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1. Clinical features and follow-up in patients with 22q11.2 deletion syndrome

Author

Cancrini, C, Puliafito, P, Digilio, M, Soresina, A, Martino, S, Rondelli, R, Consolini, R, Ruga, E, Cardinale, F, Finocchi, A, Romiti, Ml, Martire, B, Bacchetta, R, Albano, V, Carotti, A, Specchia, F, Montin, D, Cirillo, E, Cocchi, G, Trizzino, A, Bossi, G, Milanesi, O, Azzari, C, Corsello, G, Pignata, C, Aiuti, A, Pietrogrande, M, Marino, B, Ugazio, A, Plebani, A, Rossi, P, Pierani, P, Gabrielli, A, Danieli, M, De Mattia, D, Sisto, C, Dammacco, F, Ranieri, G, Pession, A, Ricci, G, Minelli, P, Lougaris, V, Badolato, R, Cattaneo, R, Airò, P, Mura, R, Cossu, F, Del Giacco, S, Manconi, P, Consarino, C, Dello Russo, A, Miniero, R, Anastasio, E, Marino, S, Russo, G, Paganelli, R, Sperlì, D, Carpino, L, Aricò, M, Gambineri, E, Lippi, F, Canessa, C, Maggi, E, Romagnani, S, Matucci, A, Vultaggio, A, Gattorno, M, Castagnola, E, Nigro, G, Presta, G, Civino, A, Buzi, F, Gambaretto, G, Fasoli, S, Salpietro, C, Gallizzi, R, Dellepiane, R, Panisi, C, Fabio, G, Carrabba, M, Roncarolo, M, Biondi, A, Vallinoto, C, Poggi, V, Menna, G, Di Nardo, R, Sottile, R, Marone, G, Spadaro, G, Carli, M, Basso, G, Putti, C, Semenzato, G, Agostini, C, D'Angelo, P, Izzi, G, Bertolini, P, Zecca, M, Marseglia, G, Maccario, R, Felici, L, Favre, C, Vecchi, V, Sacchini, P, Rinaldi, G, Livadiotti, S, Simonetti, A, Stabile, A, Duse, M, Iacobini, M, Quinti, I, Fiorilli, M, Moschese, V, Cecere, F, D'Ambrosio, A, De Zan, G, Strafella, S, Tamaro, P, Rabusin, M, Tommasini, A, Tovo, P, De Carli, M, De Carli, S, Nespoli, L, Marinoni, M, Porcellini, A, Lunardi, C, Patuzzo, G, Boner, A, Degani, D, Cancrini, C, Puliafito, P, Digilio, Mc, Soresina, A, Martino, S, Rondelli, R, Consolini, R, Ruga, Em, Cardinale, F, Finocchi, A, Romiti, Ml, Martire, B, Bacchetta, R, Albano, V, Carotti, A, Specchia, F, Montin, D, Cirillo, E, Cocchi, G, Trizzino, A, Bossi, G, Milanesi, O, Azzari, C, Corsello, G, Pignata, C, Aiuti, Alessandro, Pietrogrande, Mc, Marino, B, Ugazio, Ag, Plebani, A, Rossi, P., Cancrini, Caterina, Puliafito, Pamela, Digilio, Maria Cristina, Soresina, Annarosa, Martino, Silvana, Rondelli, Roberto, Consolini, Rita, Ruga, Ezia Maria, Cardinale, Fabio, Finocchi, Andrea, Romiti, Maria Luisa, Martire, Baldassarre, Bacchetta, Rosa, Albano, Veronica, Carotti, Adriano, Specchia, Fernando, Montin, Davide, Cirillo, Emilia, Cocchi, Guido, Trizzino, Antonino, Bossi, Grazia, Milanesi, Ornella, Azzari, Chiara, Corsello, Giovanni, Pignata, Claudio, Pietrogrande, Maria Cristina, Marino, Bruno, Ugazio, Alberto Giovanni, Plebani, Alessandro, Rossi, Paolo, Aiuti, A, Rossi, P, Pierani, P, Gabrielli, A, Danieli, Mg, De Mattia, D, Sisto, C, Dammacco, F, Ranieri, G, Pession, A, Ricci, G, Minelli, P, Lougaris, V, Badolato, R, Cattaneo, R, Airò, P, Mura, Rm, Cossu, F, Del Giacco, S, Manconi, Pe, Consarino, C, Dello Russo, Am, Miniero, R, Anastasio, E, Marino, S, Russo, G, Paganelli, R, Sperlì, D, Carpino, L, Aricò, M, Gambineri, E, Lippi, F, Canessa, C, Maggi, E, Romagnani, S, Matucci, A, Vultaggio, A, Gattorno, M, Castagnola, E, Nigro, G, Presta, G, Civino, A, Buzi, F, Gambaretto, G, Fasoli, S, Salpietro, C, Gallizzi, R, Dellepiane, Rm, Panisi, C, Fabio, G, Carrabba, M, Pietrogrande, M, Roncarolo, Mg, Biondi, A, Vallinoto, C, Poggi, V, Menna, G, Di Nardo, R, Sottile, R, Marone, G, Spadaro, G, Carli, M, Basso, G, Putti, C, Semenzato, G, Agostini, C, D'Angelo, P, Izzi, G, Bertolini, P, Zecca, M, Marseglia, G, Maccario, R, Felici, L, Favre, C, Vecchi, V, Sacchini, P, Rinaldi, G, Livadiotti, S, Simonetti, A, Stabile, A, Duse, M, Iacobini, M, Quinti, I, Fiorilli, M, Moschese, V, Cecere, F, D'Ambrosio, A, De Zan, G, Strafella, S, Tamaro, Paolo, Rabusin, M, Tommasini, A, Tovo, P, De Carli, M, De Carli, S, Nespoli, L, Marinoni, M, Porcellini, A, Lunardi, C, Patuzzo, G, Boner, A, Degani, D., Cancrini, C., Pulisfito, P., Digilio, M. C., Soresina, A., Martino, S., Rondelli, R., Consolini, R., Ruga, E. M., C. a. r. d. i. n. a. l. e., F., Finocchi, A., Romiti, M. L., Martire, B., Bacchetta, R., Albano, V., Carotti, A., Specchia, F., Montin, D., Cocchi, G., Trizzino, A., Bossi, G., Milanesi, O., Azzari, C., Corsello, G., Aiuti, A., Pietrogrande, M. C., Marino, B., Ugazio, A. G., Plebani, A., Digilio, MC, Ruga, EM, Romiti, ML, trizzino, A, Aiuti, Pietrogrande, MC, and Ugazio, AG

Subjects
Male, Pediatrics, 22q11.2 deletion, Delayed Diagnosis, Time Factors, Chromosomes, Human, Pair 22, Developmental Disabilities, digeorge syndrome, Sex Factor, Severity of Illness Index, Retrospective Studie, DiGeorge syndrome, Early Diagnosi, Age Factor, Prospective Studies, Neonatal hypocalcemia, Prospective cohort study, Child, medicine.diagnostic_test, Delayed Diagnosi, Primary immune disorders, Age Factors, del 22q, MIM, Abnormalities, Multiple, Adolescent, Adult, Child, Preschool, DiGeorge Syndrome, Early Diagnosis, Female, Follow-Up Studies, Genetic Testing, Humans, Infant, Infant, Newborn, Monitoring, Physiologic, Retrospective Studies, Risk Assessment, Sex Factors, Young Adult, Disease Progression, Cohort, Abnormalities, Multiple, Pediatrics, Perinatology and Child Health, Human, medicine.medical_specialty, Time Factor, Monitoring, Developmental Disabilitie, Italian Association of Pediatric Haematology and Oncology, Context (language use), Chromosomes, Follow-Up Studie, Severity of illness, medicine, 22q11DS, 22q11.2 deletion syndrome, AIEOP, Mendelian Inheritance in Man, Preschool, Physiologic, Genetic testing, Settore MED/38 - Pediatria Generale e Specialistica, business.industry, Retrospective cohort study, medicine.disease, Newborn, Prospective Studie, Pair 22, business
Abstract

