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  • 1
    Language: English
    In: Transfusion (Philadelphia, Pa.), 2015-04, Vol.55 (4), p.736-747
    Description: Background Extracorporeal photopheresis (ECP) has been shown as active therapy for graft‐versus‐host disease (GVHD). Study Design and Methods The aim was to ascertain the role of ECP in 71 patients with steroid‐refractory or ‐dependent acute and chronic GVHD (aGVHD and cGVHD) with special focus on hematologic variables and GVHD staging classification. A total of 34 patients were treated for aGVHD and 37 for cGVHD. Results The overall response rate (ORR) for aGVHD was 65% and the complete aGVHD‐free survival was 50% (95% confidence interval [CI], 36%‐70%). The ORR for cGVHD response was 81% while the complete cGVHD‐free survival was 50% (95% CI, 34%‐73%). The aGVHD‐free survival was associated with aGVHD grading (Grade II 81%, Grade III 33%, and Grade IV 0%, p ≤ 0.00) and the absence of visceral involvement (77% vs. 33%, p = 0.03). The cGVHD‐free survival was associated with the female sex (67% vs. 25%, p = 0.01) and with the limited form according to the Seattle classification (67% vs. 20%, p = 0.003). No role for hematologic values or apheresis cell count was found, except for the cGVHD ORR (p = 0.037). Transplant‐related mortality and overall survival were associated with ECP response 0% versus 54% (p = 0.0001) and 77% versus 45% (p = 0.03) for aGVHD patients and 7% versus 14% (p = 0.02) and 73% versus 20% (p = 0.0003) for cGVHD patients, respectively. Conclusions While confirming a higher probability of GVHD responses for early GVHD, our study shows no role of hematologic values or apheresis cell count on GVHD response.
    Subject(s): Graft vs Host Disease - therapy ; Hematologic Neoplasms - therapy ; Peripheral Blood Stem Cell Transplantation - adverse effects ; Prospective Studies ; Humans ; Immunosuppressive Agents - therapeutic use ; Middle Aged ; Child, Preschool ; Male ; Adrenal Cortex Hormones - therapeutic use ; Cord Blood Stem Cell Transplantation - adverse effects ; Adult ; Female ; Photopheresis ; Child ; Drug Resistance ; Treatment Outcome ; Combined Modality Therapy ; Graft vs Host Disease - blood ; Graft vs Host Disease - drug therapy ; Hematologic Neoplasms - blood ; Adolescent ; Survival Analysis ; Biomarkers ; Bone Marrow Transplantation - adverse effects ; Graft vs Host Disease - prevention & control ; Aged ; Transplantation Conditioning ; Blood Group Incompatibility - epidemiology ; Index Medicus ; Hemapheresis
    ISSN: 0041-1132
    E-ISSN: 1537-2995
    Source: Alma/SFX Local Collection
    Source: © ProQuest LLC All rights reserved〈img src="https://exlibris-pub.s3.amazonaws.com/PQ_Logo.jpg" style="vertical-align:middle;margin-left:7px"〉
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  • 2
    Language: English
    In: Pediatric transplantation, 2020-11, Vol.24 (7), p.e13806-n/a
    Description: aGvHD remains a major obstacle to successful HSCT. We report our experience on steroid‐refractory aGvHD III and IV from 1989 to 2017. Ninety patients with aGvHD III or IV were stratified according to the HSCT year: 1989‐1998, 1999‐2007, and 2008‐2017 and to aGvHD extension (GvHD III vs IV) and finally the probability of OS, RI, and TRM was calculated accordingly. aGvHD III patients had a substantial improvement over time: day 100 OS raised from 64% (95% CI 39‐89) in the first cohort to 100% in the latest (P = .022), and it was mainly due to a reduction of TRM (it was 28% [95% CI 12‐65] in the first cohort to 0% in the latest (P = .01). The aGvHD IV patients did not present a significant improvement. Day 100 OS was 42% (95% CI 16‐68) in the first group and 54% (95% CI 25‐83) in the year 2008‐2017 (P = NS), and the day‐100 TRM was very similar (it was 57% [95% CI 36‐90] in the first cohort and 45% [95% CI 23‐89] in the latest (P = NS). We report significant improvements in OS and TRM in patients diagnosed with grade III aGvHD. Patients with the most severe aGvHD appear to have no or fewer benefits on long‐term outcomes.
