-
Je něco špatně v tomto záznamu ?
Survival and functional outcomes of molecularly defined childhood posterior fossa ependymoma: Cure at a cost
M. Zapotocky, K. Beera, J. Adamski, N. Laperierre, S. Guger, L. Janzen, A. Lassaletta, L. Figueiredo Nobre, U. Bartels, U. Tabori, C. Hawkins, S. Urbach, DS. Tsang, PB. Dirks, MD. Taylor, E. Bouffet, DJ. Mabbott, V. Ramaswamy,
Jazyk angličtina Země Spojené státy americké
Typ dokumentu časopisecké články, Research Support, N.I.H., Extramural, práce podpořená grantem
Grantová podpora
R01 CA159859
NCI NIH HHS - United States
R01 CA148699
NCI NIH HHS - United States
CIHR - Canada
PubMed
30768777
DOI
10.1002/cncr.31995
Knihovny.cz E-zdroje
- MeSH
- analýza přežití MeSH
- dítě MeSH
- ependymom mortalita psychologie terapie MeSH
- infratentoriální nádory mortalita psychologie terapie MeSH
- kojenec MeSH
- lidé MeSH
- mladiství MeSH
- neoadjuvantní terapie škodlivé účinky MeSH
- neurochirurgické výkony škodlivé účinky MeSH
- neurokognitivní poruchy etiologie MeSH
- předškolní dítě MeSH
- radioterapie škodlivé účinky MeSH
- výsledek terapie MeSH
- Check Tag
- dítě MeSH
- kojenec MeSH
- lidé MeSH
- mladiství MeSH
- mužské pohlaví MeSH
- předškolní dítě MeSH
- ženské pohlaví MeSH
- Publikační typ
- časopisecké články MeSH
- práce podpořená grantem MeSH
- Research Support, N.I.H., Extramural MeSH
- Geografické názvy
- Ontario MeSH
BACKGROUND: Posterior fossa ependymoma (PFE) comprises 2 groups, PF group A (PFA) and PF group B (PFB), with stark differences in outcome. However, to the authors' knowledge, the long-term outcomes of PFA ependymoma have not been described fully. The objective of the current study was to identify predictors of survival and neurocognitive outcome in a large consecutive cohort of subgrouped patients with PFE over 30 years. METHODS: Demographic, survival, and neurocognitive data were collected from consecutive patients diagnosed with PFE from 1985 through 2014 at the Hospital for Sick Children in Toronto, Ontario, Canada. Subgroup was assigned using genome-wide methylation array and/or immunoreactivity to histone H3 K27 trimethylation (H3K27me3). RESULTS: A total of 72 PFE cases were identified, 89% of which were PFA. There were no disease recurrences noted among patients with PFB. The 10-year progression-free survival rate for all patients with PFA was poor at 37.1% (95% confidence interval, 25.9%-53.1%). Analysis of consecutive 10-year epochs revealed significant improvements in progression-free survival and/or overall survival over time. This pertains to the increase in the rate of gross (macroscopic) total resection from 35% to 77% and the use of upfront radiotherapy increasing from 65% to 96% over the observed period and confirmed in a multivariable model. Using a mixed linear model, analysis of longitudinal neuropsychological outcomes restricted to patients with PFA who were treated with focal irradiation demonstrated significant continuous declines in the full-scale intelligence quotient over time with upfront conformal radiotherapy, even when correcting for hydrocephalus, number of surgeries, and age at diagnosis (-1.33 ± 0.42 points/year; P = .0042). CONCLUSIONS: Data from a molecularly informed large cohort of patients with PFE clearly indicate improved survival over time, related to more aggressive surgery and upfront radiotherapy. However, to the best of the authors' knowledge, the current study is the first, in a subgrouped cohort, to demonstrate that this approach results in reduced neurocognitive outcomes over time.