Objective To investigate the clinical manifestations at diagnosis and during follow-up in patients with 22q11.2 deletion syndrome to better define the natural history of the disease. Study design A retrospective and prospective multicenter study was conducted with 228 patients in the context of the Italian Network for Primary Immunodeficiencies. Clinical diagnosis was confirmed by cytogenetic or molecular analysis. Results The cohort consisted of 112 males and 116 females; median age at diagnosis was 4 months (range 0 to 36 years 10 months). The diagnosis was made before 2 years of age in 71% of patients, predominantly related to the presence of heart anomalies and neonatal hypocalcemia. In patients diagnosed after 2 years of age, clinical features such as speech and language impairment, developmental delay, minor cardiac defects, recurrent infections, and facial features were the main elements leading to diagnosis. During follow-up (available for 172 patients), the frequency of autoimmune manifestations ( P = .015) and speech disorders ( P = .002) increased. After a median follow-up of 43 months, the survival probability was 0.92 at 15 years from diagnosis. Conclusions Our data show a delay in the diagnosis of 22q11.2 deletion syndrome with noncardiac symptoms. This study provides guidelines for pediatricians and specialists for early identification of cases that can be confirmed by genetic testing, which would permit the provision of appropriate clinical management.

Published
2014

2. Marriage and parenthood among childhood cancer survivors: a report from the Italian AIEOP Off-Therapy Registry

Author

Pivetta, E, Maule, Mm, Pisani, P, Zugna, D, Haupt, R, Jankovic, M, Aricò, M, Casale, F, Clerico, A, Cordero di Montezemolo, L, Kiren, V, Locatelli, F, Palumbo, G, Pession, A, Pillon, M, Santoro, N, Terenziani, M, Valsecchi, Mg, Dama, E, Magnani, C, Merletti, F, Pastore, G, Fagioli, F, Bona, G, Dini, G, Carnelli, V, Biondi, A, Zecca, M, Conter, V, Porta, F, Fedeli, F, Massimino, M, Nespoli, L, Roncarolo, Mg, Carli, M, Cesaro, S, Memo, L, Colleselli, P, Battisti, L, Tamaro, Paolo, Mascarin, M, Nocerino, A, Izzi, G, Paolucci, P, Borgna, Pc, Vecchi, V, Abate, Me, Acquaviva, A, Favre, C, Aversa, F, Pierani, P, Felici, L, Visani, G, Fioritoni, G, Foa, R, Riccardi, R, Frega, G, Poggi, V, Amendola, G, Filosa, A, Ladogana, S, Presta, G, Pozzi, S, De Mattia, D, Consarino, C, Nobile, F, Sperlì, D, D'Angelo, P, Marino, S, Gallisai, D, Targhetta, R., Pivetta, E, Maule, M, Pisani, P, Zugna, D, Haupt, R, Jankovic, M, Aricò, M, Casale, F, Clerico, A, Cordero di Montezemolo, L, Kiren, V, Locatelli, F, Palumbo, G, Pession, A, Pillon, M, Santoro, N, Terenziani, M, Valsecchi, M, Dama, E, Magnani, C, Merletti, F, Pastore, G, Maule, Mm, Valsecchi, Mg, Fagioli, F, Bona, G, Dini, G, Carnelli, V, Biondi, A, Zecca, M, Conter, V, Porta, F, Fedeli, F, Massimino, M, Nespoli, L, Roncarolo, Mg, Carli, M, Cesaro, S, Memo, L, Colleselli, P, Battisti, L, Tamaro, Paolo, Mascarin, M, Nocerino, A, Izzi, G, Paolucci, P, Borgna, Pc, Vecchi, V, Abate, Me, Acquaviva, A, Favre, C, Aversa, F, Pierani, P, Felici, L, Visani, G, Fioritoni, G, Foa, R, Riccardi, R, Frega, G, Poggi, V, Amendola, G, Filosa, A, Ladogana, S, Presta, G, Pozzi, S, De Mattia, D, Consarino, C, Nobile, F, Sperlì, D, D'Angelo, P, Marino, S, Gallisai, D, and Targhetta, R.

Subjects
Adult, Male, Parents, Pediatrics, medicine.medical_specialty, Total fertility rate, media_common.quotation_subject, Population, Editorials and Perspectives, Fertility, off-therapy, cancer survival, Cohort Studies, Quality of life, Marriage and parenthood, Medicine, Humans, cancer, cancer survivors, childhood, Registries, Survivors, Marriage, education, Child, media_common, education.field_of_study, business.industry, Infant, Newborn, Infant, Hematology, Original Articles, Middle Aged, childhood cancer, fertility, long-term survivors, marriage, quality of life, El Niño, Italy, Child, Preschool, Hematologic Neoplasms, Cohort, Marital status, Female, business, Cohort study, Follow-Up Studies
Abstract

Background The aim of this study was to describe the patterns of marriage and parenthood in a cohort of childhood cancer survivors included in the Off-Therapy Registry maintained by the Italian Association of Pediatric Hematology and Oncology. Design and Methods We analyzed a cohort of 6,044 patients diagnosed with cancer between 1960 and 1998, while aged 0 to 14 years and who were 18 years old or older by December 2003. They were followed up through the regional vital statistics registers until death or the end of follow up (October 30, 2006), whichever occurred first, and their marital status and date of birth of their children were recorded. The cumulative probabilities of being married and having a first child were computed by gender and compared by tumor type within the cohort. Marriage and fertility rates (the latter defined as the number of live births per woman-year) were compared with those of the Italian population of the same age, gender, area of residence and calendar period by means of the observed to expected (O/E) ratios. Results During the follow-up period, 4,633 (77%) subjects had not married. The marriage O/E ratios were 0.56 (95% CI: 0.51-0.61) and 0.70 (95% CI: 0.65-0.76) among men and women, respectively. Overall, 263 men had 367 liveborn children, and 473 women had 697 liveborn children. The female fertility O/E ratio was 0.57 (95% CI: 0.53-0.62) overall, and 1.08 (95% CI: 0.99-1.17) when analyses were restricted to married/cohabiting women Conclusions Childhood cancer survivors are less likely to marry and to have children than the general population, confirming the life-long impact of their previous disease on their social behavior and choices. The inclusion of counseling in the strategies of management and long-term surveillance of childhood cancer patients could be beneficial to survivors as they approach adulthood. ©2011 Ferrata Storti Foundation.

