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1. Diagnosis and management of acquired aplastic anemia in childhood. Guidelines from the Marrow Failure Study Group of the Pediatric Haemato-Oncology Italian Association (AIEOP)

Author

Guarina, A., Farruggia, P., Mariani, E., Saracco, P., Barone, A., Onofrillo, D., Cesaro, S., Angarano, R., Barberi, W., Bonanomi, S., Corti, P., Crescenzi, B., Dell'Orso, G., De Matteo, A., Giagnuolo, G., Iori, A.P., Ladogana, S., Lucarelli, A., Lupia, M., Martire, B., Mastrodicasa, E., Massaccesi, E., Arcuri, L., Giarratana, M.C., Menna, G., Miano, M., Notarangelo, L.D., Palazzi, G., Palmisani, E., Pestarino, S., Pierri, F., Pillon, M., Ramenghi, U., Russo, G., Saettini, F., Timeus, F., Verzegnassi, F., Zecca, M., Fioredda, F., and Dufour, C.

Published
2024
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4. 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

5. Rituximab Unveils Hypogammaglobulinemia and Immunodeficiency in Children with Autoimmune Cytopenia

Author

Ottaviano, G, Marinoni, M, Graziani, S, Sibson, K, Barzaghi, F, Bertolini, P, Chini, L, Corti, P, Cancrini, C, D'Alba, I, Gabelli, M, Gallo, V, Giancotta, C, Giordano, P, Lassandro, G, Martire, B, Angarano, R, Mastrodicasa, E, Bava, C, Miano, M, Naviglio, S, Verzegnassi, F, Saracco, P, Trizzino, A, Biondi, A, Pignata, C, Moschese, V, Ottaviano G., Marinoni M., Graziani S., Sibson K., Barzaghi F., Bertolini P., Chini L., Corti P., Cancrini C., D'Alba I., Gabelli M., Gallo V., Giancotta C., Giordano P., Lassandro G., Martire B., Angarano R., Mastrodicasa E., Bava C., Miano M., Naviglio S., Verzegnassi F., Saracco P., Trizzino A., Biondi A., Pignata C., Moschese V., Ottaviano, G, Marinoni, M, Graziani, S, Sibson, K, Barzaghi, F, Bertolini, P, Chini, L, Corti, P, Cancrini, C, D'Alba, I, Gabelli, M, Gallo, V, Giancotta, C, Giordano, P, Lassandro, G, Martire, B, Angarano, R, Mastrodicasa, E, Bava, C, Miano, M, Naviglio, S, Verzegnassi, F, Saracco, P, Trizzino, A, Biondi, A, Pignata, C, Moschese, V, Ottaviano G., Marinoni M., Graziani S., Sibson K., Barzaghi F., Bertolini P., Chini L., Corti P., Cancrini C., D'Alba I., Gabelli M., Gallo V., Giancotta C., Giordano P., Lassandro G., Martire B., Angarano R., Mastrodicasa E., Bava C., Miano M., Naviglio S., Verzegnassi F., Saracco P., Trizzino A., Biondi A., Pignata C., and Moschese V.

Abstract

Background: Rituximab (RTX; anti-CD20 mAb) is a treatment option in children with refractory immune thrombocytopenia, autoimmune hemolytic anemia (AHA), and Evans syndrome (ES). Prevalence and clinical course of RTX-induced hypogammaglobulinemia in these patients are poorly known. Objective: To evaluate the prevalence and risk factors for persistent hypogammaglobulinemia (PH) after RTX use. Methods: Clinical and immunologic data from children treated with RTX for immune thrombocytopenia, AHA, and ES were collected from 16 Italian centers and 1 UK center at pre-RTX time point (0), +6 months, and yearly, up to 4 years post-RTX. Patients with previously diagnosed malignancy or primary immune deficiency (PID) were excluded. Results: We analyzed 53 children treated with RTX for immune thrombocytopenia (n = 36), AHA (n = 13), and ES (n = 4). Median follow-up was 30 months (range, 12-48). Thirty-two percent of patients (17 of 53) experienced PH, defined as IgG levels less than 2 SD for age at last follow-up (>12 months after RTX). Significantly delayed B-cell recovery was observed in children experiencing PH (hazard ratio, 0.55; P <.05), and 6 of 17 (35%) patients had unresolved B-cell lymphopenia at last follow-up. PH was associated with IgA and IgM deficiency, younger age at RTX use (51 vs 116 months; P <.01), a diagnosis of AHA/ES, and better response to RTX. Nine patients with PH (9 of 17 [53%]) were eventually diagnosed with a PID. Conclusions: Post-RTX PH is a frequent condition in children with autoimmune cytopenia; a sizable proportion of patients with post-RTX PH were eventually diagnosed with a PID. In-depth investigation for PID is therefore recommended in these patients.

Published
2020

6. Whole-Body MRI versus an FDG-PET/CT-Based Reference Standard for Early Response Assessment and Restaging of Paediatric Hodgkin's Lymphoma: A Prospective Multicentre Study

Author

Arts-assistenten Radiologie, Cancer, MS Radiologie, Brain, PMC Medisch specialisten, Haematologie patientenzorg, Child Health, Spijkers, S., Littooij, A., Kwee, T., Tolboom, N., Beishuizen, A., Bruin, M., Enriquez, G., Sabado, C., Miller, E., Granata, C., De lange, C., Verzegnassi, F., Keizer, B., Nievelstein, R. -J., Arts-assistenten Radiologie, Cancer, MS Radiologie, Brain, PMC Medisch specialisten, Haematologie patientenzorg, Child Health, Spijkers, S., Littooij, A., Kwee, T., Tolboom, N., Beishuizen, A., Bruin, M., Enriquez, G., Sabado, C., Miller, E., Granata, C., De lange, C., Verzegnassi, F., Keizer, B., and Nievelstein, R. -J.

Published
2021

7. Normative growth charts for Shwachman-Diamond syndrome from Italian cohort of 0-8 years old

Author

Cipolli, Marco, Tridello, Gloria, Micheletto, Alessio, Perobelli, Sandra, Pintani, Emily, Cesaro, Simone, Maserati, Emanuela, Nicolis, Elena, Danesino, Cesare, Ambroni, M, Caniglia, M, Cantarini, Me, Corti, P, Farrugia, P, Frau, Mr, Fuoti, M, Indolfi, G, Ladogana, S, Lucidi, V, Rosaria Matarese SM, Menna, G, Montemitro, E, Nardi, M, Nasi, C, Nocerino, A, Pericoli, R, Raia, V, Ramenghi, U, Sainati, L, Tucci, F, Verzegnassi, F, Zecca, M, Zucchini, A., Cipolli, M, Tridello, G, Micheletto, A, Perobelli, S, Pintani, E, Cesaro, S, Maserati, E, Nicoli, Massimiliano, Danesino CAmbroni, M, Caniglia, M, Cantarini, Me, Corti, P, Farrugia, P, Frau, Mr, Fuoti, M, Indolfi, G, Ladogana, S, Lucidi, V, Rosaria Matarese, Sm, Menna, G, Montemitro, E, Nardi, M, Nasi, C, Nocerino, A, Pericoli, MARIO ANDREA, Raia, V, Ramenghi, U, Sainati, L, Tucci, F, Verzegnassi, F, Zecca, M, and Zucchini, A.

Subjects
Male, Percentile, Pediatrics, medicine.medical_specialty, Birth weight, Population, Shwachman-diamond syndrome, Shwachman–diamond syndrome, genetics, growth charts, Short stature, Body Mass Index, Medicine (all), 03 medical and health sciences, 0302 clinical medicine, Reference Values, 030225 pediatrics, medicine, Humans, Registries, Child, education, Retrospective Studies, 030304 developmental biology, 0303 health sciences, education.field_of_study, business.industry, Research, Body Weight, Infant, Newborn, Infant, Genetics and Genomics, Retrospective cohort study, General Medicine, Body Height, Italy, Child, Preschool, Mutation, Failure to thrive, Cohort, Female, medicine.symptom, business, Body mass index
Abstract

ObjectivesShwachman-Diamond syndrome (SDS) is a rare autosomal recessive disorder. Its predominant manifestations include exocrine pancreatic insufficiency, bone marrow failure and skeletal abnormalities. Patients frequently present failure to thrive and susceptibility to short stature. Average birth weight is at the 25th percentile; by the first birthday, >50% of patients drop below the third percentile for height and weight.The study aims at estimating the growth charts for patients affected by SDS in order to give a reference tool helpful for medical care and growth surveillance through the first 8 years of patient’s life.Setting and participantsThis retrospective observational study includes 106 patients (64 M) with available information from birth to 8 years, selected among the 122 patients included in the Italian National Registry of SDS and born between 1975 and 2016. Gender, birth date and auxological parameters at repeated assessment times were collected. The General Additive Model for Location Scale and Shape method was applied to build the growth charts. A set of different distributions was used, and the more appropriate were selected in accordance with the smallest Akaike information criterion.ResultsA total of 408 measurements was collected and analysed. The median number of observations per patient amounted to 3, range 1–11. In accordance with the methods described, specific SDS growth charts were built for weight, height and body mass index (BMI), separately for boys and girls.The 50th and 3rd percentiles of weight and height of the healthy population (WHO standard references) respectively correspond to the 97th and 50th percentiles of the SDS population (SDS specific growth charts), while the difference is less evident for the BMI.ConclusionsSpecific SDS growth charts obtained through our analysis enable a more appropriate classification of patients based on auxological parameters, representing a useful reference tool for evaluating their growth during childhood.

