Paediatric and adolescent medulloblastoma and CNS-PNET in Norway 1974 – 2013: Survival, regional differences, and late effects
Medulloblastoma (MB) and supratentorial primitive neuroectodermal tumor (CNS-PNET) are embryonal brain tumours, and they account for about 15%–20% and 2.5%, respectively, of malignant brain tumors in children and adolescents. The aim of the thesis was two-fold. First, to investigate whether there wi...
Published in: | Pediatric Blood & Cancer |
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Format: | Doctoral or Postdoctoral Thesis |
Language: | English |
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2021
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Online Access: | http://hdl.handle.net/10852/83146 http://urn.nb.no/URN:NBN:no-85908 |
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Universitet i Oslo: Digitale utgivelser ved UiO (DUO) |
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Medulloblastoma (MB) and supratentorial primitive neuroectodermal tumor (CNS-PNET) are embryonal brain tumours, and they account for about 15%–20% and 2.5%, respectively, of malignant brain tumors in children and adolescents. The aim of the thesis was two-fold. First, to investigate whether there within Norway were re-gional differences in survival for patients with MB and CNS-PNET in the time period 1974–2013, and if so try and identify possible explanations for such differences. The second aim was to determine frequency and severity of undesirable late effects in MB and CNS-PNET survivors, and evaluate rehabilitation need. We performed a retrospective study of patients diagnosed with MB or CNS-PNET in Norway between 1974 and 2013. Second, a clinical follow-up study focusing on late effects and rehabilitation need for MB and CNS-PNET survivors in South-Eastern Norway Regional Health Authority was performed. At the time of analysis, 46% of all Norwegian patients were alive. In univariable analysis, five-year OS, but not five-year event-free survival (EFS), for MB/CNS-PNET altogether was significantly better at University Hospital Of North Norway, St. Olavs Hospi-tal Trondheim University Hospital, and Haukeland University Hospital, compared to Oslo University Hospital (68% vs 54%). We were not able to identify any possible explanations for these regional survival differences within Norway. In the clinical follow-up study, a high burden of unwanted late effects was found in paediatric, adolescent, and adult MB and CNS-PNET survivors, in the vast majority of cases leading to unmet rehabilitation needs. It is im-portant to note that several late effects do not occur until years or even decades following completion of anti-neoplastic treatment and these patients will therefore need life-long follow-up and rehabilitation. |
format |
Doctoral or Postdoctoral Thesis |
author |
Stensvold, Einar |
spellingShingle |
Stensvold, Einar Paediatric and adolescent medulloblastoma and CNS-PNET in Norway 1974 – 2013: Survival, regional differences, and late effects |
author_facet |
Stensvold, Einar |
author_sort |
Stensvold, Einar |
title |
Paediatric and adolescent medulloblastoma and CNS-PNET in Norway 1974 – 2013: Survival, regional differences, and late effects |
title_short |
Paediatric and adolescent medulloblastoma and CNS-PNET in Norway 1974 – 2013: Survival, regional differences, and late effects |
title_full |
Paediatric and adolescent medulloblastoma and CNS-PNET in Norway 1974 – 2013: Survival, regional differences, and late effects |
title_fullStr |
Paediatric and adolescent medulloblastoma and CNS-PNET in Norway 1974 – 2013: Survival, regional differences, and late effects |
title_full_unstemmed |
Paediatric and adolescent medulloblastoma and CNS-PNET in Norway 1974 – 2013: Survival, regional differences, and late effects |
title_sort |
paediatric and adolescent medulloblastoma and cns-pnet in norway 1974 – 2013: survival, regional differences, and late effects |
publishDate |