    Subject(s): severe acute graft‐versus‐host disease ; children ; survival ; Index Medicus
    ISSN: 1397-3142
    E-ISSN: 1399-3046
    Source: Alma/SFX Local Collection
    Source: © ProQuest LLC All rights reserved〈img src="https://exlibris-pub.s3.amazonaws.com/PQ_Logo.jpg" style="vertical-align:middle;margin-left:7px"〉
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  • 3
    Language: English
    In: Bone marrow transplantation (Basingstoke), 2019-08, Vol.54 (8), p.1275-1280
    Description: To determine the current practices on the management of Adenovirus (ADV) infection after allogenic stem cell transplantation, a survey was undertook among EBMT centres. The response rate was 20% (91/446): 46% were adult, 44% were paediatric and 10% were mixed centres, respectively. The overall incidence of ADV infection was 7.1%: 4.1% in adult, 15.4% in paediatric, and 3.6% in mixed population. The determination of ADV-DNA in biological samples was used in 96% of centres; 58% of them monitored asymptomatic patients with a frequency of twice a week in 9%, once a week in 45%, every two weeks in 4% of centres. The treatment of ADV infection was mainly based on the administration of cidofovir (87%), being the schedule of 5 mg/kg/week with probenecid the most used, and the reduction of immunosuppression (84%). The threshold of ADV-DNAemia to start cidofovir in high-risk patients was most frequently 〉1000 copies/ml. Innovative treatments, such as brincidofovir and adoptive ADV-cytotoxic-T-lymphocytes, were used in 27% and 20% of centres, respectively. Almost all responding centres consider ADV infection serious enough to deserve testing asymptomatic or symptomatic patients. Cidofovir and reduction of immunosuppression represent the main therapeutic options but one fourth of responding centres experimented novel therapies.
    Subject(s): Care and treatment ; Usage ; Adenovirus diseases ; Analysis ; Stem cells ; Cidofovir ; Dosage and administration ; Transplantation ; Health surveys ; Risk factors ; Index Medicus ; Medicin och hälsovetenskap
    ISSN: 0268-3369
    ISSN: 1476-5365
    E-ISSN: 1476-5365
    Source: Nature Open Access
    Source: Elektronische Zeitschriftenbibliothek - Frei zugängliche E-Journals
    Source: Alma/SFX Local Collection
    Source: SWEPUB Freely available online
    Source: © ProQuest LLC All rights reserved〈img src="https://exlibris-pub.s3.amazonaws.com/PQ_Logo.jpg" style="vertical-align:middle;margin-left:7px"〉
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  • 4
    Language: English
    In: Archives of virology, 2017-12, Vol.162 (12), p.3639-3644
    Description: The human endogenous retroviruses (HERVs) are a family of endogenous retroviruses that integrated into the germ cell DNA of primates over 30 million years ago. HERV expression seems impaired in several diseases, ranging from autoimmune to neoplastic disorders. The purpose of this study was to evaluate the overall endogenous retroviral transcription profile in bone marrow (BM) samples. A total of 30 paediatric high-risk leukaemia patients (lymphoid and myeloid malignancies) were tested for HERVs virus gene expression. Our findings show that HERV-K expression was significantly higher in leukaemia patients when compared to healthy donors of a similar median age. We observed a significantly high expression of HERV-K in acute lymphoblastic leukemia (ALL) patients. In this study, we also found a relative overexpression of the endogenous retrovirus HERV-K in BM cells from the majority of leukemia samples analyzed, in particular in ALL. This overexpression might be related to lymphatic leukemogenesis and it warrants further investigations.