Published
2019

8. Second-line therapy in paediatric warm autoimmune haemolytic anaemia. Guidelines from the Associazione Italiana Onco-Ematologia Pediatrica (AIEOP)

Author

Ladogana, S, Maruzzi, M, Samperi, P, Condorelli, A, Casale, M, Giordano, P, Notarangelo, L, Farruggia, P, Giona, F, Nocerino, A, Fasoli, S, Casciana, M, Miano, M, Tucci, F, Casini, T, Saracco, P, Barcellini, W, Zanella, A, Perrotta, S, Russo, G, Baronci, C, Casadei, A, Cazzaniga, G, Coluzzi, S, Ciliberti, A, Del Vecchio, G, Facchini, E, Girelli, G, Lazzareschi, I, Masera, N, Perseghin, P, Peruccio, D, Petrone, A, Sau, A, Schiro, R, Tumino, M, Vasta, I, Verzegnassi, F, Ladogana S., Maruzzi M., Samperi P., Condorelli A., Casale M., Giordano P., Notarangelo L. D., Farruggia P., Giona F., Nocerino A., Fasoli S., Casciana M. L., Miano M., Tucci F., Casini T., Saracco P., Barcellini W., Zanella A., Perrotta S., Russo G., Baronci C., Casadei A. M., Cazzaniga G., Coluzzi S., Ciliberti A., Del Vecchio G. C., Facchini E., Girelli G., Lazzareschi I., Masera N., Perseghin P., Peruccio D., Petrone A., Sau A., Schiro R., Tumino M., Vasta I., Verzegnassi F., Ladogana, S, Maruzzi, M, Samperi, P, Condorelli, A, Casale, M, Giordano, P, Notarangelo, L, Farruggia, P, Giona, F, Nocerino, A, Fasoli, S, Casciana, M, Miano, M, Tucci, F, Casini, T, Saracco, P, Barcellini, W, Zanella, A, Perrotta, S, Russo, G, Baronci, C, Casadei, A, Cazzaniga, G, Coluzzi, S, Ciliberti, A, Del Vecchio, G, Facchini, E, Girelli, G, Lazzareschi, I, Masera, N, Perseghin, P, Peruccio, D, Petrone, A, Sau, A, Schiro, R, Tumino, M, Vasta, I, Verzegnassi, F, Ladogana S., Maruzzi M., Samperi P., Condorelli A., Casale M., Giordano P., Notarangelo L. D., Farruggia P., Giona F., Nocerino A., Fasoli S., Casciana M. L., Miano M., Tucci F., Casini T., Saracco P., Barcellini W., Zanella A., Perrotta S., Russo G., Baronci C., Casadei A. M., Cazzaniga G., Coluzzi S., Ciliberti A., Del Vecchio G. C., Facchini E., Girelli G., Lazzareschi I., Masera N., Perseghin P., Peruccio D., Petrone A., Sau A., Schiro R., Tumino M., Vasta I., and Verzegnassi F.

Published
2018

9. Lymphocyte-Predominant Hodgkin Lymphoma Variant: Long Term Outcome. Data From The Lh-2004 Protocol Of The Italian Association Of Pediatric Hematology And Oncology (Aieop)

Author

Mazzocco, M, additional, Mura, R, additional, De Santis, R, additional, Casini, T, additional, Porta, F, additional, Pierani, P, additional, Cellini, M, additional, Sau, A, additional, Verzegnassi, F, additional, Perruccio, K, additional, Cesaro, S, additional, Bertolini, P, additional, Pericoli, R, additional, Sperlì, D, additional, Mascarin, M, additional, and Burnelli, R, additional

Published
2020
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10. Second-line therapy in paediatric warm autoimmune haemolytic anaemia. Guidelines from the Associazione Italiana Onco-Ematologia Pediatrica (AIEOP)

Author

Ladogana S., Maruzzi M., Samperi P., Condorelli A., Casale M., Giordano P., Notarangelo L. D., Farruggia P., Giona F., Nocerino A., Fasoli S., Casciana M. L., Miano M., Tucci F., Casini T., Saracco P., Barcellini W., Zanella A., Perrotta S., Russo G., Baronci C., Casadei A. M., Cazzaniga G., Coluzzi S., Ciliberti A., Del Vecchio G. C., Facchini E., Girelli G., Lazzareschi I., Masera N., Perseghin P., Peruccio D., Petrone A., Sau A., Schiro R., Tumino M., Vasta I., Verzegnassi F., Ladogana, Saverio, Maruzzi, Matteo, Samperi, Piera, Condorelli, Annalisa, Casale, Maddalena, Giordano, Paola, Notarangelo, Lucia D., Farruggia, Piero, Giona, Fiorina, Nocerino, Agostino, Fasoli, Silvia, Casciana, Maria L., Miano, Maurizio, Tucci, Fabio, Casini, Tommaso, Saracco, Paola, Barcellini, Wilma, Zanella, Alberto, Perrotta, Silverio, Russo, Giovanna, Baronci, Carlo, Casadei, Anna Maria, Cazzaniga, Giovanni, Coluzzi, Serelina, Corti, Paola, Del Vecchio, Giovanni C., Facchini, Elena, Girelli, Gabriella, Lazzareschi, Ilaria, Masera, Nicoletta, Perseghin, Paolo, Peruccio, Daniela, Petrone, Angelamaria, Sau, Antonella, Schirò, Raffaella, Tumino, Manuela, Vasta, Isabella, Verzegnassi, Federico, Ladogana, S, Maruzzi, M, Samperi, P, Condorelli, A, Casale, M, Giordano, P, Notarangelo, L, Farruggia, P, Giona, F, Nocerino, A, Fasoli, S, Casciana, M, Miano, M, Tucci, F, Casini, T, Saracco, P, Barcellini, W, Zanella, A, Perrotta, S, Russo, G, Baronci, C, Casadei, A, Cazzaniga, G, Coluzzi, S, Ciliberti, A, Del Vecchio, G, Facchini, E, Girelli, G, Lazzareschi, I, Masera, N, Perseghin, P, Peruccio, D, Petrone, A, Sau, A, Schiro, R, Tumino, M, Vasta, I, and Verzegnassi, F

Subjects
Male, autoimmune haemolytic anaemia, child, therapy, rituximab, mycophenolate mofetil, splenectomy, therapy, Erythrocytes, Adolescent, Incidence, mycophenolate mofetil, Infant, Hematology, Guideline, splenectomy, rituximab, Settore MED/38 - PEDIATRIA GENERALE E SPECIALISTICA, Child, Preschool, Humans, Immunology and Allergy, autoimmune haemolytic anaemia, Female, Anemia, Hemolytic, Autoimmune, Child, Autoantibodies
Published
2018

11. Pillole di clavic... ultura

Author

Bevacqua M, Trombetta A, Verzegnassi F, Poropat F, BArbi E, Ventura A., Bevacqua, M, Trombetta, A, Verzegnassi, F, Poropat, F, Barbi, E, and Ventura, A.

Subjects
Chronic Recurrent Multifocal Osteomyelitis (CRMO), Clavicle, Tietze syndrome, Ewing sarcoma, Swelling
Abstract

The paper presents the cases of tree children with clavicular swelling and a different final diagnosis that offered the opportunity to discuss the key points for the differential diagnosis of clavicular swelling and to draw a rational diagnostic work-up underlining the decisive role of MRI. Although several causes (in order: Chronic Recurrent Multifocal Osteomyelitis, Ewing sarcoma, Tietze syndrome) of clavicular swelling are proposed, it is essential to highlight that main causes are represented by neoplastic and inflammatory causes. However, even if clinical and anamnestic assessment are suitable to disentangle from diagnostic incertitude, imaging is often useful to obtain a diagnosis, before (or even without) biopsy. It is anyway mandatory to rule out neoplasia in all non-traumatic clavicular swelling.

Published
2018

12. Retrospective And Prospective Study Of Childhood Autoimmune Hemolytic Anemia. A Preliminary Report From The Red Cell Working Group Of The Paediatric Hemato-Oncology Italian Associations (Aieop)

Author

Ladogana, S., Colombatti, R., Perrotta, S., Maggio, A., Maruzzi, M., Ciliberti, A., Samperi, P., Casale, M., Giordano, P., Del Vecchio, G. C., Perillo, T., Boscarol, G., Notarangelo, L. D., Casini, T., Miano, M., Fasoli, S., Corti, P., Guarina, A., Arcioni, F., Sau, A., Giona, F., Palumbo, G., Saracco, P., Petrone, A., Verzegnassi, F., Piccolo, C., and Russo, S. Cesaro and G.

Published
2019

13. Risultati preliminari degli Studi Prospettico e Retrospettivo (OEP 2015-01 e 2015-02) sull’Anemia Emolitica Autoimmune (AEA) del bambino di nuova diagnosi

Author

Ladogana, S., Colombatti, R., Perrotta, S., Maggio, A., Maruzzi, M., Ciliberti, A., Samperi, P., Casale, M., Giordano, P., Del Vecchio, G. C., Martire, B., Boscarol, G., Notarangelo, L. D., Casini, T., Miano, M., Fasoli, S., Corti, P., Masera, N., Guarina, A., Arcioni, F., Sau, A., Giona, F., Palumbo, G., Saracco, P., Petrone, A., Verzegnassi, F., Piccolo, C., Cesaro, S., and Russo, G.