2021 |
url |
http://hdl.handle.net/10852/83146 http://urn.nb.no/URN:NBN:no-85908 |
geographic |
Norway |
geographic_facet |
Norway |
genre |
North Norway |
genre_facet |
North Norway |
op_relation |
Paper 1: Children treated for medulloblastoma and supratentorial primitive neuroectodermal tumor in Norway from 1974 through 2013 – unexplainable regional differences in survival. Einar Stensvold, Tor Åge Myklebust, Johan Cappelen, Bernt J. Due-Tønnessen, Paulina Due-Tønnessen, Aleksandra Kepka, Tom Børge Johannesen, Bård Krossnes, Tryggve Lundar, Snezana Maric, Hrvoje Miletic, Viggo Moholdt, Kristin Smistad Myrmel, Terje Nordberg, Jana Rydland, Tore Stokland, Kristin Solem, Ole Solheim, Ingrid Torsvik, Gry C. Wikran, Bernward Zeller, Finn Wesenberg, Anne Grete Bechensteen and Petter Brandal. Pediatric Blood Cancer, 2019, Volume 66, Issue 10, October 2019. doi:10.1002/pbc.27910. The article is included in the thesis. Also available at: https://doi.org/10.1002/pbc.27910 Paper 2: Outcome for children treated for medulloblastoma and supratentorial primitive neuroectodermal tumor (CNS-PNET) – a retrospective analysis spanning 40 years of treatment. Einar Stensvold, Bård Kronen Krossnes, Tryggve Lundar, Bernt J. Due-Tønnessen, Paulina Due-Tønnessen, Radek Frič, Anne Grete Bechensteen, Tor Åge Myklebust, Tom Børge Johannesen & Petter Brandal. Acta Oncologica, 2017; 56, 5, 698-705. doi:10.1080/0284186X.2017.1301679. The article is not available in DUO due to publisher restrictions. The published version is available at: https://doi.org/10.1080/0284186X.2017.1301679 Paper 3: Unmet rehabilitation needs in 86% of Norwegian paediatric embryonal tumour survivors. Einar Stensvold, Kristine Stadskleiv, Tor Åge Myklebust, Finn Wesenberg, Eirik Helseth, Anne Grete Bechensteen and Petter Brandal. Acta Paediatrica. 2020; 109:1875–1886. doi:10.1111/apa.15188. Epub 2020 Feb 6. The article is included in the thesis. Also available at: https://doi.org/10.1111/apa.15188 Paper 4: Neuropsychological functioning in survivors of childhood medulloblastoma /CNS-PNET: the role of secondary medical complications. Kristine Stadskleiv, Einar Stensvold, Kjersti Stokka, Anne Grete Bechensteen and Petter Brandal. The Clinical Neuropsychologist, 2020. doi:10.1080/13854046.2020.1794045. The article is included in the thesis. Also available at: https://doi.org/10.1080/13854046.2020.1794045 https://doi.org/10.1002/pbc.27910 https://doi.org/10.1080/0284186X.2017.1301679 https://doi.org/10.1111/apa.15188 https://doi.org/10.1080/13854046.2020.1794045 http://urn.nb.no/URN:NBN:no-85908 http://hdl.handle.net/10852/83146 URN:NBN:no-85908 Fulltext https://www.duo.uio.no/bitstream/handle/10852/83146/1/PhD-Stensvold-2021.pdf |
op_doi |
https://doi.org/10.1002/pbc.27910 https://doi.org/10.1111/apa.15188 https://doi.org/10.1080/13854046.2020.1794045 https://doi.org/10.1080/0284186X.2017.1301679 |
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ftoslouniv:oai:www.duo.uio.no:10852/83146 2023-05-15T17:39:24+02:00 Paediatric and adolescent medulloblastoma and CNS-PNET in Norway 1974 – 2013: Survival, regional differences, and late effects Stensvold, Einar 2021 http://hdl.handle.net/10852/83146 http://urn.nb.no/URN:NBN:no-85908 en eng Paper 1: Children treated for medulloblastoma and supratentorial primitive neuroectodermal tumor in Norway from 1974 through 2013 – unexplainable regional differences in survival. Einar Stensvold, Tor Åge Myklebust, Johan Cappelen, Bernt J. Due-Tønnessen, Paulina Due-Tønnessen, Aleksandra Kepka, Tom Børge Johannesen, Bård Krossnes, Tryggve Lundar, Snezana Maric, Hrvoje Miletic, Viggo Moholdt, Kristin Smistad Myrmel, Terje Nordberg, Jana Rydland, Tore Stokland, Kristin Solem, Ole Solheim, Ingrid Torsvik, Gry C. Wikran, Bernward Zeller, Finn Wesenberg, Anne Grete Bechensteen and Petter Brandal. Pediatric Blood Cancer, 2019, Volume 66, Issue 10, October 2019. doi:10.1002/pbc.27910. The article is included in the thesis. Also available at: