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3. 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

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. Osteonecrosis: An emerging complication of intensive chemotherapy for childhood acute lymphoblastic leukemia

Author

Maurizio Arico', Pinta Boccalatte, M. F., Silvestri, D., Barisone, E., Messina, C., Chiesa, R., Santoro, N., Tamaro, P., Lippi, A., Gallisai, D., Basso, G., Rossi, G., Arico, M, Boccalatte, Mf, Silvestri, D, Barisone, E, Messina, C, Chiesa, R, Santoro, N, Tamaro, Paolo, Lippi, A, Gallisai, D, Basso, G, and DE ROSSI, G.

Subjects
Male, Adolescent, Antineoplastic Agents, Hormonal, Incidence, Osteonecrosis, Precursor Cell Lymphoblastic Leukemia-Lymphoma, Dexamethasone, Treatment Outcome, Risk Factors, Humans, Prednisone, Drug Therapy, Combination, Female, Child
Abstract

Osteonecrosis (ON) is a potentially disabling complication of combination chemotherapy including high doses of steroids. The incidence and main risk factors for symptomatic ON have been investigated in a large group of children treated with high-dose steroids, prednisone and dexamethasone for childhood acute lymphoblastic leukemia (ALL).From May 1995 to December 1999, 1421 patients18 years old, with newly diagnosed non-B ALL, were registered in the AIEOP-ALL 95 study. Their data were reviewed to identify patients who developed symptomatic ON. For those who were positively identified additional data were requested concerning ON-related symptoms, treatment and outcome.Overall, 15 of the 1421 patients developed symptomatic ON (1.1%) in a total of 29 sites. The estimated 5-year cumulative risk for clinically diagnosed ON was 1.6% (SE 0.4). The incidence was significantly higher among females (p=0.01) and older patients, with a peak rate of 7.4% (2.3) among those aged 10 to 17 years (p0.0001). When the two factors, i.e. age and gender were combined, there was a striking increase in the risk among female patients aged 10 to 17 years. The median time between the diagnosis of ALL and that of ON was 17 months (range 8-45). The hip was the most frequently involved (19/29) site.Symptomatic ON occurred in only 1.1% of patients treated with BFM-type, intensive chemotherapy for childhood ALL. Female adolescents appear to be the subset of patients with the highest risk of ON, especially when categorized as having high risk leukemia and thus administered higher cumulative doses of dexamethasone.

Published
2003

13. Guide lines for the auxlogical follow-up of thalassemic patients[Linee guida per il follow-up auxologico del paziente talassemico]

Author

Caruso Nicoletti, M., De Sanctis, V., Anastasi, S., Cavagnini, F., Cavallo, L., Cilla, A., Cisternino, M., DE LUCA, Filippo, De Simone, M., Di Gregorio, F., Einaudi, S., Galati, M. C., Gallisai, D., Erardi, C., Malizia, R., Mangiagli, A., Meo, A., Pasquino, A. M., Pintor, C., Ponzi, G., Quarta, G., Romondia, A., Ruggiero, L., Saviano, A., Sacco, M., and Stefano, I.

Published
1997

14. Incidence and outcome of Yersinia enterocolitica infection in thalassemic patients

Author

Cherchi, G. B., Cossellu, S., Pacifico, Lucia, Gallisai, D., Zanetti, S., Ranucci, A., Fadda, G., and Chiesa, C.

Subjects
Adult, Male, thalassemia, Adolescent, Yersinia Infections, Ceftriaxone, beta-Thalassemia, yersinia infection, Infant, Bacteremia, Enteritis, Anti-Bacterial Agents, Cephalosporins, children, Child, Preschool, Trimethoprim, Sulfamethoxazole Drug Combination, Humans, Female, Prospective Studies, Child, Yersinia enterocolitica
Published
1995

16. Disseminated poor-risk neuroblastoma. Results of an 'intensive' protocol (AIEOP NB-85). [Neuroblastoma disseminato a cattiva prognosi. Risultati di un protocollo intensivo (AIEOP NB-85)]

Author

Corciulo, P, Deb, G, Mazzei, A, Bisogno, G, Surico, Gm, Tonello, M, Garaventa, A, Arrighini, A, Lumia, F, Zanazzo, G, Gallisai, D, Massolo, F, Cornelli, Pe, Targhetta, R, DI CATALDO, Andrea, Ragusa, R, Acquaviva, A, Bernini, G, Fiorillo, A, Santoro, N, Ivani, G, Zucchetti, P, Cominetti, M, Viscardi, E, Castagnola, E, and De Bernardi, B.