Subjects
Anemia emolitica autoimmune, bambino, strategie terapeutiche
Published
2019

16. Late mortality and causes of death among 5-year survivors of childhood cancer diagnosed in the period 1960–1999 and registered in the Italian Off-Therapy Registry

Author

Bagnasco, F., Caruso, S., Andreano, A., Valsecchi, M. G., Jankovic, M., Biondi, A., Miligi, L., Casella, C., Terenziani, M., Massimino, M., Sacerdote, C., Morsellino, V., Erminio, G., Garaventa, A., Faraci, M., Micalizzi, C., Garre, M. L., Pillon, M., Basso, G., Biasin, E., Fagioli, F., Rondelli, R., Pession, A., Locatelli, Franco, Santoro, N., Indolfi, P., Palumbo, G., Russo, G., Verzegnassi, F., Favre, C., Zecca, M., Mura, R., D'Angelo, P., Cano, C., Byrne, J., Haupt, R., Pierani, P., Porta, F., Consarino, C., Burnelli, R., Fedeli, F., Cellini, M., Casale, F., Menna, G., Bertolini, P., Caniglia, M., Cecinati, V., Casazza, G., Foa, R., Clerico, A., Ladogana, S., Galimberti, D., Rabusin, M., Nespoli, L., Cesaro, S., Locatelli F. (ORCID:0000-0002-7976-3654), Bagnasco, F., Caruso, S., Andreano, A., Valsecchi, M. G., Jankovic, M., Biondi, A., Miligi, L., Casella, C., Terenziani, M., Massimino, M., Sacerdote, C., Morsellino, V., Erminio, G., Garaventa, A., Faraci, M., Micalizzi, C., Garre, M. L., Pillon, M., Basso, G., Biasin, E., Fagioli, F., Rondelli, R., Pession, A., Locatelli, Franco, Santoro, N., Indolfi, P., Palumbo, G., Russo, G., Verzegnassi, F., Favre, C., Zecca, M., Mura, R., D'Angelo, P., Cano, C., Byrne, J., Haupt, R., Pierani, P., Porta, F., Consarino, C., Burnelli, R., Fedeli, F., Cellini, M., Casale, F., Menna, G., Bertolini, P., Caniglia, M., Cecinati, V., Casazza, G., Foa, R., Clerico, A., Ladogana, S., Galimberti, D., Rabusin, M., Nespoli, L., Cesaro, S., and Locatelli F. (ORCID:0000-0002-7976-3654)

Abstract

Introduction: Advances in paediatric oncology led to the increase in long-term survival, revealing the burden of therapy-related long-term side effects. We evaluated overall and cause-specific mortality in a large cohort of Italian childhood cancer survivors (CCSs) and adolescent cancer survivors identified through the off-therapy registry. Materials and methods: CCSs alive 5 years after cancer diagnosis occurring between 1960 and 1999 were eligible; the last follow-up was between 2011 and 2014. Outcomes were reported as standardised mortality ratios (SMRs) and absolute excess risks (AERs). Results: Among 12,214 CCSs, 1113 (9.1%) deaths occurred. Survival at 35 years since diagnosis was 87% (95% confidence interval [CI]: 86–88) and at 45 years was 81% (95% CI: 77–84). CCSs had an 11-fold increased risk of death (SMR 95% CI: 10.7–12), corresponding to an AER of 48 (95% CI: 45–51). Mortality decreased by 60% for survivors treated most recently (1990–1999). The most frequent causes of death were recurrence of the original cancer (56%), a subsequent neoplasm (19%) and cardiovascular diseases (5.8%). Among those who survived at least 15 years after diagnosis, a secondary malignancy was the leading cause of death. Conclusions: This study confirms the impact of recent advances in anticancer therapy in reducing mortality, mainly attributable to recurrence but also to other causes. However, overall mortality continues to be higher than in the general population. A long-term follow-up is needed to prevent late mortality due to secondary neoplasms and non-neoplastic causes in CCSs.

Published
2019

17. Late mortality and causes of death among 5-year survivors of childhood cancer diagnosed in the period 1960–1999 and registered in the Italian Off-Therapy Registry

Author

Bagnasco, F, Caruso, S, Andreano, A, Valsecchi, M, Jankovic, M, Biondi, A, Miligi, L, Casella, C, Terenziani, M, Massimino, M, Sacerdote, C, Morsellino, V, Erminio, G, Garaventa, A, Faraci, M, Micalizzi, C, Garrè, M, Pillon, M, Basso, G, Biasin, E, Fagioli, F, Rondelli, R, Pession, A, Locatelli, F, Santoro, N, Indolfi, P, Palumbo, G, Russo, G, Verzegnassi, F, Favre, C, Zecca, M, Mura, R, D'Angelo, P, Cano, C, Byrne, J, Haupt, R, Pierani, P, Porta, F, Consarino, C, Burnelli, R, Fedeli, F, Cellini, M, Casale, F, Menna, G, Bertolini, P, Caniglia, M, Cecinati, V, Casazza, G, Foà, R, Clerico, A, Ladogana, S, Galimberti, D, Rabusin, M, Nespoli, L, Cesaro, S, Bagnasco, Francesca, Caruso, Silvia, Andreano, Anita, Valsecchi, Maria Grazia, Jankovic, Momcilo, Biondi, Andrea, Miligi, Lucia, Casella, Claudia, Terenziani, Monica, Massimino, Maura, Sacerdote, Carlotta, Morsellino, Vera, Erminio, Giovanni, Garaventa, Alberto, Faraci, Maura, Micalizzi, Concetta, Garrè, Maria Luisa, Pillon, Marta, Basso, Giuseppe, Biasin, Eleonora, Fagioli, Franca, Rondelli, Roberto, Pession, Andrea, Locatelli, Franco, Santoro, Nicola, Indolfi, Paolo, Palumbo, Giovanna, Russo, Giovanna, Verzegnassi, Federico, Favre, Claudio, Zecca, Marco, Mura, Rossella, D'Angelo, Paolo, Cano, Carmen, Byrne, Julianne, Haupt, Riccardo, Pierani, Paolo, Pession, Andreea, Porta, Fulvio, Consarino, Caterina, Burnelli, Roberta, Fedeli, Fausto, Cellini, Monica, Casale, Fiorina, Menna, Giuseppe, Bertolini, Patrizia, Caniglia, Maurizio, Cecinati, Valerio, Casazza, Gabriella, Foà, Roberto, Clerico, Anna, Ladogana, Saverio, Galimberti, Daniela, Rabusin, Marco, Nespoli, Luigi, Cesaro, Simone, Bagnasco, F, Caruso, S, Andreano, A, Valsecchi, M, Jankovic, M, Biondi, A, Miligi, L, Casella, C, Terenziani, M, Massimino, M, Sacerdote, C, Morsellino, V, Erminio, G, Garaventa, A, Faraci, M, Micalizzi, C, Garrè, M, Pillon, M, Basso, G, Biasin, E, Fagioli, F, Rondelli, R, Pession, A, Locatelli, F, Santoro, N, Indolfi, P, Palumbo, G, Russo, G, Verzegnassi, F, Favre, C, Zecca, M, Mura, R, D'Angelo, P, Cano, C, Byrne, J, Haupt, R, Pierani, P, Porta, F, Consarino, C, Burnelli, R, Fedeli, F, Cellini, M, Casale, F, Menna, G, Bertolini, P, Caniglia, M, Cecinati, V, Casazza, G, Foà, R, Clerico, A, Ladogana, S, Galimberti, D, Rabusin, M, Nespoli, L, Cesaro, S, Bagnasco, Francesca, Caruso, Silvia, Andreano, Anita, Valsecchi, Maria Grazia, Jankovic, Momcilo, Biondi, Andrea, Miligi, Lucia, Casella, Claudia, Terenziani, Monica, Massimino, Maura, Sacerdote, Carlotta, Morsellino, Vera, Erminio, Giovanni, Garaventa, Alberto, Faraci, Maura, Micalizzi, Concetta, Garrè, Maria Luisa, Pillon, Marta, Basso, Giuseppe, Biasin, Eleonora, Fagioli, Franca, Rondelli, Roberto, Pession, Andrea, Locatelli, Franco, Santoro, Nicola, Indolfi, Paolo, Palumbo, Giovanna, Russo, Giovanna, Verzegnassi, Federico, Favre, Claudio, Zecca, Marco, Mura, Rossella, D'Angelo, Paolo, Cano, Carmen, Byrne, Julianne, Haupt, Riccardo, Pierani, Paolo, Pession, Andreea, Porta, Fulvio, Consarino, Caterina, Burnelli, Roberta, Fedeli, Fausto, Cellini, Monica, Casale, Fiorina, Menna, Giuseppe, Bertolini, Patrizia, Caniglia, Maurizio, Cecinati, Valerio, Casazza, Gabriella, Foà, Roberto, Clerico, Anna, Ladogana, Saverio, Galimberti, Daniela, Rabusin, Marco, Nespoli, Luigi, and Cesaro, Simone

Abstract

Introduction: Advances in paediatric oncology led to the increase in long-term survival, revealing the burden of therapy-related long-term side effects. We evaluated overall and cause-specific mortality in a large cohort of Italian childhood cancer survivors (CCSs) and adolescent cancer survivors identified through the off-therapy registry. Materials and methods: CCSs alive 5 years after cancer diagnosis occurring between 1960 and 1999 were eligible; the last follow-up was between 2011 and 2014. Outcomes were reported as standardised mortality ratios (SMRs) and absolute excess risks (AERs). Results: Among 12,214 CCSs, 1113 (9.1%) deaths occurred. Survival at 35 years since diagnosis was 87% (95% confidence interval [CI]: 86–88) and at 45 years was 81% (95% CI: 77–84). CCSs had an 11-fold increased risk of death (SMR 95% CI: 10.7–12), corresponding to an AER of 48 (95% CI: 45–51). Mortality decreased by 60% for survivors treated most recently (1990–1999). The most frequent causes of death were recurrence of the original cancer (56%), a subsequent neoplasm (19%) and cardiovascular diseases (5.8%). Among those who survived at least 15 years after diagnosis, a secondary malignancy was the leading cause of death. Conclusions: This study confirms the impact of recent advances in anticancer therapy in reducing mortality, mainly attributable to recurrence but also to other causes. However, overall mortality continues to be higher than in the general population. A long-term follow-up is needed to prevent late mortality due to secondary neoplasms and non-neoplastic causes in CCSs.

