta P., Farruggia P., Cistaro A., Buffardi S., Bertolini P., Bianchi M., Moleti M. L., Bunkheila F., Indolfi P., Fagioli F., Garaventa A., and Burnelli R.

Abstract

Purpose: We present the results of an investigation of the role of FDG PET in response evaluation of bulky masses in paediatric patients with Hodgkin’s lymphoma (HL) enrolled in the Italian AIEOP-LH2004 trial. Methods: We analysed data derived from 703 patients (388 male, 315 female; mean age 13 years) with HL and enrolled in 41 different Italian centres from March 2004 to September 2012, all treated with the AIEOP-LH2004 protocol. The cohort comprised 309 patients with a bulky mass, of whom 263 were evaluated with FDG PET at baseline and after four cycles of chemotherapy. Responses were determined according to combined functional and morphological criteria. Patients were followed up for a mean period of 43 months and for each child we calculated time-to-progression (TTP) and relapse rates considering clinical monitoring, and instrumental and histological data as the reference standard. Statistical analyses were performed for FDG PET and morphological responses with respect to TTP. Multivariate analysis was used to define independent predictive factors. Results: Overall, response evaluation revealed 238 PET-negative patients (90.5%) and 25 PET-positive patients (9.5%), with a significant difference in TTP between these groups (mean TTP: 32.67 months for negative scans, 23.8 months for positive scans; p < 0.0001, log-rank test). In the same cohort, computed tomography showed a complete response (CR) in 85 patients (32.3%), progressive disease (PD) in 6 patients (2.3%), and a partial response (PR) in 165 patients (62.7%), with a significant difference in TTP between patients with CR and patients with PD (31.1 months and 7.9 months, respectively; p < 0.001, log-rank test). Similarly, there was a significant difference in relapse rates between PET-positive and PET-negative patients (p = 0000). In patients with PR, there was also a significant difference in TTP between PET-positive and PET-negative patients (24.6 months and 34.9 months, respectively; p < 0.0001). In the

Published
2019

49. Nationwide central diagnosis review for childhood solid tumors: From concept to realization of an Associazione Italiana Ematologia Oncologia Pediatrica (AIEOP) integrated project

Author

Spreafico, F, Quarello, P, Alaggio, R, Collini, P, Haupt, R, Mastronuzzi, A, Prete, A, Rabusin, M, Rizzari, C, Schumacher, F, Zecca, M, Pession, A, Fagioli, F, Spreafico F., Quarello P., Alaggio R., Collini P., Haupt R., Mastronuzzi A., Prete A., Rabusin M., Rizzari C., Schumacher F., Zecca M., Pession A., Fagioli F., Spreafico, F, Quarello, P, Alaggio, R, Collini, P, Haupt, R, Mastronuzzi, A, Prete, A, Rabusin, M, Rizzari, C, Schumacher, F, Zecca, M, Pession, A, Fagioli, F, Spreafico F., Quarello P., Alaggio R., Collini P., Haupt R., Mastronuzzi A., Prete A., Rabusin M., Rizzari C., Schumacher F., Zecca M., Pession A., and Fagioli F.

Published
2019

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