Department of Paediatric Laboratory Medicine Hospital for Sick Children Toronto Ontario Canada
Department of Psychology Hospital for Sick Children Toronto Ontario Canada
Department of Radiation Oncology Princess Margaret Cancer Center Toronto Ontario Canada
Division of Endocrinology Hospital for Sick Children Toronto Ontario Canada
Division of Haematology Oncology Hospital for Sick Children Toronto Ontario Canada
Programme in Neuroscience and Mental Health Hospital for Sick Children Toronto Ontario Canada
Citace poskytuje Crossref.org
- 000
- 00000naa a2200000 a 4500
- 001
- bmc20006594
- 003
- CZ-PrNML
- 005
- 20200521085255.0
- 007
- ta
- 008
- 200511s2019 xxu f 000 0|eng||
- 009
- AR
- 024 7_
- $a 10.1002/cncr.31995 $2 doi
- 035 __
- $a (PubMed)30768777
- 040 __
- $a ABA008 $b cze $d ABA008 $e AACR2
- 041 0_
- $a eng
- 044 __
- $a xxu
- 100 1_
- $a Zapotocky, Michal $u Division of Haematology/Oncology, Hospital for Sick Children, Toronto, Ontario, Canada. Department of Paediatric Haematology and Oncology, Second Medical School, Charles University and University Hospital Motol, Prague, Czech Republic.
- 245 10
- $a Survival and functional outcomes of molecularly defined childhood posterior fossa ependymoma: Cure at a cost / $c M. Zapotocky, K. Beera, J. Adamski, N. Laperierre, S. Guger, L. Janzen, A. Lassaletta, L. Figueiredo Nobre, U. Bartels, U. Tabori, C. Hawkins, S. Urbach, DS. Tsang, PB. Dirks, MD. Taylor, E. Bouffet, DJ. Mabbott, V. Ramaswamy,
- 520 9_
- $a BACKGROUND: Posterior fossa ependymoma (PFE) comprises 2 groups, PF group A (PFA) and PF group B (PFB), with stark differences in outcome. However, to the authors' knowledge, the long-term outcomes of PFA ependymoma have not been described fully. The objective of the current study was to identify predictors of survival and neurocognitive outcome in a large consecutive cohort of subgrouped patients with PFE over 30 years. METHODS: Demographic, survival, and neurocognitive data were collected from consecutive patients diagnosed with PFE from 1985 through 2014 at the Hospital for Sick Children in Toronto, Ontario, Canada. Subgroup was assigned using genome-wide methylation array and/or immunoreactivity to histone H3 K27 trimethylation (H3K27me3). RESULTS: A total of 72 PFE cases were identified, 89% of which were PFA. There were no disease recurrences noted among patients with PFB. The 10-year progression-free survival rate for all patients with PFA was poor at 37.1% (95% confidence interval, 25.9%-53.1%). Analysis of consecutive 10-year epochs revealed significant improvements in progression-free survival and/or overall survival over time. This pertains to the increase in the rate of gross (macroscopic) total resection from 35% to 77% and the use of upfront radiotherapy increasing from 65% to 96% over the observed period and confirmed in a multivariable model. Using a mixed linear model, analysis of longitudinal neuropsychological outcomes restricted to patients with PFA who were treated with focal irradiation demonstrated significant continuous declines in the full-scale intelligence quotient over time with upfront conformal radiotherapy, even when correcting for hydrocephalus, number of surgeries, and age at diagnosis (-1.33 ± 0.42 points/year; P = .0042). CONCLUSIONS: Data from a molecularly informed large cohort of patients with PFE clearly indicate improved survival over time, related to more aggressive surgery and upfront radiotherapy. However, to the best of the authors' knowledge, the current study is the first, in a subgrouped cohort, to demonstrate that this approach results in reduced neurocognitive outcomes over time.
- 650 _2
- $a mladiství $7 D000293
- 650 _2
- $a dítě $7 D002648
- 650 _2
- $a předškolní dítě $7 D002675
- 650 _2
- $a ependymom $x mortalita $x psychologie $x terapie $7 D004806
- 650 _2
- $a ženské pohlaví $7 D005260
- 650 _2
- $a lidé $7 D006801
- 650 _2
- $a kojenec $7 D007223
- 650 _2
- $a infratentoriální nádory $x mortalita $x psychologie $x terapie $7 D015192
- 650 _2
- $a mužské pohlaví $7 D008297
- 650 _2
- $a neoadjuvantní terapie $x škodlivé účinky $7 D020360
- 650 _2
- $a neurokognitivní poruchy $x etiologie $7 D019965
- 650 _2
- $a neurochirurgické výkony $x škodlivé účinky $7 D019635
- 650 _2
- $a radioterapie $x škodlivé účinky $7 D011878
- 650 _2
- $a analýza přežití $7 D016019
- 650 _2
- $a výsledek terapie $7 D016896
- 651 _2
- $a Ontario $7 D009864
- 655 _2
- $a časopisecké články $7 D016428
- 655 _2
- $a Research Support, N.I.H., Extramural $7 D052061
- 655 _2
- $a práce podpořená grantem $7 D013485
- 700 1_
- $a Beera, Kiran $u Programme in Neuroscience and Mental Health, Hospital for Sick Children, Toronto, Ontario, Canada.