Published
2019

18. Second-line therapy in paediatric warm autoimmune haemolytic anaemia. Guidelines from the Associazione Italiana Onco-Ematologia Pediatrica (AIEOP)

Author

Ladogana, S., Maruzzi, M., Samperi, P., Condorelli, A., Casale, M., Giordano, P., Notarangelo, L. D., Farruggia, P., Giona, F., Nocerino, A., Fasoli, S., Casciana, M. L., Miano, M., Tucci, F., Casini, T., Saracco, P., Barcellini, W., Zanella, A., Perrotta, S., Russo, G., Baronci, C., Casadei, A. M., Cazzaniga, G., Coluzzi, S., Ciliberti, A., Del Vecchio, G. C., Facchini, E., Girelli, G., Lazzareschi, I., Masera, N., Perseghin, P., Peruccio, D., Petrone, A., Sau, A., Schiro, R., Tumino, M., Vasta, I., Verzegnassi, F., Zanella A., Cazzaniga G., Ciliberti A., Facchini E., Lazzareschi I. (ORCID:0000-0001-7221-2983), Vasta I., Ladogana, S., Maruzzi, M., Samperi, P., Condorelli, A., Casale, M., Giordano, P., Notarangelo, L. D., Farruggia, P., Giona, F., Nocerino, A., Fasoli, S., Casciana, M. L., Miano, M., Tucci, F., Casini, T., Saracco, P., Barcellini, W., Zanella, A., Perrotta, S., Russo, G., Baronci, C., Casadei, A. M., Cazzaniga, G., Coluzzi, S., Ciliberti, A., Del Vecchio, G. C., Facchini, E., Girelli, G., Lazzareschi, I., Masera, N., Perseghin, P., Peruccio, D., Petrone, A., Sau, A., Schiro, R., Tumino, M., Vasta, I., Verzegnassi, F., Zanella A., Cazzaniga G., Ciliberti A., Facchini E., Lazzareschi I. (ORCID:0000-0001-7221-2983), and Vasta I.

Abstract

No abstract available

Published
2018

19. Autoimmune neutropenia of childhood secondary to other autoimmune disorders: Data from the Italian neutropenia registry

Author

Farruggia, P, Puccio, G, Fioredda, F, Lanza, T, Porretti, L, Ramenghi, U, Barone, A, Bonanomi, S, Finocchi, A, Ghilardi, R, Ladogana, S, Marra, N, Martire, B, Notarangelo, Ld, Onofrillo, D, Pillon, M, Russo, G, Lo Valvo, L, Serafinelli, J, Tucci, F, Zunica, F, Verzegnassi, F, and Dufour, C

Subjects
Male, Neutropenia, Infant, Newborn, Immunoglobulins, Intravenous, Immunoglobulins, Infant, Diseases, Infant, Premature, Diseases, Hematology, Newborn, Autoimmune Diseases, Child, Disease Susceptibility, Female, Humans, Immunosuppressive Agents, Infant, Premature, Italy, Prevalence, Registries, Settore MED/38, Intravenous, Premature
Published
2017

20. Rischio di recidiva nel linfoma di Hodgkin pediatrico: dalla esperienza AIEOP ad una strategia europea

Author

Mascarin, M., Bernasconi, S., Bertolini, P., Bianchi, M., Buffardi, S., Casale, F., Casini, T., Cellini, M., Cesaro, S., Civino, A., Consarino, C., Cosmi, C., D’Amico, S., De Santis, R., Facchini, E., Fagioli, F., Farruggia, P., Favre, C., Felici, L., Galimberti, D., Garaventa, A., Iaria, G., Indolfi, P., Locatelli, F., Moleti, M. L., Muggeo, P., Mura, R. M., Pericoli, R., Perruccio, K., Pierani, P., Pillon, M., Porta, F., Provenzi, M., Rinieri, S., Rondelli, R., Russo, G., Sala, A., Santoro, N., Sau, A., Sperli, D., Todesco, A., Tolva, A., Varasso, G., Verzegnassi, F., Vinti, L., Zecca, M., Lopci, E., Elia, C., Birri, S., Sabattini, E., D’Amore, E., and Burnelli, R.

Subjects
pediatria, linfoma hodgkin, risultati, linfoma hodgkin, pediatria, risultati
Published
2017

21. Diagnosis and management of newly diagnosed childhood autoimmune haemolytic anaemia. Recommendations from the Red Cell Study Group of the Paediatric Haemato-Oncology Italian Association

Author

Ladogana, S, Maruzzi, Maria Pia, Samperi, Pietro, Perrotta, S, Del Vecchio, Gc, Notarangelo, Ld, Farruggia, P, Verzegnassi, F, Masera, N, Saracco, P, Fasoli, S, Miano, M, Girelli, Gabriella, Barcellini, W, Zanella, A, Russo, G., Ladogana, Saverio, Maruzzi, Matteo, Samperi, Piera, Perrotta, Silverio, Del Vecchio, Giovanni C, Notarangelo, Lucia D, Farruggia, Piero, Verzegnassi, Federico, Masera, Nicoletta, Saracco, Paola, Fasoli, Silvia, Miano, Maurizio, Girelli, Gabriella, Barcellini, Wilma, Zanella, Alberto, and Russo, Giovanna

Subjects
child, therapy, diagnosis, autoimmune haemolytic anaemia, child, DAT, diagnosis, therapy, Disease Management, Hematology, DAT, Guideline, Pediatrics, diagnosis therapy, Coombs Test, Immunoglobulin M, Italy, Humans, Blood Transfusion, Steroids, autoimmune haemolytic anaemia, Anemia, Hemolytic, Autoimmune, autoimmune haemolytic anaemia, child, DAT, diagnosis therapy, Societies, Medical
Abstract

Autoimmune haemolytic anaemia is an uncommon disorder to which paediatric haematology centres take a variety of diagnostic and therapeutic approaches. The Red Cell Working Group of the Italian Association of Paediatric Onco-haematology (Associazione Italiana di Ematologia ed Oncologia Pediatrica, AIEOP) developed this document in order to collate expert opinions on the management of newly diagnosed childhood autoimmune haemolytic anaemia.The diagnostic process includes the direct and indirect antiglobulin tests; recommendations are given regarding further diagnostic tests, specifically in the cases that the direct and indirect antiglobulin tests are negative. Clear-cut definitions of clinical response are stated. Specific recommendations for treatment include: dosage of steroid therapy and tapering modality for warm autoimmune haemolytic anaemia; the choice of rituximab as first-line therapy for the rare primary transfusion-dependent cold autoimmune haemolytic anaemia; the indications for supportive therapy; the need for switching to second-line therapy. Each statement is provided with a score expressing the level of appropriateness and the agreement among participants.

Published
2017

22. Studio Multicentrico prospettico osserva zionale sui sintomi muscolo scheletrici all’esordio in oncologia pediatrica e i fattori predittivi nella diagnosi differenziale con l’atrite idiopatica giovanile. Analisi preliminare

Author

Civino, A., Alighieri, G., Davi, S., Pession, A., Ravelli, A., Santoro, N., Belotti, T, Martino, S., Cesaro, Simone, Filocamo, G., Marino, S., Magnolato, A., Colombini, A., Ricci, F., Suffia, C., Galizzi, R., Palmisani, E., Verzegnassi, F., Olivieri, A. N., Tirtei, E., Gerloni, V, Gorio, C., Lattanzi, B., Pizzati, C., Soscia, F., Vinti, L., De Fanti, A., Ficara, M., Magni Manzoni, S., Boaro, M. P., Prete, E., Quartulli, L., La Torre, F., Onofrillo, D., Rigante, D., Capolsini, I., Maggio, C., Ladogana, S., Marsali, M., Burnelli, R., Coassin, E., Pelegatti, M. A., Arlotta, A., Lepore, L., Conter, V., Biondi, A., Fagioli, F., and Rondelli, R.

Subjects
diagnosi, leucemia acuta, sintomi, diagnosi, sintomi, leucemia acuta
Published
2016

23. Antiretroviral use in Italian children with perinatal HIV infection over a 14-year period

Author

Chiappini E, Galli L, Tovo PA, Gabiano C, Lisi C, Giacomet V, Bernardi S, Esposito S, Rosso R, Giaquinto C, Badolato R, GUARINO, ALFREDO, Maccabruni A, Masi M, Cellini M, Salvini F, Di Bari C, Dedoni M, Dodi I, de Martino M, Osimani P, Cordiali R., Larovere D, De Serio G, Giannini AM, Quercia M., Ruggeri M., Miniaci A, Specchia F, Ciccia M, Faldella G, Baldi F, Lanari M. Ciccia M, Bertulli C, Sorlini A, Ricci F, Dessy C, Pintor M, Fenu ML, Cavallini R, Aliffi A, Anastasio E, Aloe M., Abbagnato L., Merlino S, Fiumana E, Burnelli R, Bonsignori F, Gervaso P, Sollai S., Viscoli C, Cosso D, Timitilli A, Amoretti M., Vigano` A, Zuccotti GV, Mameli C, Fabiano V, Coletto S, Nello F, Riva E, Bettiga C, Picciolli, Irene, Preti Valentina, Tagliaferri Laura, Lipreri R, Mancini L., Mariotti I, Manzotti E, Giubbarelli F., GIANNATTASIO, ANTONIETTA, LO VECCHIO, ANDREA, BRUZZESE, EUGENIA, Tarallo L, Buffolano W., Pennazzato M, Rampon O., Dalle Nogare ER, Sanfilippo A, Romano A, Saitta M, Bandello MA, Tchana I, Maccabruni A., Felici L., Verrotti M., Consolini R, Palla G, Magnani C., Palma P, Pontrelli G, Tchidjou K. H, Genovese O, Falconieri P, Casadei M, Valentini P, Casadei, Martino, Anzidei, Cerilli, Catania Ajissa, Castelli Gattinara G., Cristiano R, Labalestra G, Portelli V., Mazza A, Chiarello P, Garazzino S, Mignone F, Calitri C., Rabusin M, Verzegnassi F., Pellegatta A., Boscardini L, Fortunati P, Da Riol R., Chiappini, E, Galli, L, Tovo, Pa, Gabiano, C, Lisi, C, Giacomet, V, Bernardi, S, Esposito, S, Rosso, R, Giaquinto, C, Badolato, R, Guarino, Alfredo, Maccabruni, A, Masi, M, Cellini, M, Salvini, F, Di Bari, C, Dedoni, M, Dodi, I, de Martino, M, Osimani, P, Cordiali, R., Larovere, D, De Serio, G, Giannini, Am, Quercia, M., Ruggeri, M., Miniaci, A, Specchia, F, Ciccia, M, Faldella, G, Baldi, F, Lanari M., Ciccia M, Bertulli, C, Sorlini, A, Ricci, F, Dessy, C, Pintor, M, Fenu, Ml, Cavallini, R, Aliffi, A, Anastasio, E, Aloe, M., Abbagnato, L., Merlino, S, Fiumana, E, Burnelli, R, Bonsignori, F, Gervaso, P, Sollai, S., Viscoli, C, Cosso, D, Timitilli, A, Amoretti, M., Vigano`, A, Zuccotti, Gv, Mameli, C, Fabiano, V, Coletto, S, Nello, F, Riva, E, Bettiga, C, Picciolli, Irene, Preti, Valentina, Tagliaferri, Laura, Lipreri, R, Mancini, L., Mariotti, I, Manzotti, E, Giubbarelli, F., Giannattasio, Antonietta, LO VECCHIO, Andrea, Bruzzese, Eugenia, Tarallo, L, Buffolano, W., Pennazzato, M, Rampon, O., Dalle Nogare, Er, Sanfilippo, A, Romano, A, Saitta, M, Bandello, Ma, Tchana, I, Maccabruni, A., Felici, L., Verrotti, M., Consolini, R, Palla, G, Magnani, C., Palma, P, Pontrelli, G, Tchidjou, K. H., Genovese, O, Falconieri, P, Casadei, M, Valentini, P, Casadei, Martino, Anzidei, Cerilli, Catania, Ajissa, Castelli Gattinara, G., Cristiano, R, Labalestra, G, Portelli, V., Mazza, A, Chiarello, P, Garazzino, S, Mignone, F, Calitri, C., Rabusin, M, Verzegnassi, F., Pellegatta, A., Boscardini, L, Fortunati, P, and Da Riol, R.