Published
1993

17. Stage IV-S neuroblastoma. 76 Cases treated by the Cooperative Group for Neuroblastoma of the Italian Association of Paediatric Haematology and Oncology [NEUROBLASTOMA STADIO IV-S. 76 CASI TRATTATI DAL GRUPPO COOPERATIVO NEUROBLASTOMA DELL'ASSOCIAZIONE ITALIANA DI EMATOLOGIA ED ONCOLOGIA PEDIATRICA]

Author

De Bernardi, B, Mancini, A, Carli, M, Casale, F, Bagnulo, S, Corciulo, P, Deb, G, Donfrancesco, A, Rosito, P, Arrighini, A, Provenzi, P, DI CATALDO, Andrea, Fabbretti, G, Felici, L, Izzi, Gc, Provenzano, G, Massolo, F, Mazzei, A, Fiorillo, A, Santoro, R, Targhetta, R, Tamaro, P, Fagnani, Am, Gallisai, D, Conte, M, Tonello, M, Cordero di Montezemolo LRiv Ital Pediatr 18:414 426, and 1992

Published
1992

24. Costs, quality of life, treatment satisfaction and compliance in patients with beta-thalassemia major undergoing iron chelation therapy: the ITHACA study.

Author

Scalone L, Mantovani LG, Krol M, Rofail D, Ravera S, Bisconte MG, Borgna-Pignatti C, Borsellino Z, Cianciulli P, Gallisai D, Prossomariti L, Stefàno I, Cappellini MD, Scalone, Luciana, Mantovani, Lorenzo G, Krol, Marieke, Rofail, Diana, Ravera, Simona, Bisconte, Maria Grazia, and Borgna-Pignatti, Caterina

Abstract

Objectives: Iron chelation treatment (ICT) in beta-thalassemia major (beta-TM) patients undergoing blood transfusions can cause low satisfaction, low compliance, with possible negative consequences on treatment success, patients' wellbeing, and costs. The purpose was to estimate the societal burden attributable to beta-TM in terms of direct and indirect costs, health-related quality-of-life (HRQoL), satisfaction and compliance with ICT in patients undergoing transfusions and ICT.Research Design and Methods: The naturalistic, multicenter, longitudinal Italian-THAlassemia-Cost-&-Outcomes-Assessment (ITHACA) cost-of-illness study was conducted involving patients of any age, on ICT for at least 3 years, who were enrolled at 8 Italian Thalassemia Care Centers. Costs were estimated from the societal perspective, quantified with tariffs, prices, or net earnings valid in 2006.Results: One-hundred and thirty-seven patients were enrolled (median age = 28.3, 3-48 years, 49.6% male) and retrospectively observed for a median of 11.6 months. Mean direct costs were euro1242/patient/month, 55.5% attributable to ICT, 33.2% attributable to transfusions. Relevant quantity and quality of productivity was lost. Both physical and mental components of HRQoL were compromised. Little difficulties remembering to take ICT and positive satisfaction with the perceived effectiveness of therapy were declared, but not good levels of satisfaction with acceptance, perception of side effects and burden of ICT.Conclusions: The management of beta-TM patients undergoing transfusions and ICT is efficacious, although costly, but overall benefits were not always perceived as optimal by patients. Efforts must be focused to improve patients' acceptance and satisfaction with their therapy; this would contribute to a better compliance and hence an increase in treatment effectiveness and patients' overall wellbeing, with expected improved allocation of human and economic resources. [ABSTRACT FROM AUTHOR]

Published
2008
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26. Interferon-α Therapy in Sicilian and Sardinian Polytransfused Thalassaemic Patients with Chronic Hepatitis C.

Author

Pizzarelli, G., Di Gregorio, F., Romeo, M.A., Carboni, F., Gallisai, D., Solinas, A., Malaguarnera, M., and Musumeci, S.