Published
2011

3. The Quality of Life of Children and Adolescents with X-Linked Agammaglobulinemia

Author

Soresina A., Nacinovich R., Bomba M., Cassani M., Molinaro A., Sciotto A., Martino S., Cardinale F., De Mattia D., Putti C., Dellepiane R.M., Felici L., Parrinello G., Neri F., Plebani A., Pierani P., DeMattia D., Martire B., Armenio L., Dammacco F., Ranieri G., Masi M., Miniaci A., Pession A., Rondelli R., Notarangelo L. D., Cao, Cossu F., Del Giacco S., Manconi P., Evangelista I., Magro S., Morgione S., STRISCIUGLIO, PIETRO, Anastasio E., Schillirò G., Paganelli R., Sticca M., Sperlì D., Carpino L., Bernini G., Azzari C., Maggi E., Romagnani S., Matucci A., Vultaggio A., Castagnola E., Gattorno M., Presta G., Civino A., Gambaretto G., Fasoli S., Salpietro C., Pietrogrande M.C., DellePiane R.M., Panisi C., Cambiaghi G., Pietrogrande M., Roncarolo M.G., Aiuti A., Masera G., Biondi A., Sala A., PIGNATA, CLAUDIO, Poggi V., Menna G., Di Nardo R., D'Apuzzo A., Pelliccia A., Correra A., Marone G., SPADARO, GIUSEPPE, Carli M., Zanesco L., Basso G., Putti M.C., Semenzato G., Agostini C., Amato G.M., Aricò M., Trizzino A., Izzi G., Bertolini P., Locatelli F., Zecca M., Rondini G., Marseglia G.L., Maccario R., Bossi G., Favre C., Consolini R., Vecchi V., Sacchini P., Rinaldi G., Ugazio A.G., Rossi P., Livadiotti S., Cancrini C., Finocchi A., Stabile A., Duse M., Iacobini M., Quinti I., Moschese V., Cecere F., Morgese G., Acquaviva A., De Zan G., Strafella S., Tamaro P., Rabusin M., Tovo P.A., Nespoli L., Marinoni M., Porcellini A., Cazzola G.A., Annarosa, Soresina, Renata, Nacinovich, Monica, Bomba, Morena, Cassani, Molinaro, Anna, Antonella, Sciotto, Silvana, Martino, Fabio, Cardinale, Domenico, Mattia, Caterina, Putti, Rosa Maria, Dellepiane, Leonardo, Felici, Giovanni, Parrinello, Francesca, Neri, Alessandro, Plebani, Soresina, A, Nacinovich, R, Bomba, M, Cassani, M, Molinaro, A, Sciotto, A, Martino, S, Cardinale, F, De Mattia, D, Putti, C, Dellepiane, R, Felici, L, Parrinello, G, Neri, F, Plebani, A, Soresina, A., Nacinovich, R., Bomba, M., Cassani, M., Molinaro, A., Sciotto, A., Martino, S., Cardinale, F., De Mattia, D., Putti, C., Dellepiane, R. M., Felici, L., Parrinello, G., Neri, F., Plebani, A., Pierani, P., Demattia, D., Martire, B., Armenio, L., Dammacco, F., Ranieri, G., Masi, M., Miniaci, A., Pession, A., Rondelli, R., Notarangelo, L. D., Cao, Cossu, F., Del Giacco, S., Manconi, P., Evangelista, I., Magro, S., Morgione, S., Strisciuglio, Pietro, Anastasio, E., Schillirò, G., Paganelli, R., Sticca, M., Sperlì, D., Carpino, L., Bernini, G., Azzari, C., Maggi, E., Romagnani, S., Matucci, A., Vultaggio, A., Castagnola, E., Gattorno, M., Presta, G., Civino, A., Gambaretto, G., Fasoli, S., Salpietro, C., Pietrogrande, M. C., Panisi, C., Cambiaghi, G., Pietrogrande, M., Roncarolo, M. G., Aiuti, A., Masera, G., Biondi, A., Sala, A., Pignata, Claudio, Poggi, V., Menna, G., Di Nardo, R., D'Apuzzo, A., Pelliccia, A., Correra, A., Marone, G., Spadaro, Giuseppe, Carli, M., Zanesco, L., Basso, G., Putti, M. C., Semenzato, G., Agostini, C., Amato, G. M., Aricò, M., Trizzino, A., Izzi, G., Bertolini, P., Locatelli, F., Zecca, M., Rondini, G., Marseglia, G. L., Maccario, R., Bossi, G., Favre, C., Consolini, R., Vecchi, V., Sacchini, P., Rinaldi, G., Ugazio, A. G., Rossi, P., Livadiotti, S., Cancrini, C., Finocchi, A., Stabile, A., Duse, M., Iacobini, M., Quinti, I., Moschese, V., Cecere, F., Morgese, G., Acquaviva, A., De Zan, G., Strafella, S., Tamaro, P., Rabusin, M., Tovo, P. A., Nespoli, L., Marinoni, M., Porcellini, A., and Cazzola, G. A.

Subjects
Male, Pediatrics, medicine.medical_specialty, x-linked agammaglobulinemia, Activities of daily living, Adolescent, X-linked agammaglobulinemia, Health Status, Immunology, pedsql 4.0 generic core scale, Quality of life, children, Agammaglobulinemia, Surveys and Questionnaires, Activities of Daily Living, health-related quality of life, parents, medicine, Humans, Immunology and Allergy, PedsQL 4.0 Generic Core Scale, Child, Settore MED/38 - Pediatria Generale e Specialistica, Health related quality of life, quality of live, business.industry, Immunoglobulins, Intravenous, Genetic Diseases, X-Linked, medicine.disease, Socioeconomic Factors, Child, Preschool, Mutation, Quality of Life, Female, X-linked agammaglobulinemia - children - parents - health-related quality of life - PedsQL 4.0 Generic Core Scale, business
Abstract

Introduction: The health-related quality of life in X-linked agammaglobulinemia was investigated in 25 children and adolescents patients through the Italian version of Pediatric Quality of Life Inventory 4.0 Generic Core Scale for patients aged less then 18 years, comparing child perception to that of the parents and the physician's evaluation. The data were compared with the ones of 80 healthy controls and the literature data of a group of patients with rheumatic diseases. Discussion: The agammaglobulinemia subjects perceived a lower global quality of life than the healthy subjects, but significantly higher than the rheumatic diseases controls. The clinical relevance of health-related quality of life assessment in X-linked agammaglobulinemia pediatric patients is discussed. © 2008 Springer Science+Business Media, LLC.

Published
2009

4. Patterns of domestic migrations and access to childhood cancer care centres in Italy: A report from the hospital based registry of the Italian Association of Pediatric Hematology and Oncology (AIEOP)

Author

Dama, E, Rondelli, R, De Rosa, M, Aricò, M, Carli, M, Bellani, Ff, Magnani, C, Merletti, F, Pastore, G, Pession, A, Madon, E, Dini, G, Carnelli, V, Fedeli, F, Fossati Bellani, F, Masera, G, Locatelli, F, Cornelli, Pe, Notarangelo, L, Nespoli, L, Bagnulo, S, Marradi, P, Musi, L, Rodeghiero, F, Grotto, P, Rossetti, F, Battisti, L, Tamaro, Paolo, Mascarin, M, Nocerino, A, Izzi, G, Paolucci, P, Ambrosioni, G, Picci, P, Borgna Pignatti, C, Vecchi, V, Bernini, G, Morgese, G, Favre, C, Zucchetti, P, Pierani, P, Felici, L, Visani, G, Di Bartolomeo, P, Ballati, G, Castello, Ma, De Rossi, G, Donfrancesco, A, Foà, R, Menichelli, A, Riccardi, R, Di Tullio MT, Fiorillo, A, Poggi, V, Amendola, G, Ladogana, S, Ruggiero, L, Pozzi, S, De Mattia, D, Magro, S, Nobile, F, Sperlì, D, Schilirò, G, Gallisai, D, Biddau, P., Dama, E, Rondelli, R, De Rosa, M, Aricò, M, Carli, M, Bellani, Ff, Magnani, C, Merletti, F, Pastore, G, Pession, A, Madon, E, Dini, G, Carnelli, V, Fedeli, F, Fossati Bellani, F, Masera, G, Locatelli, F, Cornelli, Pe, Notarangelo, L, Nespoli, L, Bagnulo, S, Marradi, P, Musi, L, Rodeghiero, F, Grotto, P, Rossetti, F, Battisti, L, Tamaro, Paolo, Mascarin, M, Nocerino, A, Izzi, G, Paolucci, P, Ambrosioni, G, Picci, P, Borgna Pignatti, C, Vecchi, V, Bernini, G, Morgese, G, Favre, C, Zucchetti, P, Pierani, P, Felici, L, Visani, G, Di Bartolomeo, P, Ballati, G, Castello, Ma, De Rossi, G, Donfrancesco, A, Foà, R, Menichelli, A, Riccardi, R, Di Tullio, Mt, Fiorillo, A, Poggi, V, Amendola, G, Ladogana, S, Ruggiero, L, Pozzi, S, De Mattia, D, Magro, S, Nobile, F, Sperlì, D, Schilirò, G, Gallisai, D, Biddau, P., Dama E, Rondelli R, De Rosa M, Aricò M, Carli M, Bellani FF, Magnani C, Merletti F, Pastore G, Pession A, and Italian Association of Pediatric Hematology and Oncology (AIEOP).