Abstract

BACKGROUND: Information on the use of new antiretroviral drugs in children in the real setting of clinical fields is largely unknown. METHODS: Data from 2554 combined antiretroviral therapy (cART) regimens administered to 911 children enrolled in the Italian Register for HIV infection in children, between 1996 and 2009, were analysed. Factors potentially associated with undetectable viral load and immunological response to cART were explored by Cox regression analysis. RESULTS: Proportion of protease inhibitor (PI)-based regimens significantly decreased from 88.0% to 51.2% and 54.9%, while proportion on non-nucleoside reverse transcriptase inhibitor (NNRTI)-based regimens increased from 4.5% to 38.8% and 40.2% in 1996-1999, 2000-2004 and 2005-2009, respectively (p < 0.0001). Significant change in the use of each antiretroviral drug occurred over the time periods (p < 0.0001). Factors independently associated with virological and immunological success were as follows: later calendar periods, younger age at regimen (only for virological success) and higher CD4(+) T-lymphocyte percentage at baseline. Use of unboosted PI was associated with lower adjusted hazard ratio (aHR) of virological or immunological success with respect to NNRTI- and boosted PI-based regimens, with no difference among these two latter types. CONCLUSION: Use of new generation antiretroviral drugs in Italian HIV-infected children is increasing. No different viro-immunological outcomes between NNRTI- and boosted PI-based cART were observed

Published
2012

26. Aplasie midollari acquisite in età pediatrica: Raccomandazioni diagnostico-terapeutiche [Acquired aplastic anaemia in children: Diagnostic-therapeutic recommendations]

Author

Barone, A, Lucarelli, A, Onofrillo, D, Verzegnassi, F, Bonanomi, S, Cesaro, S, Cugno, C, Fioredda, F, Iori, Ap, Ladogana, S, Locasciulli, A, Longoni, D, Lanciotti, M, Macaluso, A, Mandaglio, R, Marra, N, Martire, B, Maruzzi, M, Menna, G, Notarangelo, Ld, Palazzi, G, Pillon, M, Ramenghi, U, Russo, Giovanna, Svahn, J, Timeus, F, Tucci, F, Zecca, M, Farruggia, P, Dufour, C, and Saracco, P.

Published
2014

27. Acquired aplastic anaemia in children: Diagnostic-therapeutic recommendations

Author

Barone, A, Lucarelli, A, Onofrillo, D, Verzegnassi, F, Bonanomi, S, Longoni, D, Cesaro, S, Cugno, C, Zecca, M, Fioredda, F, Svahn, J, Dufour, C, Iori, Ap, Ladogana, S, Maruzzi, M, Locasciulli, A, Lanciotti, M, Macaluso, A, Mandaglio, R, Marra, N, Menna, G, Martire, B, Notarangelo, Ld, Palazzi, G, Pillon, M, Ramenghi, Ugo, Saracco, P, Russo, G, Timeus, F, Tucci, F, and Farruggia, P.

Subjects
Acquired aplastic anemia, Childhood
Published
2014

30. DENTAL PHENOTYPE IN A PATIENT WITH HYPOIDROTIC ECTODERMAL DYSPLASIA AND SEVERE IMMUNODEFICIENCY.

Author

Callea, M., Faletra, F., Maestro, A., Verzegnassi, F., Rabusin, M., Vinciguerra, A., Radovich, F., Clarich, G., Yavuz, I., and Tumen, E. C.

Subjects
ECTODERMAL dysplasia, IMMUNODEFICIENCY, DENTITION, DENTAL occlusion
Abstract

Ectodermal dysplasia is a rare disease which affects at least two ectoderm-derived structures such as hair, nails, skin, sweat glands and teeth. The dentition is altered in number and shape. A 14-year-old male patient with hypodontia, micrognathia, ankylosed teeth and conical shaped teeth was referred for examination, evaluation and treatment. The child exhibited the classic dental phenotype of Ectodermal Dysplasia plus a severe immunodeficiency. Radiographic examination revealed ankylosed primary molars. Ocular findings are reported. Conservative dentistry to reduce the abnormal shape was carried out , and an ultrasound scaling every 4 months, with a strong follow up established. The child fulfilled a good occlusion. Every 3 months the patient has been seen in our department for control of hard and soft tissue in the mouth and after 36 months the dental situation is very well accomplished by patient, family and dental staff. Oral rehabilitation must be carried out at the earliest age possible in order to maintain and correct the oral functions, alignment, good occlusion and a good compliance in smiling and feeding. [ABSTRACT FROM AUTHOR]

Published
2011

33. Il registro italiano dei fuori terapia (ROT).

Author

Haupt, R., Bagnasco, F., Caruso, S., Silvestri, D., Rossi, E., Valsecchi, M. G., Jankovic, M., Fraschini, D., Faraci, M., Fioredda, F., Hanau, G., Polastri, D., Pillon, M., Varotto, S., Rondelli, R., Cano, C., Verzegnassi, F., Puma, M., Mele, D., and Biondi, L.

Published
2012

35. Acquired aplastic anaemia in children: Diagnostic-therapeutic recommendations | Aplasie midollari acquisite in età pediatrica: Raccomandazioni diagnostico-terapeutiche

Author

Barone, A., Lucarelli, A., Onofrillo, D., Verzegnassi, F., Bonanomi, S., Longoni, D., Cesaro, S., Cugno, C., Zecca, M., Fioredda, F., Svahn, J., Dufour, C., Iori, A. P., Ladogana, S., Maruzzi, M., Locasciulli, A., Lanciotti, M., Macaluso, A., Mandaglio, R., Marra, N., Menna, G., Martire, B., Notarangelo, L. D., Palazzi, G., Pillon, M., Ramenghi, U., Saracco, P., Giovanna Russo, Timeus, F., Tucci, F., and Farruggia, P.

36. The Italian Registry of the Off-Therapy (ROT) | Il registro Italiano dei Fuori terapia (ROT)

Author

Haupt, R., Bagnasco, F., Caruso, S., Silvestri, D., Emanuela Rossi, Valsecchi, M. G., Jankovic, M., Fraschini, D., Faraci, M., Fioredda, F., Hanau, G., Polastri, D., Pillon, M., Varotto, S., Rondelli, R., Cano, C., Verzegnassi, F., Puma, M., Mele, D., Biondi, L., Robustelli, G., Tonioli, C., Palumbo, G., Baronci, C., Ilari, I., La Spina, M., Parasole, R., Santoro, N., Mura, R. M., Bertin, D., Comotti, C., Rosa, E., Tropia, S., Grigoli, A., and Biondi, A.

39. Acquired aplastic anaemia in children: Diagnostic-therapeutic recommendations,Aplasie midollari acquisite in età pediatrica: Raccomandazioni diagnostico-terapeutiche

Author

Barone, A., Lucarelli, A., Onofrillo, D., Verzegnassi, F., Bonanomi, S., Longoni, D., Cesaro, S., Cugno, C., Zecca, M., Fioredda, F., Johanna Svahn, Dufour, C., Iori, A. P., Ladogana, S., Maruzzi, M., Locasciulli, A., Lanciotti, M., Macaluso, A., Mandaglio, R., Marra, N., Menna, G., Martire, B., Notarangelo, L. D., Palazzi, G., Pillon, M., Ramenghi, U., Saracco, P., Russo, G., Timeus, F., Tucci, F., and Farruggia, P.

40. The Italian Registry of the Off-Therapy (ROT),Il registro Italiano dei Fuori terapia (ROT)

Author

Haupt, R., Francesca Bagnasco, Caruso, S., Silvestri, D., Rossi, E., Valsecchi, M. G., Jankovic, M., Fraschini, D., Faraci, M., Fioredda, F., Hanau, G., Polastri, D., Pillon, M., Varotto, S., Rondelli, R., Cano, C., Verzegnassi, F., Puma, M., Mele, D., Biondi, L., Robustelli, G., Tonioli, C., Palumbo, G., Baronci, C., Ilari, I., La Spina, M., Parasole, R., Santoro, N., Mura, R. M., Bertin, D., Comotti, C., Rosa, E., Tropia, S., Grigoli, A., and Biondi, A.