    Subject(s): Medical Microbiology ; Infectious Diseases ; Biomedicine ; Virology ; Gene Expression ; Leukemia - pathology ; Endogenous Retroviruses - enzymology ; Humans ; Child, Preschool ; Infant ; Male ; Gene Expression Profiling ; Gene Products, pol - analysis ; Bone Marrow - pathology ; Adolescent ; Female ; Child ; Medical research ; Retroviruses ; Leukemia ; Genes ; Transplantation ; Gene expression ; Analysis ; Stem cells ; Medicine, Experimental ; Genetic research ; Genetic aspects ; Public health ; Cancer ; Index Medicus
    ISSN: 0304-8608
    E-ISSN: 1432-8798
    Source: Alma/SFX Local Collection
    Source: © ProQuest LLC All rights reserved〈img src="https://exlibris-pub.s3.amazonaws.com/PQ_Logo.jpg" style="vertical-align:middle;margin-left:7px"〉
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  • 5
    Language: English
    In: Journal of bone and mineral research, 2017-01, Vol.32 (1), p.99-105
    Description: ABSTRACT Autosomal recessive osteopetroses (AROs) are rare, genetically heterogeneous skeletal diseases with increased bone density that are often lethal if left untreated. A precise molecular classification is relevant for the patient's management, because in some subgroups hematopoietic stem cell transplantation (HSCT), which is the only curative therapy, is contraindicated. In two unrelated ARO patients, the molecular analysis revealed the presence of a synonymous variant in known ARO genes, namely in the TCIRG1 gene in one patient and in the CLCN7 in the other patient, predicted to impact on the splicing process. In the latter case, sequencing of the transcript confirmed the splicing defect, whereas in the former, for whom an RNA sample was not available, the defect was reconstructed in vitro by the minigene technology. These results strongly suggest that these synonymous changes were responsible for the disease in our patients. Our findings are novel with respect to ARO and add to the few reports in literature dealing with different diseases, underlining the importance of cDNA analysis for the correct assessment of exonic changes, even when exome sequencing is performed. In particular, we highlight the possibility that at least in some cases ARO is due to synonymous changes, erroneously considered clinically silent, in the genes already described in literature, and suggest carefully reevaluating the sequencing results of these genes when mutations are not found at a first analysis. In addition, with respect to the CLCN7 gene, we suggest that synonymous variants might also contribute to the large spectrum of severity typical of CLCN7‐dependent osteopetrosis through more subtle, but not negligible, effects on protein availability and functionality. © 2016 American Society for Bone and Mineral Research.
    Subject(s): CLCN7 ; SYNONYMOUS MUTATION ; TCIRG1 ; OSTEOPETROSIS ; MINIGENE ; Amino Acid Sequence ; Osteopetrosis - diagnostic imaging ; Vacuolar Proton-Translocating ATPases - genetics ; Humans ; Infant ; Male ; Chloride Channels - genetics ; Pregnancy ; Sequence Alignment ; Base Sequence ; Vacuolar Proton-Translocating ATPases - chemistry ; Fatal Outcome ; Osteopetrosis - diagnosis ; Female ; Osteopetrosis - genetics ; Silent Mutation - genetics ; Osteopetrosis ; Analysis ; Genetic research ; Bones ; Genetic aspects ; Diagnosis ; Density ; Hematopoietic stem cells ; Index Medicus
    ISSN: 0884-0431
    E-ISSN: 1523-4681
    Source: Alma/SFX Local Collection
    Source: © ProQuest LLC All rights reserved〈img src="https://exlibris-pub.s3.amazonaws.com/PQ_Logo.jpg" style="vertical-align:middle;margin-left:7px"〉
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  • 6
    Language: English
    In: European journal of haematology, 2018-03, Vol.100 (3), p.315-322
    Description: Background Posaconazole is a triazole with limited pharmacokinetic information in children. This study assessed the correlation between posaconazole oral solution daily dosage/kg/body weight and trough plasma level. Methods A total of 97 hematology‐oncology pediatric patients with ≥1 posaconazole plasma concentration level (PPC) assessment in the first 6 weeks after the start of posaconazole treatment were included. Results Posaconazole was used as prophylaxis in 84 of 97 (87%) patients and as therapy in 13 of 97 (13%). The median daily dose/kg/bw ranged from 10 to 12 mg in the prophylaxis group and 12.5 to 16.5 mg in the therapy group. The median value of PPC for the prophylaxis group was 0.9 and 0.8 μg/mL at the first and second/third determinations, respectively. Posaconazole prophylaxis failed in 4 of 84 patients (5%). The median value of PPC for the therapy group was 1.5 and 1.4 μg/mL at the first/second and the third determination, respectively. Posaconazole‐related side effects were reported in 6 patients and all regressed with the suspension of the drug. In the prophylaxis group, the use of proton‐pump inhibitors was significantly associated with a lower PPC, P = 0.04. Conclusions Posaconazole may be a valuable antifungal agent in children despite the incomplete knowledge of its pharmacokinetic characteristics.