Subjects
Oncology, Male, Cancer Research, medicine.medical_specialty, Adolescent, Paediatric haematology, care access, childhood cancer, italy, specialised cancer centres, Childhood cancer, Child Health Services, Regional Medical Programs, Tertiary care, Health Services Accessibility, domestic migration, Internal medicine, Neoplasms, Oncology Service, Hospital, medicine, Humans, Child, Specialised cancer centres, Care access, Italy, business.industry, Infant, Newborn, Infant, Hospital based, El Niño, Child, Preschool, Residence, Female, Health Services Research, Pediatric hematology, business, Delivery of Health Care
Abstract

Tertiary care centres, grouped in the Italian Association of Paediatric Haematology and Oncology (AIEOP) are unevenly distributed across the country. In an attempt to describe their perceived efficacy, we matched the residence and the location of the treatment centre in 18,441 patients aged ⩽15 years treated in the AIEOP network between 1989 and 2005. Overall, centres located in the central and southern regions were less appealing than those located in the North, although this trend decreased over the study period. Patients with solid tumours migrated more frequently than those with leukaemia or lymphoma. Information resulting from better knowledge of the non-random migrations for treatment of children with cancer will be useful to refine planning of the national paediatric haematology-oncology network with social and economic implications.

Published
2008

5. Survival of children with cancer in Italy, 1989-98. A report from the hospital based registry of the Italian Association of Pediatric Haematology and Oncology (AIEOP)

Author

Pession A, Dama E, Rondelli R, Magnani C, De Rosa M, Locatelli F, Fagioli F, Haupt R, Jankovic M, Terracini B, Merletti F, Pastore G, Italian Association of Paediatric Haematology, Oncology Madon E, Dini G, Carnelli V, Fedeli F, Fossati Bellani F, Masera G, Cornelli PE, Porta F, Dorizzi A, Nespoli A, Carli M, Marradi P, Rodeghiero F, Musi L, Mascarin M, Nocerino A, Izzi G, Paolucci P, Ambrosioni G, Picci P, Borgna Pignatti C, Bernini G, Morgese G, Favre C, Aversa F, Pierani P, Di Marzio A, Foà R, De Rossi G, Donfrancesco A, Castello MA, Casale F, Poggi V, Auricchio S, Antonelli P, Ladogana S, De Mattia D, Magro S, Nobile F, Aricò M, Schilirò G, Gallisai D, Argiolu F., TAMARO, PAOLO, Pession, A, Dama, E, Rondelli, R, Magnani, C, De Rosa, M, Locatelli, F, Fagioli, F, Haupt, R, Jankovic, M, Terracini, B, Merletti, F, Pastore, G, Italian Association of Paediatric, Haematology, Oncology Madon, E, Dini, G, Carnelli, V, Fedeli, F, Fossati Bellani, F, Masera, G, Cornelli, Pe, Porta, F, Dorizzi, A, Nespoli, A, Carli, M, Marradi, P, Rodeghiero, F, Musi, L, Tamaro, Paolo, Mascarin, M, Nocerino, A, Izzi, G, Paolucci, P, Ambrosioni, G, Picci, P, Borgna Pignatti, C, Bernini, G, Morgese, G, Favre, C, Aversa, F, Pierani, P, Di Marzio, A, Foà, R, De Rossi, G, Donfrancesco, A, Castello, Ma, Casale, F, Poggi, V, Auricchio, S, Antonelli, P, Ladogana, S, De Mattia, D, Magro, S, Nobile, F, Aricò, M, Schilirò, G, Gallisai, D, Argiolu, F., Pession A, Dama E, Rondelli R, Magnani C, De Rosa M, Locatelli F, Fagioli F, Haupt R, Jankovic M, Terracini B, Merletti F, Pastore G, and on behalf of the Italian Association of Paediatric Haematology and Oncology

Subjects
Oncology, Male, Cancer Research, medicine.medical_specialty, Pediatrics, Adolescent, Lymphoproliferative disorders, Cancer registration, survival, children cancer, Internal medicine, Neuroblastoma, Neoplasms, medicine, Humans, Survivors, Sex Distribution, Child, Survival rate, Childhood cancer, Gender, Italy, Survival, Hematology, business.industry, Hazard ratio, Cancer, Infant, medicine.disease, Lymphoma, El Niño, Child, Preschool, Female, business, Epidemiologic Methods
Abstract

We describe the survival patterns of 10,791 Italian children (age 0-14) diagnosed with cancer during 1989-1998 and who were included in the hospital-based registry of the Italian Association of Paediatric Haematology and Oncology. Five-year cumulative survival percentages were 76% for lymphoproliferative disorders and 68% for solid tumours. Survival rates in 1994-1998 significantly improved for acute lymphocytic leukaemia (ALL), acute non-lymphocytic leukaemia, Hodgkin's lymphoma and Wilms' tumour. Gender and age were determinants of survival for some specific types of cancer. Girls with ALL and neuroblastoma exhibited a significant advantage (hazard ratio HR 0.72, 0.62-0.83) and disadvantage (HR 0.73, 0.59-0.90) over boys, respectively. Children with a Wilms' tumour diagnosed above age 3 had a worse prognosis than younger children (HR 2.3, 1.4-4.1). The persisting gender-related difference in survival rate for ALL requires understanding as to whether it is attributable to delays in the adoption of more recent therapeutic protocols, while the corresponding findings for Wilms' tumour and neuroblastoma deserve further biological interpretation.

Published
2008

6. Bloodstream infections in children with cancer: a multicentre surveillance study of the Italian Association of Paediatric Haematology and Oncology. Supportive Therapy Group-Infectious Diseases Section

Author

Viscoli, Claudio, Castagnola, Elio, Giacchino, M, Cesáro, S, Properzi, E, Tucci, F, Mura, R. M, Alvisi, P, Zanazzo, G, Surico, G, Bonetti, F, De Sio, L, Izzi, G, Di Cataldo, A, Ziino, O, Massolo, F, Nardi, M, Santoro, N, and Binda, S.

Subjects
Male, bacteraemia, Neutropenia, Drug Resistance, fungaemia, Bacteremia, Drug Resistance, Microbial, Microbial, Italy, Neoplasms, Humans, Child, Female, Fungemia, Prospective Studies, cancer infections
Abstract

A one-year prospective, multicentre surveillance study on aetiology, main clinical features and outcome of bloodstream infections in children with cancer was conducted in 18 paediatric haematology centres belonging to the Italian Association for Paediatric Haematology and Oncology. A total of 191 bloodstream infections were reported during the study period. Of them, 123 (64%) occurred in neutropenic and 68 (36%) in non-neutropenic patients. Gram-positive cocci caused 45% (85/191) of the episodes, gram-negative rods 41% (78/191), and fungi 9% (18/191). The remaining 5% (10/191) of the episodes were poly-microbial infections. A total of 204 pathogens were isolated (46% gram-positive cocci; 44% gram-negative rods; and 10% fungi). The aetiologic distribution was similar among neutropenic and non-neutropenic patients. A correlation between the infection and the presence of an indwelling central venous catheter was found in 20% (23/114) of the episodes among neutropenic patients and in 55% (23/62) among non-neutropenic patients. Gram-negative micro-organisms were isolated in an unusually high proportion of catheter-related infections (48%). The overall mortality rate from any cause within 30 days from the first positive blood culture was 11%, and was higher among patients who were neutropenic at the onset of the infection than among those who were not neutropenic (15 versus 4%, P = 0.03). In addition, the mortality was significantly higher in recipients of bone marrow transplantation than in patients with acute leukaemia or solid tumour (21, 11 and 6%, respectively) and was also higher in fungaemias and poly-microbial infections (22 and 30%) than in single gram-positive and gram-negative bacteraemias (11 and 6%).