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

Author

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

Subjects
medicine.medical_specialty, business.industry, Immunology, Arthritis, Cancer, Odds ratio, Musculoskeletal manifestation, Juvenile idiopathic arthritis, medicine.disease, Histiocytosis, Rheumatology, Settore MED/38 - PEDIATRIA GENERALE E SPECIALISTICA, Prednisone, Internal medicine, Joint pain, Arthropathy, Musculoskeletal manifestations, childhood cancer, juvenile idiopathic arthritis, medicine, childhood cancer, Immunology and Allergy, Differential diagnosis, medicine.symptom, business, medicine.drug
Abstract

Summary 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 by patients with Langerhans histiocytosis (16 [47%] of 34), leukaemia (189 [32%] of 582), soft-tissue sarcomas (16 [24%] of 68), and neuroblastoma (21 [19%] of 109). In the 324 patients with cancer and musculoskeletal symptoms, the most common complaints were joint pain (199 [61%]), followed by limb bone pain (112 [35%]). Joint involvement had a prevalent monoarticular pattern (100 [48%] of 207) and oligoarticular pattern (86 [42%] had 2–4 joints involved and 20 [10%] had >4 joints involved), with the most frequently involved joints being the hip (88 [43%] of 207) and knee (81 [39%]). On multivariable analysis, limb bone pain was the independent variable most strongly associated with cancer (odds ratio [OR] 87·80 [95% CI 18·89–408·12]), followed by weight loss (59·88 [6·34–565·53]), thrombocytopenia (12·67 [2·40–66·92]), monoarticular involvement (11·30 [4·09–31·19]), hip involvement (3·30 [1·13–9·61]), and male sex (2·40 [1·03–5·58]). Factors independently associated with juvenile idiopathic arthritis were morning stiffness (OR 0·04 [95% CI 0·01–0·20]), joint swelling (0·03 [0·01–0·09]), and involvement of the small hand joints (0·02 [0–1·05]). Interpretation Our study provides detailed information about presenting musculoskeletal manifestations of childhood cancers and highlights the clinical and laboratory features that are most helpful in the differential diagnosis with juvenile idiopathic arthritis. Funding Associazione Lorenzo Risolo.

Published
2021

42. Rituximab unveils hypogammaglobulinemia and immunodeficiency in children with autoimmune cytopenia

Author

Viviana Moschese, Giuseppe Lassandro, Caterina Cancrini, Paola Saracco, Keith Sibson, Claudio Pignata, Antonino Trizzino, Maurizio Miano, Carmela Giancotta, Maria Gabelli, Cecilia Bava, Federica Barzaghi, Simona Graziani, Rosa Angarano, Andrea Biondi, Samuele Naviglio, Baldassare Martire, Federico Verzegnassi, Patrizia Bertolini, Maddalena Marinoni, Paola Giordano, Giorgio Ottaviano, Elena Mastrodicasa, Loredana Chini, Vera Gallo, Irene D'Alba, Paola Corti, Ottaviano, Gianmarco, Marinoni, M., Graziani, S., Sibson, K., Barzaghi, F., Bertolini, P., Chini, L., Corti, P., Cancrini, C., D'Alba, I., Gabelli, M., Gallo, V., Giancotta, C., Giordano, P., Lassandro, G., Martire, B., Angarano, R., Mastrodicasa, E., Bava, Anna, Miano, M., Naviglio, S., Verzegnassi, F., Saracco, P., Trizzino, A., Biondi, A., Pignata, C., Moschese, V., Ottaviano, G, Marinoni, M, Graziani, S, Sibson, K, Barzaghi, F, Bertolini, P, Chini, L, Corti, P, Cancrini, C, D'Alba, I, Gabelli, M, Gallo, V, Giancotta, C, Giordano, P, Lassandro, G, Martire, B, Angarano, R, Mastrodicasa, E, Bava, C, Miano, M, Naviglio, S, Verzegnassi, F, Saracco, P, Trizzino, A, Biondi, A, Pignata, C, and Moschese, V

Subjects
medicine.medical_specialty, Evans syndrome, Hypogammaglobulinemia, Autoimmunity, Gastroenterology, 03 medical and health sciences, 0302 clinical medicine, Agammaglobulinemia, hemic and lymphatic diseases, Internal medicine, medicine, Immunology and Allergy, Humans, 030212 general & internal medicine, Child, Immunodeficiency, Settore MED/38 - Pediatria Generale e Specialistica, Cytopenia, Purpura, Thrombocytopenic, Idiopathic, Primary immunodeficiency, business.industry, Autoimmune Cytopenia, medicine.disease, Thrombocytopenia, Rituximab, Treatment Outcome, 030228 respiratory system, Anemia, Hemolytic, Autoimmune, Autoimmune hemolytic anemia, business, medicine.drug
Abstract

Background: Rituximab (RTX; anti-CD20 mAb) is a treatment option in children with refractory immune thrombocytopenia, autoimmune hemolytic anemia (AHA), and Evans syndrome (ES). Prevalence and clinical course of RTX-induced hypogammaglobulinemia in these patients are poorly known. Objective: To evaluate the prevalence and risk factors for persistent hypogammaglobulinemia (PH) after RTX use. Methods: Clinical and immunologic data from children treated with RTX for immune thrombocytopenia, AHA, and ES were collected from 16 Italian centers and 1 UK center at pre-RTX time point (0), +6 months, and yearly, up to 4 years post-RTX. Patients with previously diagnosed malignancy or primary immune deficiency (PID) were excluded. Results: We analyzed 53 children treated with RTX for immune thrombocytopenia (n = 36), AHA (n = 13), and ES (n = 4). Median follow-up was 30 months (range, 12-48). Thirty-two percent of patients (17 of 53) experienced PH, defined as IgG levels less than 2 SD for age at last follow-up (>12 months after RTX). Significantly delayed B-cell recovery was observed in children experiencing PH (hazard ratio, 0.55; P

Published
2020

43. Multicenter randomized, double-blind controlled trial to evaluate the efficacy of laser therapy for the treatment of severe oral mucositis induced by chemotherapy in children: laMPO RCT

Author

Maria Livia Mariuzzi, Alessandra Piras, Federico Verzegnassi, Elena Bardellini, Maria Grazia Petris, Patrizia Defabianis, Simone Bagattoni, Margherita Gobbo, Elisabetta Merigo, Fraia Melchionda, Davide Zanon, Matteo Biasotto, Alessandra Majorana, Giulio Andrea Zanazzo, Massimo Berger, Angelica Barone, Nunzia Decembrino, Giulia Ottaviani, Marina Consuelo Vitale, Luca Ronfani, Rosamaria Mura, Gobbo M., Verzegnassi F., Ronfani L., Zanon D., Melchionda F., Bagattoni S., Majorana A., Bardellini E., Mura R., Piras A., Petris M.G., Mariuzzi M.L., Barone A., Merigo E., Decembrino N., Vitale M.C., Berger M., Defabianis P., Biasotto M., Ottaviani G., Zanazzo G.A., Gobbo, M., Verzegnassi, F., Ronfani, L., Zanon, D., Melchionda, F., Bagattoni, S., Majorana, A., Bardellini, E., Mura, R., Piras, A., Petris, M. G., Mariuzzi, M. L., Barone, A., Merigo, E., Decembrino, N., Vitale, M. C., Berger, M., Defabianis, P., Biasotto, M., Ottaviani, G., and Zanazzo, G. A.

Subjects
Male, genetic structures, medicine.medical_treatment, Clinical trial, Laser, Mucositis, Pediatric hemato-oncology, Supportive care, Pediatrics, Perinatology and Child Health, Hematology, Oncology, Pediatrics, law.invention, Antineoplastic Agent, 0302 clinical medicine, Randomized controlled trial, law, Neoplasms, Child, Stomatitis, pediatric hemato-oncology, clinical trial, Perinatology and Child Health, Treatment Outcome, 030220 oncology & carcinogenesis, Female, Human, medicine.medical_specialty, Adolescent, Analgesic, chemical and pharmacologic phenomena, Antineoplastic Agents, macromolecular substances, Placebo, 03 medical and health sciences, Double-Blind Method, Internal medicine, medicine, Humans, Low-Level Light Therapy, Adverse effect, laser, mucositis, supportive care, Chemotherapy, business.industry, mucositi, fungi, 030206 dentistry, medicine.disease, Stomatiti, Neoplasm, business
Abstract

Objectives: To demonstrate the efficacy of laser photobiomodulation (PBM) compared to that of placebo on severe oral mucositis (OM) in pediatric oncology patients. The primary objective was the reduction of OM grade (World Health Organization [WHO] scale) 7 days after starting PBM. Secondary objectives were reduction of pain, analgesic consumption, and incidence of side effects. Methods: One hundred and one children with WHO grade>2 chemotherapy-induced OM were enrolled in eight Italian hospitals. Patients were randomized to either PBM or sham treatment for four consecutive days (days +1 to +4). On days +4, +7, and +11, OM grade, pain (following a 0–10 numeric pain rating scale, NRS) and need for analgesics were evaluated by an operator blinded to treatment. Results: Fifty-one patients were allocated to the PBM group, and 50 were allocated to the sham group. In total, 93.7% of PBM patients and 72% of sham patients had OM grade&nbsp

Published
2017

44. IL-1 blockade with anakinra for severe inflammatory symptoms during chemotherapy for acute lymphoblastic leukemia

Author

Maria Luisa Coniglio, Samuele Naviglio, Marco Rabusin, Alberto Tommasini, Elena Sieni, Erica Valencic, Valentina Kiren, Federico Verzegnassi, Nagua Giurici, Giada Zanella, Naviglio, S., Zanella, G., Verzegnassi, F., Kiren, V., Giurici, N., Sieni, E., Coniglio, M. L., Valencic, E., Tommasini, A., and Rabusin, M.