    Subject(s): posaconazole ; hematopoietic stem cell transplantation ; treatment ; invasive fungal infection ; prophylaxis ; pediatric malignancy ; Pediatrics ; Triazoles ; Antifungal agents ; Mycoses ; Transplantation ; Children ; Health aspects ; Oral medication ; Hematopoietic stem cells ; Index Medicus
    ISSN: 0902-4441
    E-ISSN: 1600-0609
    Source: Academic Search Ultimate
    Source: Alma/SFX Local Collection
    Source: © ProQuest LLC All rights reserved〈img src="https://exlibris-pub.s3.amazonaws.com/PQ_Logo.jpg" style="vertical-align:middle;margin-left:7px"〉
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  • 7
    Language: English
    In: Pediatric blood & cancer, 2018-08, Vol.65 (8), p.e27098-n/a
    Description: 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 〈 3 WHO on day +7 (P = 0.01). A significant reduction of pain was registered on day +7 in the PBM versus sham group (NRS 1 [0–3] vs. 2.5 [1–5], P 〈 0.006). Reduced use of analgesics was reported in the PBM group, although it was not statistically significant. No significant adverse events attributable to treatment were recorded. Conclusions PBM is a safe, feasible, and effective treatment for children affected by chemotherapy‐induced OM, as it accelerates mucosal recovery and reduces pain.
    Subject(s): clinical trial ; mucositis ; pediatric hemato‐oncology ; laser ; supportive care ; Stomatitis - radiotherapy ; Low-Level Light Therapy - methods ; Double-Blind Method ; Antineoplastic Agents - adverse effects ; Humans ; Stomatitis - chemically induced ; Adolescent ; Female ; Male ; Treatment Outcome ; Child ; Neoplasms - drug therapy ; Evaluation ; Complications and side effects ; Stomatitis ; Care and treatment ; Chemotherapy ; Hospitals ; Lasers in surgery ; Clinical trials ; Lasers in medicine ; Cancer ; Index Medicus
    ISSN: 1545-5009
    E-ISSN: 1545-5017
    Source: Alma/SFX Local Collection
    Source: © ProQuest LLC All rights reserved〈img src="https://exlibris-pub.s3.amazonaws.com/PQ_Logo.jpg" style="vertical-align:middle;margin-left:7px"〉
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  • 8
    Language: English
    In: Mycoses, 2019-02, Vol.62 (2), p.165-170
    Description: Summary Background Invasive mucormycosis is a rare but frequently fatal fungal disease. The acute and rapidly progressive evolution causes unfavourable outcome in 22%‐59% of patients and its treatment represents a clinical challenge, especially in immunocompromised patients. Current data in paediatric oncological patients are limited. Objectives The infection Working Group of the Italian Association of Pediatric Hematology and Oncology (AIEOP) analysed the episodes of invasive mucormycosis occurred between 2009 and 2016. Patients Fifteen cases of proven mucormycosis (male/female 8/7; median age 14.1 years, range 7.7‐18.6) were reported after chemotherapy for acute leukaemia and lymphoma (12) and allogeneic stem cell transplantation (3). The aetiology was Rhizopus oryzae 4, Lichtheimia corymbifera 3 and Mucor spp. 8. Results Paranasal sinus was the primary site of infection in 14/15 patients combined with orbital involvement (9), central nervous system (8), lung (4), thyroid gland and kidney (1). All patients received liposomal Amphotericin B (L‐AmB) (3‐10 mg/kg), with surgical debridement in 14/15 cases. Eleven patients received maintenance treatment with posaconazole (9) or isavuconazole (2). Eight out of fifteen patients (53.3%) died, after 3‐6 months. Conclusions Mucormycosis involved mainly the sinu‐orbital site and affected children 〉10 years. Despite aggressive treatment with high‐dose L‐AmB and timely surgical debridement, the mortality rate remains still high.