Published
1999

7. Italian cancer figures, report 2012: Cancer in children and adolescents

Author

Pisani, P, Buzzoni, C, Crocetti, E, Maso, Ld, Rondelli, R, Alessi, D, Aricò, M, Bidoli, E, Ferrari, A, Fusco, M, Gatta, G, Guzzinati, S, Jankovic, M, Locatelli, F, Maule, M, Mosso, Ml, Pastore, G, Pession, A, Rashid, I, Sacerdote, C, Terracini, B, Tognazzo, S, Trama, A, Ferretti, S, Porta, F, Mazzoleni, G, Tschugguel, B, De Valiere, E, Facchinelli, G, Falk, M, Cappello, Td, Giacomin, A, Vercellino, Pc, Andreone, S, Marzola, L, Migliari, E, Carletti, N, Nenci, I, Caldarella, A, Corbinelli, A, Intrieri, T, Manneschi, G, Nemcova, L, Ocello, C, Sacchettini, C, Zappa, M, Paci, E, Serraino, D, Angelin, T, Birri, S, De Dottori, M, De Santis, E, Forgiarini, O, Zucchetto, A, Zanier, L, Vercelli, M, Orengo, Ma, Casella, C, Marani, E, Puppo, A, Celesia, Mv, Cogno, R, Levrieri, I, Manenti, S, Garrone, E, Quaglia, A, Pannozzo, F, Busco, S, Ramazzotti, V, Cercato, Mc, Battisti, W, Sperduti, I, Macci, L, Bugliarello, E, Valerio, O, Bernazza, E, Tamburo, L, Rossi, M, Curatella, S, Tamburrino, S, Fattoruso, S, Bisanti, L, Autelitano, M, Cuccaro, F, Randi, G, Ghilardi, S, Leone, R, Filipazzi, L, Bonini, A, Giubelli, C, Federico, M, Artioli, Me, Valla, K, Braghiroli, B, Cirilli, C, Luminari, S, Pirani, M, Ferrari, L, Bellatalla, C, Panico, M, Perrotta, C, Vassante, B, Vitale, Mf, Michiara, M, Bozzani, F, Sgargi, P, Tumino, R, Rosa, Mg, Cascone, G, Frasca, G, Giurdanella, Mc, Martorana, C, Morana, G, Nicita, C, Rollo, Pc, Ruggeri, Mg, Sigona, A, Spata, E, Vacirca, S, Mangone, L, Di Felice, E, Pezzarossi, A, Caroli, S, Pellegri, C, Vicentini, M, D'Angelo, S, Balottari, P, Romagna, Rt, Falcini, F, Colamartini, A, Bucchi, L, Balducci, C, Ravegnani, M, Vitali, B, Cordaro, C, Caprara, L, Giuliani, O, Giorgetti, S, Palumbo, M, Vattiato, R, Ravaioli, A, Mancini, S, Tonelli, C, Amadori, M, Cremone, L, Iannelli, A, Senatore, G, Zevola, A, Sechi, O, Budroni, M, Cesaraccio, R, Pirino, D, Carboni, D, Fiori, G, Soddu, M, Mameli, G, Mura, F, Contrino, Ml, Madeddu, A, Tisano, F, Sciacca, S, Muni, A, Mizzi, M, Russo, M, Sacco, G, Aletta, P, Ziino, Ac, Tessandori, R, Fanetti, Ac, Maspero, S, Cecconami, L, Annulli, Ml, Moroni, E, Gonzalez, Me, Zanetti, R, Rosso, S, Patriarca, S, Prandi, R, Sobrato, I, Gilardi, F, Busso, P, Piffer, S, Rizzello, R, Gentilini, Ma, Cappelletti, M, Rosa, Fl, Stracci, F, D'Alò, D, Scheibel, M, Costarelli, D, Spano, F, Rossini, S, Santucci, C, Petrinelli, Am, Solimene, C, Bianconi, F, Brunori, V, Crosignani, P, Tagliabue, G, Contiero, P, Preto, L, Tittarelli, A, Maghini, A, Codazzi, T, Frassoldi, E, Gada, D, Costa, E, Grazia, Ld, Zambon, P, Baracco, M, Bovo, E, Cin, Ad, Fiore, Ar, Greco, A, Monetti, D, Rosano, A, Stocco, C, Donato, F, Limina, Rm, Adorni, A, Andreis, P, Zani, G, Piovani, F, Salvi, O, Puleio, M, Vitarelli, S, Candela, G, Pappalardo, G, Scuderi, T, Lottero, B, Ribaudo, M, Ricci, P, Guarda, L, Gatti, L, Bozzeda, A, Dall'Acqua, M, Pironi, V, Sardo, As, Mazzei, A, Sirianni, N, Lavecchia, Am, Mancuso, P, Vitale, F, Cusimano, R, Amodio, R, Guttadauro, A, Ravazzolo, B, Bucalo, G, Trapani, C, Sunseri, R, Cascio, Ma, Brucculeri, Ma, Adamo, Ms, Mannino, R, Mistretta, A, Costa, A, Rudisi, G, Gola, G, Corti, M, Prosperini, E, Grandi, L, Sciacchitano, S, Fidelbo, M, Benedetto, G, Vasquez, E, Torrisi, A, Pesce, P, Calabretta, L, Bella, F, Leone, A, Ieni, A, Varvarà, M, Castaing, M, Di Prima, A, Benedetto, A, Paderni, F, Usala, M, Pala, F, Sini, Gm, Pintori, N, Canu, L, Demurtas, G, Doa, N, Lazzarato, F, Magnani, C, Merletti, F, Sarcerdote, C, Macerata, V, de Leon MP, Domati, F, Rossi, G, Goldoni, Ca, Rossi, F, De Gaetani, C, Benatti, P, Roncucci, L, Di Gregorio, C, Pedroni, M, Pezzi, A, Maffei, S, Mariani, F, Borsi, E, Cocchioni, M, Pascucci, C, Gennaro, V, Lazzarotto, A, Benfatto, L, Mazzucco, G, Montanaro, F, Traina, A, Carruba, G, Dolcemascolo, C, Staiti, R, Zarcone, M, Bisogno, G, Cuttini, M, De Rosa, M, Haupt, R, Soresina, A, Valsecchi, Mg, Fagioli, F, Bona, G, Dini, G, Conter, V, Biondi, A, Zecca, M, Fedeli, F, Massimino, M, Nespoli, L, Roncarolo, Mg, Basso, G, Cesaro, S, Memo, L, Bellettato, M, Tamaro, Paolo, Mascarin, M, Rosolen, A, Izzi, G, Paolucci, P, Borgna Pignatti, C, Vecchi, V, Abate, Me, D'Ambrosio, A, Favre, C, Caniglia, M, Pierani, P, Felici, L, Visani, G, Fioritoni, G, Foà, R, Riccardi, R, Frega, G, Clerico, A, Casale, F, Poggi, V, Amendola, G, Filosa, A, Ladogana, S, Presta, G, Tornesello, A, De Mattia, D, Consarino, C, Nobile, F, Sperlì, D, D'Angelo, P, Marino, S, Cosmi, C, Mura, Rm, Battisti, L., Pisani, P, Buzzoni, C, Crocetti, E, Maso, Ld, Rondelli, R, Alessi, D, Aricò, M, Bidoli, E, Ferrari, A, Fusco, M, Gatta, G, Guzzinati, S, Jankovic, M, Locatelli, F, Maule, M, Mosso, Ml, Pastore, G, Pession, A, Rashid, I, Sacerdote, C, Terracini, B, Tognazzo, S, Trama, A, Ferretti, S, Porta, F, Mazzoleni, G, Tschugguel, B, De