Subjects
INTERFERONS, THALASSEMIA treatment, HEPATITIS treatment
Abstract

Objective: Our study was designed to evaluate the effects of 2 dosage schedules of recombinant interferon (IFN)-α (IFNα-2a and IFNα-2b) in reducing serum ALT and eradicating serum hepatitis C virus (HCV) RNA in β-thalassaemic patients with chronic hepatitis C. Design: 38 Sicilian β-thalassaemic patients (22 males and 16 females) received intramuscular IFNα-2a (Roferon-A; Roche) 5 MU/m 3 times weekly for 6 months, followed by 3 MU/m 3 times weekly for a further 6 months. 13 Sardinian β-thalassaemic patients (7 males and 6 females) received intramuscular IFNα-2b (Intron®; Schering-Plough) 3 MU/m 3 times weekly for 12 months. Parallel control groups (n = 20 and n = 8, respectively) did not receive IFNα. All patients received continuous subcutaneous desferoxamine infusion. Results: 24 (63%) Sicilian patients had a positive clinical response to IFNα-2a therapy. Two different patterns of response were apparent: (i) early and progressive decrease in ALT values until stable normalisation; and (ii) slower reduction of ALT values, which fluctuated on the way to normalisation. Five (21%) patients relapsed during the 12-month follow-up period. ALT levels decreased early in 5 (38%) Sardinian patients and one patient (20%) relapsed during the 12-month follow-up period. In the control groups, ALT values spontaneously normalised in 3 (10%) untreated patients. None of the patients treated with IFNα developed anti-IFNα antibodies. Viral clearance was demonstrated in 19 (50%) of 38 patients in the Sicilian group and 4 of 13 patients (31%) in the Sardinian group. Conclusion: Treatment with intramuscular recombinant IFNα-2a 5 MU/m 3 times weekly for 6 months, followed by 3 MU/m 3 times weekly for 6 months, appeared to be more effective than intramuscular IFNα-2b 3 MU/m 3 times weekly for 12 months. [ABSTRACT FROM AUTHOR]

Published
1999
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37. The unreliability of mean corpuscular volume and mean cellular hemoglobin determinations in the diagnosis of alpha-thalassemia in newborn infants.

Author

Meloni, T., Solinas, L., Erre, S., Dore, A., Gallisai, D., and Porqueddu, F.

Abstract

Mean corpuscular volume (MCV) and mean cellular hemoglobin (MCH) were determined by means of a Hemalog 8/90 electronic counter in 51 full-term newborn infants with alpha-thalassemia-2 and 15 with alpha-thalassemia-1, as well as in 150 normal newborn infants. The mean MCV and MCH values were 92 fl +/- 06 and 33.26 pg +/- 2.22 in the normal newborn infants, 82 fl +/- 07 and 29.40 pg +/- 2.60 in the alpha-thalassemia-2 subjects, and 73 fl +/- 06 and 26.7 +/- 2.05 in the alpha-thalassemia-1 subjects. Four of the 150 normal newborn infants had MCV's < 79 fl and MCH's < 29.00 pg whereas 5 of the alpha-thalassemic subjects had MCV's > 90 fl and MCH's > 32.00 pg. We conclude that MCV and MCH determinations are unreliable in the diagnosis of alpha-thalassemia in the neonatal period. [ABSTRACT FROM AUTHOR]

Published
1980
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38. The incidence and natural course of transfusion-associated GB virus C/hepatitis G virus infection in a cohort of thalassemic patients. The Cooleycare Cooperative Group

Author

Daniele Prati, Zanella A, Bosoni P, Rebulla P, Farma E, De Mattei C, Capelli C, Mozzi F, Gallisai D, Magnano C, Melevendi C, and Sirchia G

Subjects
Adult, Male, Adolescent, Hepatitis, Viral, Human, Flaviviridae, beta-Thalassemia, Infant, Newborn, Infant, Transfusion Reaction, RNA-Directed DNA Polymerase, Polymerase Chain Reaction, Immunoenzyme Techniques, Risk Factors, Child, Preschool, Humans, RNA, Viral, Female, Longitudinal Studies, Prospective Studies, Child
Abstract