Subjects
Oncology, Anakinra, medicine.medical_specialty, Chemotherapy, business.industry, Il-1, Lymphoblastic Leukemia, medicine.medical_treatment, Hematology, acute lymphoblastic leukemia, Precursor Cell Lymphoblastic Leukemia-Lymphoma, Blockade, Interleukin 1 Receptor Antagonist Protein, Internal medicine, Pediatrics, Perinatology and Child Health, Acute Disease, medicine, Humans, business, medicine.drug, Interleukin-1
Abstract

Anakinra is a recombinant interleukin (IL)-1 receptor antagonistusedforseveralinflammatoryconditionsincludingsystemicjuvenileidiopathicarthritis,recurrentpericarditis,andfamilialMediterraneanfever (FMF). Its efficacy in blocking IL-1 and its manageable safetyprofilesupportitsusealsoinotherconditions,suchassepsis,acutemyocardialinfarction,andCOVID-19

Published
2022

45. Late mortality and causes of death among 5-year survivors of childhood cancer diagnosed in the period 1960–1999 and registered in the Italian Off-Therapy Registry

Author

Francesca Bagnasco, Silvia Caruso, Anita Andreano, Maria Grazia Valsecchi, Momcilo Jankovic, Andrea Biondi, Lucia Miligi, Claudia Casella, Monica Terenziani, Maura Massimino, Carlotta Sacerdote, Vera Morsellino, Giovanni Erminio, Alberto Garaventa, Maura Faraci, Concetta Micalizzi, Maria Luisa Garrè, Marta Pillon, Giuseppe Basso, Eleonora Biasin, Franca Fagioli, Roberto Rondelli, Andrea Pession, Franco Locatelli, Nicola Santoro, Paolo Indolfi, Giovanna Palumbo, Giovanna Russo, Federico Verzegnassi, Claudio Favre, Marco Zecca, Rossella Mura, Paolo D'Angelo, Carmen Cano, Julianne Byrne, Riccardo Haupt, Paolo Pierani, Andreea Pession, Fulvio Porta, Caterina Consarino, Roberta Burnelli, Fausto Fedeli, Monica Cellini, Fiorina Casale, Giuseppe Menna, Patrizia Bertolini, Maurizio Caniglia, Valerio Cecinati, Gabriella Casazza, Roberto Foà, Anna Clerico, Saverio Ladogana, Daniela Galimberti, Marco Rabusin, Luigi Nespoli, Simone Cesaro, Bagnasco, F, Caruso, S, Andreano, A, Valsecchi, M, Jankovic, M, Biondi, A, Miligi, L, Casella, C, Terenziani, M, Massimino, M, Sacerdote, C, Morsellino, V, Erminio, G, Garaventa, A, Faraci, M, Micalizzi, C, Garrè, M, Pillon, M, Basso, G, Biasin, E, Fagioli, F, Rondelli, R, Pession, A, Locatelli, F, Santoro, N, Indolfi, P, Palumbo, G, Russo, G, Verzegnassi, F, Favre, C, Zecca, M, Mura, R, D'Angelo, P, Cano, C, Byrne, J, Haupt, R, Pierani, P, Porta, F, Consarino, C, Burnelli, R, Fedeli, F, Cellini, M, Casale, F, Menna, G, Bertolini, P, Caniglia, M, Cecinati, V, Casazza, G, Foà, R, Clerico, A, Ladogana, S, Galimberti, D, Rabusin, M, Nespoli, L, and Cesaro, S

Subjects
0301 basic medicine, Male, Pediatrics, Cancer Research, Cardiotoxicity, Causes of death, Childhood cancer, Childhood cancer long-term survivors, Late mortality, Second malignant neoplasms, Oncology, 0302 clinical medicine, Cancer Survivors, Cause of Death, Neoplasms, Second malignant neoplasm, Prospective Studies, Registries, Age of Onset, Child, Cause of death, education.field_of_study, Paediatric oncology, Italy, Settore MED/38 - PEDIATRIA GENERALE E SPECIALISTICA, 030220 oncology & carcinogenesis, Child, Preschool, Female, Adult, medicine.medical_specialty, Adolescent, Population, 03 medical and health sciences, Young Adult, Age Distribution, medicine, Humans, education, business.industry, Infant, Newborn, Secondary Malignancy, Cancer, Infant, medicine.disease, Confidence interval, 030104 developmental biology, Increased risk, Childhood cancer long-term survivor, business
Abstract

Introduction Advances in paediatric oncology led to the increase in long-term survival, revealing the burden of therapy-related long-term side effects. We evaluated overall and cause-specific mortality in a large cohort of Italian childhood cancer survivors (CCSs) and adolescent cancer survivors identified through the off-therapy registry. Materials and methods CCSs alive 5 years after cancer diagnosis occurring between 1960 and 1999 were eligible; the last follow-up was between 2011 and 2014. Outcomes were reported as standardised mortality ratios (SMRs) and absolute excess risks (AERs). Results Among 12,214 CCSs, 1113 (9.1%) deaths occurred. Survival at 35 years since diagnosis was 87% (95% confidence interval [CI]: 86–88) and at 45 years was 81% (95% CI: 77–84). CCSs had an 11-fold increased risk of death (SMR 95% CI: 10.7–12), corresponding to an AER of 48 (95% CI: 45–51). Mortality decreased by 60% for survivors treated most recently (1990–1999). The most frequent causes of death were recurrence of the original cancer (56%), a subsequent neoplasm (19%) and cardiovascular diseases (5.8%). Among those who survived at least 15 years after diagnosis, a secondary malignancy was the leading cause of death. Conclusions This study confirms the impact of recent advances in anticancer therapy in reducing mortality, mainly attributable to recurrence but also to other causes. However, overall mortality continues to be higher than in the general population. A long-term follow-up is needed to prevent late mortality due to secondary neoplasms and non-neoplastic causes in CCSs.

Published
2019

46. The Challenge of Next Generation Sequencing in a Boy With Severe Mononucleosis and EBV-related Lymphoma

Author

Nagua Giurici, Annalisa Marcuzzi, Alberto Tommasini, Flavio Faletra, Federico Verzegnassi, Diego Vozzi, Valentina Kiren, Anna Monica Bianco, Erica Valencic, Verzegnassi, F., Valencic, E., Kiren, V., Giurici, N., Bianco, A. M., Marcuzzi, A., Vozzi, D., Tommasini, A., and Faletra, F.

Subjects
0301 basic medicine, Male, Herpesvirus 4, Human, Mononucleosis, Adolescent, Lymphoma, Infectious Mononucleosi, medicine.disease_cause, NO, whole exome sequencing, CTPS2, 03 medical and health sciences, 0302 clinical medicine, Epstein Barr virus, hemophagocytic lymphohistiocytosis, lymphoma, Humans, Exome, High-Throughput Nucleotide Sequencing, Infectious Mononucleosis, Mutation, hemophagocytic lymphohistiocytosi, medicine, Exome sequencing, Hemophagocytic lymphohistiocytosis, business.industry, Herpesvirus 4, Hematology, medicine.disease, Penetrance, Epstein–Barr virus, 030104 developmental biology, Oncology, Epstein Barr viru, Pediatrics, Perinatology and Child Health, Immunology, Primary immunodeficiency, business, 030215 immunology, Human
Abstract

A severe course of infectious mononucleosis should always lead up to the suspicion of a primary immunodeficiency. We describe the case of a boy with severe mononucleosis accompanied by the development of hemophagocytic lymphohistiocytosis and lymphoma. By whole exome sequencing, we identified a mutation of uncertain significance in CTPS2, a gene closely related to CTPS1, which is involved in a primary immune deficiency with susceptibility to herpesviruses. We discuss the challenge of a correct interpretation of data from whole exome sequencing, questioning whether the CTPS2 variant found in our patient is just an incidental finding or a mutation with variable penetrance.

Published
2017

47. Molecular analysis of Fanconi anemia: the experience of the Bone Marrow Failure Study Group of the Italian Association of Pediatric Onco-Hematology

Author

Fabio Corsolini, Roberta Bottega, Federico Verzegnassi, Adriana Borriello, Elena Nicchia, Silverio Perrotta, Simona Cavani, Marta Pillon, Paola Grammatico, Johanna Svahn, Fulvio Della Ragione, Walter Barberi, Chiara Greco, Anna Locasciulli, Maria Criscuolo, Enrico Cappelli, Ugo Ramenghi, Piero Farruggia, Gabriella Casazza, Daniela Longoni, Fabio Tucci, Chiara Cugno, Daniela De Rocco, Cristina Mecucci, Anna Savoia, Helmut Hanenberg, Carlo Dufour, De Rocco, D, Bottega, R, Cappelli, E, Cavani, S, Criscuolo, M, Nicchia, E, Corsolini, F, Greco, C, Borriello, Adriana, Svahn, J, Pillon, M, Mecucci, C, Casazza, G, Verzegnassi, F, Cugno, C, Locasciulli, A, Farruggia, P, Longoni, D, Ramenghi, U, Barberi, W, Tucci, F, Perrotta, Silverio, Grammatico, P, Hanenberg, H, DELLA RAGIONE, Fulvio, Dufour, C, Savoia, A, Bone Marrow Failure Study Group of the Italian Association of Pediatric Onco, Hematology, DE ROCCO, Daniela, Bottega, Roberta, Nicchia, Elena, Borriello, A, Perrotta, S, Della Ragione, F, and Savoia, Anna