- 700 1_
- $a Adamski, Jenny $u Division of Haematology/Oncology, Hospital for Sick Children, Toronto, Ontario, Canada. Department of Pediatric Oncology, Birmingham Children's Hospital NHS Foundation Trust, Birmingham, United Kingdom.
- 700 1_
- $a Laperierre, Normand $u Department of Radiation Oncology, Princess Margaret Cancer Center, Toronto, Ontario, Canada.
- 700 1_
- $a Guger, Sharon $u Department of Psychology, Hospital for Sick Children, Toronto, Ontario, Canada.
- 700 1_
- $a Janzen, Laura $u Programme in Neuroscience and Mental Health, Hospital for Sick Children, Toronto, Ontario, Canada. Department of Psychology, Hospital for Sick Children, Toronto, Ontario, Canada.
- 700 1_
- $a Lassaletta, Alvaro $u Division of Haematology/Oncology, Hospital for Sick Children, Toronto, Ontario, Canada. Department of Paediatric Hematology and Oncology, Child Jesus Hospital, Madrid, Spain.
- 700 1_
- $a Figueiredo Nobre, Liana $u Division of Haematology/Oncology, Hospital for Sick Children, Toronto, Ontario, Canada.
- 700 1_
- $a Bartels, Ute $u Division of Haematology/Oncology, Hospital for Sick Children, Toronto, Ontario, Canada.
- 700 1_
- $a Tabori, Uri $u Division of Haematology/Oncology, Hospital for Sick Children, Toronto, Ontario, Canada.
- 700 1_
- $a Hawkins, Cynthia $u Department of Paediatric Laboratory Medicine, Hospital for Sick Children, Toronto, Ontario, Canada.
- 700 1_
- $a Urbach, Stacey $u Division of Endocrinology, Hospital for Sick Children, Toronto, Ontario, Canada.
- 700 1_
- $a Tsang, Derek S $u Department of Radiation Oncology, Princess Margaret Cancer Center, Toronto, Ontario, Canada.
- 700 1_
- $a Dirks, Peter B $u Division of Neurosurgery, Hospital for Sick Children, Toronto, Ontario, Canada. Programme in Developmental and Stem Cell Biology, Hospital for Sick Children, Toronto, Ontario, Canada.
- 700 1_
- $a Taylor, Michael D $u Division of Neurosurgery, Hospital for Sick Children, Toronto, Ontario, Canada. Programme in Developmental and Stem Cell Biology, Hospital for Sick Children, Toronto, Ontario, Canada.
- 700 1_
- $a Bouffet, Eric $u Division of Haematology/Oncology, Hospital for Sick Children, Toronto, Ontario, Canada.
- 700 1_
- $a Mabbott, Donald J $u Programme in Neuroscience and Mental Health, Hospital for Sick Children, Toronto, Ontario, Canada. Department of Psychology, University of Toronto, Toronto, Ontario, Canada.
- 700 1_
- $a Ramaswamy, Vijay $u Division of Haematology/Oncology, Hospital for Sick Children, Toronto, Ontario, Canada. Programme in Developmental and Stem Cell Biology, Hospital for Sick Children, Toronto, Ontario, Canada.
- 773 0_
- $w MED00001031 $t Cancer $x 1097-0142 $g Roč. 125, č. 11 (2019), s. 1867-1876
- 856 41
- $u https://pubmed.ncbi.nlm.nih.gov/30768777 $y Pubmed
- 910 __
- $a ABA008 $b sig $c sign $y a $z 0
- 990 __
- $a 20200511 $b ABA008
- 991 __
- $a 20200521085252 $b ABA008
- 999 __
- $a ok $b bmc $g 1525452 $s 1096650
- BAS __
- $a 3
- BAS __
- $a PreBMC
- BMC __
- $a 2019 $b 125 $c 11 $d 1867-1876 $e 20190215 $i 1097-0142 $m Cancer $n Cancer $x MED00001031
- GRA __
- $a R01 CA159859 $p NCI NIH HHS $2 United States
- GRA __
- $a R01 CA148699 $p NCI NIH HHS $2 United States
- GRA __
- $p CIHR $2 Canada
- LZP __
- $a Pubmed-20200511