https://doi.org/10.1002/pbc.27910 Paper 2: Outcome for children treated for medulloblastoma and supratentorial primitive neuroectodermal tumor (CNS-PNET) – a retrospective analysis spanning 40 years of treatment. Einar Stensvold, Bård Kronen Krossnes, Tryggve Lundar, Bernt J. Due-Tønnessen, Paulina Due-Tønnessen, Radek Frič, Anne Grete Bechensteen, Tor Åge Myklebust, Tom Børge Johannesen & Petter Brandal. Acta Oncologica, 2017; 56, 5, 698-705. doi:10.1080/0284186X.2017.1301679. The article is not available in DUO due to publisher restrictions. The published version is available at: https://doi.org/10.1080/0284186X.2017.1301679 Paper 3: Unmet rehabilitation needs in 86% of Norwegian paediatric embryonal tumour survivors. Einar Stensvold, Kristine Stadskleiv, Tor Åge Myklebust, Finn Wesenberg, Eirik Helseth, Anne Grete Bechensteen and Petter Brandal. Acta Paediatrica. 2020; 109:1875–1886. doi:10.1111/apa.15188. Epub 2020 Feb 6. The article is included in the thesis. Also available at: https://doi.org/10.1111/apa.15188 Paper 4: Neuropsychological functioning in survivors of childhood medulloblastoma /CNS-PNET: the role of secondary medical complications. Kristine Stadskleiv, Einar Stensvold, Kjersti Stokka, Anne Grete Bechensteen and Petter Brandal. The Clinical Neuropsychologist, 2020. doi:10.1080/13854046.2020.1794045. The article is included in the thesis. Also available at: https://doi.org/10.1080/13854046.2020.1794045 https://doi.org/10.1002/pbc.27910 https://doi.org/10.1080/0284186X.2017.1301679 https://doi.org/10.1111/apa.15188 https://doi.org/10.1080/13854046.2020.1794045 http://urn.nb.no/URN:NBN:no-85908 http://hdl.handle.net/10852/83146 URN:NBN:no-85908 Fulltext https://www.duo.uio.no/bitstream/handle/10852/83146/1/PhD-Stensvold-2021.pdf Doctoral thesis Doktoravhandling 2021 ftoslouniv https://doi.org/10.1002/pbc.27910 https://doi.org/10.1111/apa.15188 https://doi.org/10.1080/13854046.2020.1794045 https://doi.org/10.1080/0284186X.2017.1301679 2021-02-17T23:31:00Z Medulloblastoma (MB) and supratentorial primitive neuroectodermal tumor (CNS-PNET) are embryonal brain tumours, and they account for about 15%–20% and 2.5%, respectively, of malignant brain tumors in children and adolescents. The aim of the thesis was two-fold. First, to investigate whether there within Norway were re-gional differences in survival for patients with MB and CNS-PNET in the time period 1974–2013, and if so try and identify possible explanations for such differences. The second aim was to determine frequency and severity of undesirable late effects in MB and CNS-PNET survivors, and evaluate rehabilitation need. We performed a retrospective study of patients diagnosed with MB or CNS-PNET in Norway between 1974 and 2013. Second, a clinical follow-up study focusing on late effects and rehabilitation need for MB and CNS-PNET survivors in South-Eastern Norway Regional Health Authority was performed. At the time of analysis, 46% of all Norwegian patients were alive. In univariable analysis, five-year OS, but not five-year event-free survival (EFS), for MB/CNS-PNET altogether was significantly better at University Hospital Of North Norway, St. Olavs Hospi-tal Trondheim University Hospital, and Haukeland University Hospital, compared to Oslo University Hospital (68% vs 54%). We were not able to identify any possible explanations for these regional survival differences within Norway. In the clinical follow-up study, a high burden of unwanted late effects was found in paediatric, adolescent, and adult MB and CNS-PNET survivors, in the vast majority of cases leading to unmet rehabilitation needs. It is im-portant to note that several late effects do not occur until years or even decades following completion of anti-neoplastic treatment and these patients will therefore need life-long follow-up and rehabilitation. Doctoral or Postdoctoral Thesis North Norway Universitet i Oslo: Digitale utgivelser ved UiO (DUO) Norway Pediatric Blood & Cancer |