To evaluate the risk of transmitting blood-borne GB virus C/hepatitis G virus (GBV-C/HGV) and to define the natural course of infection, we performed a prospective study in a cohort of multitransfused beta-thalassemics during a 6-year follow-up period. We analyzed serum samples of 150 patients collected at 3-year intervals from 1990 to 1996. GBV-C/HGV RNA was determined by reverse transcriptase-polymerase chain reaction and antibodies to E2-protein by an enzyme immunoassay. At baseline, 14.5% of patients had viremia and 18.5% anti-E2. None of the patients with anti-E2 in 1990 subsequently became viremic. Of the 100 GBV-C/HGV RNA-, anti-E2- patients, 10 acquired infection during follow-up, as indicated by positivity of GBV-C/HGV RNA (n = 2), anti-E2 (n = 7), or both markers (n = 1) in 1996. The incidence was 1.7 per 100 person-years (95% confidence interval [CI], 0.8 to 3). Since approximately 19,000 blood units were transfused to these patients during follow-up, the risk of infection was 5.3 in 10,000 units (95% CI, 2 to 8.5). Six of 22 viremic patients cleared the virus during follow-up; 4 of them became anti-E2+. Twelve of 28 patients lost anti-E2 reactivity during follow-up. In conclusion, more than 25% of infections resolve within 6 years; the presence of anti-E2 seems to be protective against infection. Anti-E2 reactivity may decrease with time.

45. beta-Thalassemia trait and hyperbilirubinemia in G-6-PD deficient newborn infants.

Author

Meloni, T, Erre, S, Gallisai, D, and Cutillo, S

Abstract

Hb A2 was determined in 50 subjects with erythrocyte G-6-PD deficiency who presented with hyperbilirubinemia in the neonatal period and in 100 non-hyperbilirubinemic G-6-PD deficient newborn infants, at the age of 12 months or more. Six subjects in the first group and 13 in the second were found to be carriers of the beta-thalassemia trait. Statistical analysis of the data did not show any significant difference between the two groups. It seems that the beta-thalassemia trait does not provide any protection against neonatal hyperbilirubinemia associated with G-6-PD deficiency. [ABSTRACT FROM AUTHOR]

Published
1980

48. Plasma oxysterols in normal and cholestatic children as indicators of the two pathways of bile acid synthesis

Author

Carla Colombo, Marzia Galli Kienle, Maria Graziella Mela, Emma De Fabiani, M.L. Melzi, Marina Del Puppo, Domenico Pietro Paolo Gallisai, Donatella Caruso, Andrea Crosignani, Crosignani, A, DEL PUPPO, M, De Fabiani, E, Caruso, D, Gallisai, D, Mela, M, Melzi, M, Kienle, M, and Colombo, C

Subjects
Adult, Male, medicine.medical_specialty, 27-hydroxycholesterol, Adolescent, Clinical Biochemistry, Biochemistry, Intestinal absorption, Bile Acids and Salts, chemistry.chemical_compound, Liver disease, Internal medicine, polycyclic compounds, medicine, Humans, 7α-Hydroxycholesterol, Child, Cholestasis, Cholesterol, Biochemistry (medical), 7alpha-hydroxycholesterol, Infant, General Medicine, medicine.disease, Hydroxycholesterols, Endocrinology, chemistry, Child, Preschool, 27-Hydroxycholesterol, Chronic Disease, Bile acid synthesi, lipids (amino acids, peptides, and proteins), Female, Cholestatic liver disease, Liver function, Bile acid synthesis, Pediatric cholestasis
Abstract

Background: No information is available on the hepatic and extrahepatic pathways of bile acid synthesis in normal children and in pediatric cholestatic liver diseases. Methods: To explore the changes of the two pathways of bile acid synthesis during development, plasma concentrations of 7α-hydroxycholesterol and 27-hydroxycholesterol were measured in 50 healthy children (1 month-14 years) and compared to 18 adult controls. We also measured plasma oxysterols in 31 patients with pediatric cholestatic liver disease. Results: A progressive increase of plasma concentrations of both 27-hydroxycholesterol and 7α-hydroxycholesterol was found with age. In children with cystic fibrosis-associated liver disease plasma concentrations of 27-hydroxycholesterol were significantly lower compared to age-matched controls (5.6 ± 0.5 vs. 12.8 ± 1.1 μg/dl; p < 0.001) and paralleled significantly lower concentrations of total cholesterol. In infants with biliary atresia plasma concentrations of 27-hydroxycholesterol were significantly higher compared to age-matched controls (8.8 ± 0.8 vs. 4.4 ± 0.6 μg/dl, p < 0.001) paralleling significantly higher concentrations of total cholesterol while 7α-hydroxycholesterol resulted significantly lower (1.2 ± 0.2 vs. 2.3 ± 0.3 μg/100 mg of total cholesterol; p = 0.011). Conclusions: Our data suggest that both pathways of bile acid synthesis reach a state of maturity only after the age of 4 years and are significantly influenced also in children by liver function and intestinal absorption of cholesterol. © 2008 Elsevier B.V. All rights reserved.