Valiere, E, Facchinelli, G, Falk, M, Cappello, Td, Giacomin, A, Vercellino, Pc, Andreone, S, Marzola, L, Migliari, E, Carletti, N, Nenci, I, Caldarella, A, Corbinelli, A, Intrieri, T, Manneschi, G, Nemcova, L, Ocello, C, Sacchettini, C, Zappa, M, Paci, E, Serraino, D, Angelin, T, Birri, S, De Dottori, M, De Santis, E, Forgiarini, O, Zucchetto, A, Zanier, L, Vercelli, M, Orengo, Ma, Casella, C, Marani, E, Puppo, A, Celesia, Mv, Cogno, R, Levrieri, I, Manenti, S, Garrone, E, Quaglia, A, Pannozzo, F, Busco, S, Ramazzotti, V, Cercato, Mc, Battisti, W, Sperduti, I, Macci, L, Bugliarello, E, Valerio, O, Bernazza, E, Tamburo, L, Rossi, M, Curatella, S, Tamburrino, S, Fattoruso, S, Bisanti, L, Autelitano, M, Cuccaro, F, Randi, G, Ghilardi, S, Leone, R, Filipazzi, L, Bonini, A, Giubelli, C, Federico, M, Artioli, Me, Valla, K, Braghiroli, B, Cirilli, C, Luminari, S, Pirani, M, Ferrari, L, Bellatalla, C, Panico, M, Perrotta, C, Vassante, B, Vitale, Mf, Michiara, M, Bozzani, F, Sgargi, P, Tumino, R, Rosa, Mg, Cascone, G, Frasca, G, Giurdanella, Mc, Martorana, C, Morana, G, Nicita, C, Rollo, Pc, Ruggeri, Mg, Sigona, A, Spata, E, Vacirca, S, Mangone, L, Di Felice, E, Pezzarossi, A, Caroli, S, Pellegri, C, Vicentini, M, D'Angelo, S, Balottari, P, Romagna, Rt, Falcini, F, Colamartini, A, Bucchi, L, Balducci, C, Ravegnani, M, Vitali, B, Cordaro, C, Caprara, L, Giuliani, O, Giorgetti, S, Palumbo, M, Vattiato, R, Ravaioli, A, Mancini, S, Tonelli, C, Amadori, M, Cremone, L, Iannelli, A, Senatore, G, Zevola, A, Sechi, O, Budroni, M, Cesaraccio, R, Pirino, D, Carboni, D, Fiori, G, Soddu, M, Mameli, G, Mura, F, Contrino, Ml, Madeddu, A, Tisano, F, Sciacca, S, Muni, A, Mizzi, M, Russo, M, Sacco, G, Aletta, P, Ziino, Ac, Tessandori, R, Fanetti, Ac, Maspero, S, Cecconami, L, Annulli, Ml, Moroni, E, Gonzalez, Me, Zanetti, R, Rosso, S, Patriarca, S, Prandi, R, Sobrato, I, Gilardi, F, Busso, P, Piffer, S, Rizzello, R, Gentilini, Ma, Cappelletti, M, Rosa, Fl, Stracci, F, D'Alò, D, Scheibel, M, Costarelli, D, Spano, F, Rossini, S, Santucci, C, Petrinelli, Am, Solimene, C, Bianconi, F, Brunori, V, Crosignani, P, Tagliabue, G, Contiero, P, Preto, L, Tittarelli, A, Maghini, A, Codazzi, T, Frassoldi, E, Gada, D, Costa, E, Grazia, Ld, Zambon, P, Baracco, M, Bovo, E, Cin, Ad, Fiore, Ar, Greco, A, Monetti, D, Rosano, A, Stocco, C, Donato, F, Limina, Rm, Adorni, A, Andreis, P, Zani, G, Piovani, F, Salvi, O, Puleio, M, Vitarelli, S, Candela, G, Pappalardo, G, Scuderi, T, Lottero, B, Ribaudo, M, Ricci, P, Guarda, L, Gatti, L, Bozzeda, A, Dall'Acqua, M, Pironi, V, Sardo, A, Mazzei, A, Sirianni, N, Lavecchia, Am, Mancuso, P, Vitale, F, Cusimano, R, Amodio, R, Guttadauro, A, Ravazzolo, B, Bucalo, G, Trapani, C, Sunseri, R, Cascio, Ma, Brucculeri, Ma, Adamo, M, Mannino, R, Mistretta, A, Costa, A, Rudisi, G, Gola, G, Corti, M, Prosperini, E, Grandi, L, Sciacchitano, S, Fidelbo, M, Benedetto, G, Vasquez, E, Torrisi, A, Pesce, P, Calabretta, L, Bella, F, Leone, A, Ieni, A, Varvarà, M, Castaing, M, Di Prima, A, Benedetto, A, Paderni, F, Usala, M, Pala, F, Sini, Gm, Pintori, N, Canu, L, Demurtas, G, Doa, N, Lazzarato, F, Magnani, C, Merletti, F, Sarcerdote, C, Macerata, V, de Leon, Mp, Domati, F, Rossi, G, Goldoni, Ca, Rossi, F, De Gaetani, C, Benatti, P, Roncucci, L, Di Gregorio, C, Pedroni, M, Pezzi, A, Maffei, S, Mariani, F, Borsi, E, Cocchioni, M, Pascucci, C, Gennaro, V, Lazzarotto, A, Benfatto, L, Mazzucco, G, Montanaro, F, Traina, A, Carruba, G, Dolcemascolo, C, Staiti, R, Zarcone, M, Bisogno, G, Cuttini, M, De Rosa, M, Haupt, R, Soresina, A, Valsecchi, Mg, Fagioli, F, Bona, G, Dini, G, Conter, V, Biondi, A, Zecca, M, Fedeli, F, Massimino, M, Nespoli, L, Roncarolo, Mg, Basso, G, Cesaro, S, Memo, L, Bellettato, M, Tamaro, Paolo, Mascarin, M, Rosolen, A, Izzi, G, Paolucci, P, Borgna Pignatti, C, Vecchi, V, Abate, Me, D'Ambrosio, A, Favre, C, Caniglia, M, Pierani, P, Felici, L, Visani, G, Fioritoni, G, Foà, R, Riccardi, R, Frega, G, Clerico, A, Casale, F, Poggi, V, Amendola, G, Filosa, A, Ladogana, S, Presta, G, Tornesello, A, De Mattia, D, Consarino, C, Nobile, F, Sperlì, D, D'Angelo, P, Marino, S, Cosmi, C, Mura, Rm, Battisti, L., Maso, L, Mosso, M, Cappello, T, Vercellino, P, Orengo, M, Celesia, M, Cercato, M, Artioli, M, Vitale, M, Rosa, M, Giurdanella, M, Rollo, P, Ruggeri, M, Romagna, R, Contrino, M, Ziino, A, Fanetti, A, Annulli, M, Gonzalez, M, Gentilini, M, Rosa, F, Petrinelli, A, Grazia, L, Cin, A, Fiore, A, Limina, R, Lavecchia, A, Cascio, M, Brucculeri, M, Sini, G, de Leon, M, Goldoni, C, Valsecchi, M, Roncarolo, M, Tamaro, P, Abate, M, Mura, R, Battisti, L, AIRTUM Working Group[.., Enrica Borsi, ], CCM, and AIEOP Working Group