Subjects
Candidate gene, gene amplification, genotype, cytogenetics and molecular genetics, genetic analysis, Bioinformatics, Western blotting, hematopoietic stem cell, bone marrow failure, fanconi anemia, Cohort Studies, genetic heterogeneity, single nucleotide polymorphism, FANCG, Fanconi anemia, hemic and lymphatic diseases, Genotype, genetics, gene mutation, DNA extraction, Genetics, biology, pathogenesis, Fanconi anemia group A protein, Articles, bioinformatics, cell line, genetic screening, Hematology, cohort analysis, Fanconi Anemia Complementation Group Proteins, founder effect, Italy, nucleic acid database, Errata Corrige, Databases, Nucleic Acid, amino acid substitution, Fanconi anemia group C protein, Fanconi anemia group D2 protein, Fanconi anemia group G protein, Fanconi anemia proteinarticle, bone marrow depression, controlled study, gene sequence, human, human cell, missense mutation, molecular diagnosis, molecular genetics, protein analysis, mosaicism, mutation, congenital, hereditary, and neonatal diseases and abnormalities, Biology, Polymorphism, Single Nucleotide, FANCD2, medicine, Humans, Genetic heterogeneity, Computational Biology, nutritional and metabolic diseases, medicine.disease, FANCA, FANCB
Abstract

Fanconi anemia is an inherited disease characterized by congenital malformations, pancytopenia, cancer predisposition, and sensitivity to cross-linking agents. The molecular diagnosis of Fanconi anemia is relatively complex for several aspects including genetic heterogeneity with mutations in at least 16 different genes. In this paper, we report the mutations identified in 100 unrelated probands enrolled into the National Network of the Italian Association of Pediatric Hematoly and Oncology. In approximately half of these cases, mutational screening was carried out after retroviral complementation analyses or protein analysis. In the other half, the analysis was performed on the most frequently mutated genes or using a next generation sequencing approach. We identified 108 distinct variants of the FANCA, FANCG, FANCC, FANCD2, and FANCB genes in 85, 9, 3, 2, and 1 families, respectively. Despite the relatively high number of private mutations, 45 of which are novel Fanconi anemia alleles, 26% of the FANCA alleles are due to 5 distinct mutations. Most of the mutations are large genomic deletions and nonsense or frameshift mutations, although we identified a series of missense mutations, whose pathogenetic role was not always certain. The molecular diagnosis of Fanconi anemia is still a tiered procedure that requires identifying candidate genes to avoid useless sequencing. Introduction of next generation sequencing strategies will greatly improve the diagnostic process, allowing a rapid analysis of all the genes.

Published
2014

48. Class IV laser therapy as treatment for chemotherapy-induced oral mucositis in onco-haematological paediatric patients: a prospective study

Author

Margherita Gobbo, Matteo Biasotto, Luca Ronfani, Federico Verzegnassi, Giulio Andrea Zanazzo, Maddalena Chermetz, Roberto Di Lenarda, Nathaniel S. Treister, Serena Zacchigna, Giulia Ottaviani, Chermetz, Maddalena, Gobbo, Margherita, Ronfani, L, Ottaviani, Giulia, Zanazzo, Ga, Verzegnassi, F, Treister, N, DI LENARDA, Roberto, Biasotto, Matteo, and Zacchigna, S.

Subjects
Stomatitis, medicine.medical_specialty, Chemotherapy, Visual analogue scale, business.industry, medicine.medical_treatment, Antineoplastic Agents, Total body irradiation, medicine.disease, Surgery, Transplantation, Oral mucositis, Laser therapy, Hematologic Neoplasms, medicine, Mucositis, Humans, Laser Therapy, Prospective Studies, Child, Prospective cohort study, business, General Dentistry, Paediatric patients
Abstract

Background Oral mucositis is a debilitating side effect of chemotherapy. Laser therapy has recently demonstrated efficacy in the management of oral mucositis (OM). Aim This prospective study was conducted to evaluate the efficacy of class IV laser therapy in patients affected by OM. Design Eighteen onco-haematological paediatric patients receiving chemotherapy and/or haematopoietic stem cell transplantation, prior to total body irradiation, affected by OM, were enrolled in this study. Patients were treated with class IV laser therapy for four consecutive days; the assessment of OM was performed through WHO Oral Mucositis Grading Objective Scale, and pain was evaluated through visual analogue scale. Patients completed a validated questionnaire, and photographs of lesions were taken during each session. Patients were re-evaluated 11 days after the first day of laser therapy. Results All patients demonstrated improvement in pain sensation, and all mucositis was fully resolved at the 11-day follow-up visit, with no apparent side effects. Laser therapy was well tolerated with remarkable reduction in pain associated with oral mucositis after 1–2 days of laser therapy. Conclusions Given class IV laser therapy appears to be safe, non-invasive, and potentially effective, prospective, randomized, controlled trials are necessary to further assess efficacy and to determine optimal treatment parameters.

Published
2014

49. TNF-α SNP rs1800629 and risk of relapse in childhood acute lymphoblastic leukemia: relation to immunophenotype

Author

Giuliana Decorti, Alberto Tommasini, Maria Grazia Valsecchi, Federico Verzegnassi, Paola Rebora, Raffaella Franca, Marco Rabusin, Giuseppe Basso, Diego Favretto, Emmanouil Athanasakis, Franca, R, Rebora, P, Athanasakis, E, Favretto, D, Verzegnassi, F, Basso, G, Tommasini, A, Valsecchi, M, Decorti, G, Rabusin, M, Franca, Raffaella, Paola, Rebora, Athanasakis, Emmanouil, Diego, Favretto, Federico, Verzegnassi, Giuseppe, Basso, Tommasini, Alberto, Maria Grazia Valsecchi, Decorti, Giuliana, and Marco, Rabusin

Subjects
Male, Oncology, medicine.medical_specialty, Adolescent, Genotype, rs1800629, acute lymphoblastic leukemia, TNF-α, Antineoplastic Agents, Precursor T-Cell Lymphoblastic Leukemia-Lymphoma, Lower risk, Risk Assessment, Immunophenotyping, Recurrence, hemic and lymphatic diseases, Internal medicine, Genetics, Humans, Medicine, SNP, Child, Childhood Acute Lymphoblastic Leukemia, Pharmacology, Tumor Necrosis Factor-alpha, business.industry, Proportional hazards model, Infant, Precursor Cell Lymphoblastic Leukemia-Lymphoma, medicine.disease, Leukemia, Lymphocytic, Chronic, B-Cell, Minor allele frequency, Leukemia, Phenotype, Drug Resistance, Neoplasm, Child, Preschool, childhood acute lymphoblastic leukemia, Immunology, Molecular Medicine, Female, Steroids, business, Risk assessment
Abstract

Aim: In the AIEOP-BFM ALL (Associazione Italiana Ematologia Oncologia Pediatrica-Berlin Frankfurt Münster acute lymphoblastic leukemia) 2000 protocol, 70% of relapsed patients had favorable prognostic features and fell within less intensive polychemotherapeutic regimens, suggesting the need for better assessing lower risk stratification. Materials & methods: A novel two-phase study design selected 614 children to be genotyped for TNF-α SNP rs1800629 (-308G>A). A weighted Cox model was applied to evaluate the SNP effect on hazard of relapse, adjusting for immunophenotype, risk group, age and gender and including interaction terms. Results: Significant interaction was found with immunophenotypes (p = 0.0007, with minor allele genotypes being adverse genetic markers in B-cell acute lymphoblastic leukemia and protective ones in T-cell acute lymphoblastic leukemia), and also with risk protocols (p = 0.0041, with minor allele genotypes as prognostic factor of relapse for standard risk patients [only one T-cell acute lymphoblastic leukemia in the subgroup analyzed]). Conclusion: The presence of at least one A allele in TNF-α SNP rs1800629 should suggest a closer monitoring in B-cell acute lymphoblastic leukemia standard risk patients. Original submitted 12 September 2013; Revision submitted 16 December 2013

Published
2014

50. Multilocus genotypes of relevance for drug metabolizing enzymes and therapy with thiopurines in patients with acute lymphoblastic leukemia

Author

Gabriele eStocco, Raffaella eFranca, Federico eVerzegnassi, Margherita eLondero, Marco eRabusin, Giuliana eDecorti, Stocco, Gabriele, Franca, Raffaella, Verzegnassi, F., Londero, Margherita, Rabusin, M., and Decorti, Giuliana

Subjects
lcsh:QH426-470, PACSIN2, Azathioprine, acute lymphoblastic leukemia, Review Article, Pharmacology, mercaptopurine, multiloucs genotypes, Bioinformatics, ITPA, thiopurine, TPMT, Genotype, medicine, Genetics, Adverse effect, Genetics (clinical), multilocus genotype, Thiopurine methyltransferase, biology, business.industry, thiopurines, multilocus genotypes, Acute Lymphoblastic Leukemia, Mercaptopurine, lcsh:Genetics, Pharmacogenetics, Pharmacogenomics, biology.protein, Molecular Medicine, business, medicine.drug
Abstract

Multilocus genotypes have been shown to be of relevance for using pharmacogenomic principles to individualize drug therapy. As it relates to thiopurine therapy, genetic polymorphisms of TPMT are strongly associated with the pharmacokinetics and clinical effects of thiopurines (mercaptopurine and azathioprine), influencing their toxicity and efficacy. We have recently demonstrated that TPMT and ITPA genotypes constitute a multilocus genotype of pharmacogenetic relevance for children with acute lymphoblastic leukemia (ALL) receiving thiopurine therapy. The use of high-throughput genomic analysis allows identification of additional candidate genetic factors associated with pharmacogenetic phenotypes, such as TPMT enzymatic activity: PACSIN2 polymorphisms have been identified by a genome-wide analysis, combining evaluation of polymorphisms and gene expression, as a significant determinant of TPMT activity in the HapMap CEU cell lines and the effects of PACSIN2 on TPMT activity and mercaptopurine induced adverse effects were confirmed in children with ALL. Combination of genetic factors of relevance for thiopurine metabolizing enzyme activity, based on the growing understanding of their association with drug metabolism and efficacy, is particularly promising for patients with pediatric ALL. The knowledge basis and clinical applications for multilocus genotypes of importance for therapy with mercaptopurine in pediatric ALL is discussed in the present review.

Published
2013

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