    Subject(s): mycoses ; cancer ; children ; mucormycosis ; Invasive Fungal Infections - pathology ; Mucorales - isolation & purification ; Mucormycosis - epidemiology ; Invasive Fungal Infections - epidemiology ; Humans ; Child, Preschool ; Infant ; Male ; Antifungal Agents - therapeutic use ; Mucorales - classification ; Invasive Fungal Infections - drug therapy ; Mucormycosis - microbiology ; Adolescent ; Mucormycosis - pathology ; Survival Analysis ; Female ; Italy - epidemiology ; Retrospective Studies ; Hematologic Neoplasms - complications ; Mucormycosis - drug therapy ; Child ; Invasive Fungal Infections - microbiology ; Infant, Newborn ; Pediatrics ; Mucormycosis ; Research ; Oncology, Experimental ; Cancer ; Index Medicus
    ISSN: 0933-7407
    E-ISSN: 1439-0507
    Source: Academic Search Ultimate
    Source: Alma/SFX Local Collection
    Source: © ProQuest LLC All rights reserved〈img src="https://exlibris-pub.s3.amazonaws.com/PQ_Logo.jpg" style="vertical-align:middle;margin-left:7px"〉
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  • 9
    Language: English
    In: European journal of haematology, 2018-01, Vol.100 (1), p.75-82
    Description: Objectives Posterior reversible encephalopathy syndrome (PRES) is one of the most common neurological complications in hematology‐oncology pediatric patients. Despite an increasingly recognized occurrence, no clear consensus exists regarding how best to manage the syndrome, because most cases of PRES have reported in single‐case reports or small series. Aim of this paper is to identify incidence, clinical features, management, and outcome of PRES in a large series of hematology‐oncology pediatric patients. Methods The cases of PRES occurred in twelve centers of the Italian Association of Pediatric Hematology and Oncology were reported. Results One hundred and twenty‐four cases of PRES in 112 pediatric patients were recorded with an incidence of 2.1% and 4.7%, respectively, in acute lymphoblastic leukemia in first complete remission and hematopoietic stem cell transplantation (HSCT). The majority of cases occurred after a cycle of chemotherapy rather than after stem cell transplant. PRES after chemotherapy significantly differs from that after HSCT for diagnosis, time of presentation, risk factors, management, and outcome. Conclusions This study demonstrates that PRES is a common neurological complication and occurring preferentially in course of induction treatment of some hematologic malignancies, as ALL and after HSCT. It also highlights great clinical differences in the management and outcome in patients with PRES occurring after chemotherapy or after HSCT.
    Subject(s): posterior reversible encephalopathy syndrome ; pediatric oncology, and hematology ; Surveys ; Pediatrics ; Medical research ; Chemotherapy ; Encephalopathy ; Leukemia ; Medicine, Experimental ; Transplantation ; Children ; Health aspects ; Hematopoietic stem cells ; Cancer ; Index Medicus
    ISSN: 0902-4441
    E-ISSN: 1600-0609
    Source: Academic Search Ultimate
    Source: Alma/SFX Local Collection
    Source: © ProQuest LLC All rights reserved〈img src="https://exlibris-pub.s3.amazonaws.com/PQ_Logo.jpg" style="vertical-align:middle;margin-left:7px"〉
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  • 10
    Language: English
    In: The New England journal of medicine, 2008-12-04, Vol.359 (23), p.2502-2504
    Description: To the Editor: There have been few reports of osteosarcoma after allogeneic bone marrow transplantation. 1 We report the development of osteosarcoma in a recipient 17 years after stem-cell transplantation. A 23-month-old boy with β-thalassemia received a bone marrow transplant from his HLA-identical 11-year-old brother in September 1989. Norrie's disease had been diagnosed in the donor at 12 months of age, after enucleation of the right eye was performed because a bilateral retinoblastoma was suspected. No genetic analysis was conducted at the time of surgery. Norrie's disease is an X-linked recessive disease caused by mutations in the NDP gene on Xp11.4. . . .
    Subject(s): Retinal Diseases - genetics ; beta-Thalassemia - therapy ; Humans ; Osteosarcoma - etiology ; Immunophenotyping ; In Situ Hybridization, Fluorescence ; Infant ; Male ; Nerve Tissue Proteins - genetics ; Transplantation Chimera ; Bone Neoplasms - etiology ; Transplantation, Homologous ; Pelvic Bones ; Fatal Outcome ; Cell Line, Tumor ; Bone Neoplasms - genetics ; Eye Proteins - genetics ; Osteosarcoma - genetics ; Genetic Diseases, X-Linked ; Stem Cell Transplantation - adverse effects
    ISSN: 0028-4793
    E-ISSN: 1533-4406
    Source: Single Journals
    Source: © ProQuest LLC All rights reserved〈img src="https://exlibris-pub.s3.amazonaws.com/PQ_Logo.jpg" style="vertical-align:middle;margin-left:7px"〉
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