Subjects
Male, Parents, cancer incidence, Adolescent, Epidemiology, Aftercare, cancer in adolescents, Young Adult, children, Neoplasms, cancer in children, Italian population, cancer, Cluster Analysis, Humans, Registries, Survivors, Age of Onset, Mortality, Child, cancer survival, Incidence, Environmental and Occupational Health, Infant, Newborn, Disease Management, Infant, pediatric, Survival Rate, Italy, Child, Preschool, Public Health, Environmental and Occupational Health, Female, Public Health, Morbidity, Forecasting
Abstract

OBJECTIVES: This study describes up-to-date cancer incidence and survival in Italian paediatric and adolescent patients, based on data collected by the network of Italian cancer registries (AIRTUM). It updates the monograph published on the same topic in 2008. The main objective of this monograph is to present the statistics according to standard rigorous epidemiological methods and disseminate them to a wide range of readers, including the lay public. Given the deep impact of the 2008 monograph on the general public, in this update we complement descriptive statistics with additional data and commentaries on issues of importance for public health, in order to provide unambiguous criteria on how to interpret the statistics. The study is the result of the collaboration between AIRTUM and AIEOP (Italian Association of Paediatric Haematology and Oncology) with contributions from interested parties, including representatives of parent associations. The monograph is divided into three parts. The first part presents incidence rates, survival probabilities, and time trends, by sex, age, geographical area, and cancer site or type, by means of tables and graphs as in the previous monograph, to facilitate direct comparisons. Four articles summarize and comment the results. The second part uses data from AIRTUM and AIEOP to outline patient management and health care issues; it includes estimates of the number of new cases in the next decade and of young adults living after a paediatric cancer diagnosis. Health organizational aspects of treatment services for paediatric patients, based on the AIEOP database, are also discussed, along with long-term complications in cured patients. The third section describes the changes in mortality trends due to improving therapies and healthcare services, and discusses risk factors and prevention of childhood cancer, late adverse events in cured patients, and other related issues. MATERIAL AND METHODS: Data herein presented were provided by AIRTUM population-based cancer registries, covering 47%of the Italian population below age 20 years, in the period 2003-2008. Quality of cancer registration in Italy is elevated, with high proportions of microscopically verified diagnoses (91%in the 0-14 years age group and 96% between 15 and 19 years of age) and a very small proportion of cases collected through death certificate only (0.1%).The proportion of cases in diagnostic groups XI (other malignant epithelial neoplasms) and XII (other and unspecified neoplasms) of the International Classification for Childhood Cancer (ICCC), based on the third revision of the International Classification of Diseases for Oncology (ICD-O-3), were 7.0% in the 0-14 years age group and 26.0%in the 15-19 years age group.The ratio between mortality and incidence was 17.7% in both children and adolescents. Detailed results are presented in 24 fact sheets for the 12 major ICCC-3 diagnostic groups and 10 sub-groups of special interest; the series is completed by a sheet on all malignant tumours and one on all tumours including non-malignant neoplasms of the central nervous system. All sheets include results for three age groups (0-14, 15-19, and 0-19 years) and are followed by two commentaries on incidence in the recent period, one on trends and the other on survival. Incidence rates were age-standardized on the European population and presented per million children. Incidence rates are also presented by age group, sex, and geographical area. Incidence trends were evaluated for two periods, 1988-2008 and 1998-2008, using estimated annual percent changes, and survival estimates were calculated by age and period. Indicators and corresponding 95% confidence intervals are shown in forms of graphics and tables at the end of the monograph and online at http://www.registri-tumori.it. Geographical analyses were conducted rearranging cancer registries into four macroareas (North-West, North-East, Centre, and South and Islands). Age groups were the same used in descriptive studies on children worldwide (0, 1-4, 5-9, 10- 14 years for paediatric tumours and 15-19 years for adolescents). Incidence trend analyses included cancer registries with three or more years of registration in the 5-year period, using Poisson regression models. Observed survival was computed according to the Kaplan-Meier method. The estimate of expected cases in the next decade was based on observed incidence rates in the most recent period, extended to the Italian estimated population of children and adolescents in the periods 2011-2015 and 2016-2020. The AIEOP database (Modello 1.01) allowed us to compare the number of patients treated and followed-up in specialized centres with expected cases based on AIRTUM estimates. The AIEOP database also provided information regarding health care migration throughout Italian regions and the number of foreign (immigrated) children treated in Italian AIEOP centres. RESULTS: In the period 2003-2008, 31 cancer registries reported 4,473 incident malignant neoplasms, 2,855 in children and 1,618 in adolescents. Cancer incidence rates were 164 cases per million in children aged 14 years or below and 269 cases per million in patients aged 15-19 years. Limited geographical variations emerged. In children (0-14 years) a significant increase in malignant cancer incidence was observed until 1997 (APC: +3.2%), followed by a plateau (APC: -1.1%not statistically significant).Until the late Nineties, a statistically significant increase was also observed in the incidence of all leukaemias in males (APC: +5.7%), lymphoid leukaemias (APC: +5.6%), representing 80% of all leukaemias, Hodgkin and non- Hodgkin lymphomas (APC: +6.3%). A significant decrease emerged for lymphoid leukaemia starting in 1995 (APC: -1.9%), while no substantial change in cancer incidence rates was observed in the last decade of observation for all malignant neoplasms and lymphomas. In addition, no variation emerged for malignant (according to the most recent classification) central nervous system (CNS) neoplasms, while an annual increase of 1.8% (significant) was observed in the period 1988-2008, when non-malignant tumours were included. Increases in cancer incidence were observed throughout the study period for neuroblastoma (APC: +1.9%) and epithelial tumours or melanoma (APC: +4.1%). In the period 1998-2008, in addition to lymphoid leukaemias, a significant decrease was observed for all malignant neoplasms, lymphomas in girls, CNS tumours (males and females), and renal tumours in girls, while no increases were observed in this age group. In adolescents (15-19 years) between 1988 and 2008, a significant increase in incidence rates was observed (APC: +2.0%) for all malignant neoplasms, all lymphomas (APC: +2.9%; in particular Hodgkin lymphoma, APC: +3.6%), thyroid cancer (APC: +6.1%), and melanoma (APC: +8.1%). Conversely, lymphoid leukaemia is the only neoplasm showing a long-term decrease in adolescents. Recent trends (1998-2008) confirm the long-term increases only for all malignant neoplasms in girls and thyroid cancer (APC: +7.9%, boys and girls), while a decrease in bone tumour incidence emerged in girls, albeit based only on 46 cases. Cancer mortality in children showed a persistent decrease for all neoplasms and even for more frequent cancer sites or types, and mortality rates for cancer were three-fold higher in the early Seventies than in 2008. In addition, five-year survival after cancer diagnosis increased in the last three decades and was still increasing in the period 2003- 2008, reaching 82% in children and 86% in adolescents. In the period 2008-2010, 4,488 children (0-14 years) were treated in one of the AIEOP clinical centres and we estimate, based on the above-presented incidence rates, that they represented 92% of all cancer cases in Italy. However, in adolescents, the proportion of patients treated in AIEOP centres was only 25%. A migration of patients living in the South of Italy to Central and Northern Italy emerged from AIEOP information. The expected number of cancer cases in children aged between 0 and 14 years of age is approximately 7,000 in the period 2016- 2020, while the corresponding figure for adolescents between 15 and 19 years of age is 4,000, with no relevant variation in comparison with the previous five-year period. COMMENTS: The present findings update descriptive cancer epidemiology in children and adolescents in Italy based on data provided by an extensive network of general and specialized population-based cancer registries. Data obtained from cancer registries are supplemented by additional information collected by specialized clinical AIEOP centres and mortality reports collected by the National Institute of Statistics (ISTAT). Incidence rates reported in Italy were slightly higher in comparison to other developed Countries, but relatively consistent between different Italian areas. Our results also showed that the significant increase in cancer incidence observed until the end of Nineties has halted, with the exception solely of thyroid cancer in adolescents. Efficacy of therapeutic protocols has improved constantly since the Seventies, and recent findings confirm this trend in all age groups and, in particular, for rarer tumours and cancer types that have very poor prognosis. Findings derived from cross-analysis with AIEOP data suggest that it is possible to further improve the efficiency of our healthcare system, in particular for adolescents; migration can be reduced with a more rational use of hospitals throughout Italy.