Published
2008

49. Rituximab-based immunosuppression for autoimmune haemolytic anaemia in infants.

Author

Svahn J, Fioredda F, Calvillo M, Molinari AC, Micalizzi C, Banov L, Schmidt M, Caprino D, Marinelli D, Gallisai D, and Dufour C

Subjects
Anemia, Hemolytic, Autoimmune immunology, Antibodies, Monoclonal, Murine-Derived, Cyclosporine therapeutic use, Female, Follow-Up Studies, Hemolysis drug effects, Humans, Infant, Male, Remission Induction, Rituximab, Treatment Outcome, Anemia, Hemolytic, Autoimmune drug therapy, Antibodies, Monoclonal therapeutic use, Immunosuppressive Agents therapeutic use
Abstract

We report a case series of four infants with severe autoimmune haemolytic anaemia (AIHA) who responded to treatment with rituximab and cyclosporine after having failed first line therapy with high-dose steroid (prednisolone 4-8 mg/kg/d). Rituximab was started at 11-90 d from onset due to continued haemolysis; three infants also received cyclosporine A. Three of four infants reached complete response, defined as normal haemoglobin, reticulocytes and negative indices of haemolysis, at 7-21 months from diagnosis. In long-term follow-up two infants remained disease-free with normal immunology, one had undefined immunodeficiency and one had autoimmune lymphoproliferative syndrome.

Published
2009
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50. Plasma oxysterols in normal and cholestatic children as indicators of the two pathways of bile acid synthesis.

Author

Crosignani A, Del Puppo M, De Fabiani E, Caruso D, Gallisai D, Mela MG, Melzi ML, Galli Kienle M, and Colombo C

Subjects
Adolescent, Adult, Child, Child, Preschool, Cholesterol metabolism, Chronic Disease, Female, Humans, Hydroxycholesterols metabolism, Infant, Male, Bile Acids and Salts biosynthesis, Cholestasis blood, Cholesterol blood, Hydroxycholesterols blood
Abstract

Background: No information is available on the hepatic and extrahepatic pathways of bile acid synthesis in normal children and in pediatric cholestatic liver diseases., Methods: To explore the changes of the two pathways of bile acid synthesis during development, plasma concentrations of 7alpha-hydroxycholesterol and 27-hydroxycholesterol were measured in 50 healthy children (1 month-14 years) and compared to 18 adult controls. We also measured plasma oxysterols in 31 patients with pediatric cholestatic liver disease., Results: A progressive increase of plasma concentrations of both 27-hydroxycholesterol and 7alpha-hydroxycholesterol was found with age. In children with cystic fibrosis-associated liver disease plasma concentrations of 27-hydroxycholesterol were significantly lower compared to age-matched controls (5.6+/-0.5 vs. 12.8+/-1.1 microg/dl; p<0.001) and paralleled significantly lower concentrations of total cholesterol. In infants with biliary atresia plasma concentrations of 27-hydroxycholesterol were significantly higher compared to age-matched controls (8.8+/-0.8 vs. 4.4+/-0.6 microg/dl, p<0.001) paralleling significantly higher concentrations of total cholesterol while 7alpha-hydroxycholesterol resulted significantly lower (1.2+/-0.2 vs. 2.3+/-0.3 microg/100 mg of total cholesterol; p=0.011)., Conclusions: Our data suggest that both pathways of bile acid synthesis reach a state of maturity only after the age of 4 years and are significantly influenced also in children by liver function and intestinal absorption of cholesterol.

Published
2008
Full Text
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