9. Noonan syndrome-like disorder with loose anagen hair: a second case with neuroblastoma

Author

Garavelli, Livia, Cordeddu, Viviana, Errico, Stefania, Bertolini, Patrizia, Street, Maria Elisabeth, Rosato, Simonetta, Pollazzon, Marzia, Wischmeijer, Anita, Ivanovski, Ivan, Daniele, Paola, Bacchini, Ermanno, Lombardi, Alfonsa Anna, Izzi, Giancarlo, Biasucci, Giacomo, Del Rossi, Carmine, Corradi, Domenico, Cazzaniga, Giovanni, Dominici, Carlo, Rossi, Cesare, De Luca, Alessandro, Bernasconi, Sergio, Riccardi, Riccardo, Legius, Eric, Tartaglia, Marco, Garavelli, L, Cordeddu, V, Errico, S, Bertolini, P, Street, M, Rosato, S, Pollazzon, M, Wischmeijer, A, Ivanovski, I, Daniele, P, Bacchini, E, Lombardi, A, Izzi, G, Biasucci, G, Del Rossi, C, Corradi, D, Cazzaniga, G, Dominici, C, Rossi, C, De Luca, A, Bernasconi, S, Riccardi, R, Legius, E, and Tartaglia, M

Subjects
Male, Acute leukemia, SHOC2, MED/03 - GENETICA MEDICA, Noonan Syndrome, Infant, Newborn, cancer predisposition, mazzanti syndrome, Brain tumor, Neuroblastoma, Genetic, RASopathie, acute leukemias, noonan-like syndrome with loose anagen hair, brain tumors, Humans, RASopathies, neuroblastoma, rhabdomyosarcoma, Genetics (clinical), Human
Abstract

Noonan-like syndrome with loose anagen hair (NSLH), also known as Mazzanti syndrome, is a RASopathy characterized by craniofacial features resembling Noonan syndrome, cardiac defects, cognitive deficits and behavioral issues, reduced growth generally associated with GH deficit, darkly pigmented skin, and an unique combination of ectodermal anomalies. Virtually all cases of NSLH are caused by an invariant and functionally unique mutation in SHOC2 (c.4A>G, p.Ser2Gly). Here, we report on a child with molecularly confirmed NSLH who developed a neuroblastoma, first suspected at the age 3 months by abdominal ultrasound examination. Based on this finding, scanning of the SHOC2 coding sequence encompassing the c.4A>G change was performed on selected pediatric cohorts of malignancies documented to occur in RASopathies (i.e., neuroblastoma, brain tumors, rhabdomyosarcoma, acute lymphoblastic, and myeloid leukemia), but failed to identify a functionally relevant cancer-associated variant. While these results do not support a major role of somatic SHOC2 mutations in these pediatric cancers, this second instance of neuroblastoma in NSLAH suggests a possible predisposition to this malignancy in subjects heterozygous for the c.4A>G SHOC2 mutation.

Published
2015

10. Rotavirus and not age determines gastroenteritis severity in children: a hospital-based study

Author

Nicola Principi, Paola Pecco, Lucina Titone, Raffaele Virdis, Serenella Arista, Giancarlo Izzi, Massimo Fontana, Eugenia Bruzzese, Fabio Albano, Alfredo Guarino, Antonino Bella, Antonio Cascio, F., Albano, Bruzzese, Eugenia, A., Bella, A., Cascio, L., Titone, S., Arista, G., Izzi, R., Virdi, P., Pecco, N., Principi, M., Fontana, Guarino, Alfredo, ALBANO F, BRUZZESE E, BELLA A, CASCIO A, TITONE L, ARISTA S, IZZI G, VIRDIS R, PECCO P, PRINCIPI N, FONTANA M, and GUARINO A

Subjects
Male, Rotavirus, Pediatrics, medicine.medical_specialty, Reoviridae, macromolecular substances, medicine.disease_cause, Gastroenterology, Severity of Illness Index, Rotavirus Infections, Hospital based study, Age Distribution, Internal medicine, medicine, Humans, Children, Gastroenteriti, Dehydration, biology, business.industry, musculoskeletal, neural, and ocular physiology, Infant, Newborn, Infant, Length of Stay, Rotaviru, biology.organism_classification, Diarrhoea, Gastroenteritis, Diarrhea, nervous system, El Niño, Italy, Child, Preschool, Pediatrics, Perinatology and Child Health, Female, medicine.symptom, business
Abstract

Background The severity of childhood gastroenteritis is generally believed to be age-related rather than aetiologyrelated. Rotavirus-induced gastroenteritis is more severe than gastroenteritis caused by other enteric pathogens and is also age-related. We thus addressed the question of whether the increased severity of rotavirus-induced gastroenteritis is related to age or to features intrinsic to the agent. Study design In this multicentre, hospital-based, prospective survey, we evaluated the severity of diarrhoea in rotavirus-positive and rotavirus-negative children up to 4 years of age. Severity was assessed with a score in four groups of age-matched children. Results Rotavirus was detected in 381 of 911 children. Disease severity was evaluated in 589 cases for which clinical data were complete. The rotavirus-positive and rotavirus-negative groups differed with regards to diarrhoea duration, hospital stay, degree of dehydration and the number of episodes of vomiting. Gastroenteritis was more severe in rotavirus-positive than in rotavirus-negative children. In contrast, none of the main severity parameters differed in the four age groups, irrespective of the presence of rotavirus. Conclusions These data provide the evidence that aetiology and not age determines diarrhoeal severity. The demonstration that diarrhoea was more severe in rotavirus-positive children supports the need for a rotavirus vaccine and for studies that address the duration of vaccine protection.

Published
2006

11. Topotecan plus ifosfamide in patients with platinum refractory advanced/metastatic non-small cell lung cancer: A phase II trial

Author

Nicola Silvestris, Giuseppe Nettis, Domenico Galetta, Domenico Germano, Giovanni Izzi, Marianna Giampaglia, Michele Guida, Grazia Cassano, Giuseppe Colucci, Cosimo Brunetti, Vito Lorusso, Vittorio Gebbia, M. Spada, LORUSSO V, GEBBIA V, SPADA M, GUIDA M, CASSANO G, BRUNETTI C, GERMANO D, NETTIS G, IZZI G, GALETTA D, GIAMPAGLIA M, SILVESTRIS N, and COLUCCI G

Subjects
Adult, Male, Cancer Research, medicine.medical_specialty, Lung Neoplasms, Neutropenia, medicine.medical_treatment, Gastroenterology, chemistry.chemical_compound, Carcinoma, Non-Small-Cell Lung, Internal medicine, Antineoplastic Combined Chemotherapy Protocols, medicine, Humans, Ifosfamide, Infusions, Intravenous, Lung cancer, Survival rate, Aged, Platinum, Chemotherapy, business.industry, Alopecia, Anemia, General Medicine, Middle Aged, medicine.disease, Thrombocytopenia, Carboplatin, Squamous carcinoma, Surgery, Oxaliplatin, Treatment Outcome, Oncology, chemistry, Drug Resistance, Neoplasm, Female, Topotecan, business, medicine.drug
Abstract

A number of second line treatments have been proposed in patients with advanced pretreated non-small cell lung cancer (NSCLC). However, either single agents or two or three drug combinations achieved very poor results with no superiority of any combination over monotherapy. We have treated 42 patients (30 males) affected by advanced/metastatic NSCLC progressing during front line cisplatin-based chemotherapy with a combination of topotecan (1.2 mg/m2) plus ifosfamide (1200 mg/m2) for 3 consecutive days every 3 weeks. The median age was 63 years (range 43-76); cell types were: squamous carcinoma (n=17), adenocarcinoma (n=16), large cell carcinoma (n=3), broncho-alveolar carcinoma (n=2) and undifferentiated carcinoma (n=4). All patients were treated with a platinum containing chemotherapy: 39 patients with cisplatin, 2 patients with carboplatin and 1 patient with oxaliplatin, respectively. The ECOG PS was 0 in 8 patients (19%), 1 in 11 patients (26%), and 2 in 23 patients (55%). The median number of courses administered was 3 (range 1-8). Grade 3-4 neutropenia was the dose limiting toxicity, observed in 36% of patients. Moreover, grade 3-4 anemia and thrombocytopenia were observed in 17% and in 12% of patients, respectively. One PS 2 patient died of grade 4 hematological toxicity after the first cycle. No complete response was observed. Six (14.2%) subjects obtained a partial response (PR). In addition, 1 (2.4%) minimal response (MR) plus 14 (34%) stable diseases (SD) and 21 (51%) progressive diseases (PD) were observed. Median time to disease progression and median survival were 9 weeks (range 1-13) and 26 weeks (range 1-91+), respectively. The 1-year survival rate was 14%. Combination of topotecan and ifosfamide demonstrated antitumor activity in patients with relapsing or refractory NCSLC with a modest side effect profile and an overall disease control (PR + MR + SD) of 50.7%. Nevertheless, the still low response rate and the shortness of median survival indicates the need for more effective second line treatments in this disease.

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