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@ARTICLE{Sharma:143580,
author = {T. Sharma$^*$ and E. C. Schwalbe and D. Williamson and M.
Sill$^*$ and V. Hovestadt and M. Mynarek and S. Rutkowski
and G. W. Robinson and A. Gajjar and F. Cavalli and V.
Ramaswamy and M. D. Taylor and J. C. Lindsey and R. M. Hill
and N. Jäger$^*$ and A. Korshunov$^*$ and D. Hicks and S.
Bailey and M. Kool$^*$ and L. Chavez and P. A. Northcott and
S. Pfister$^*$ and S. C. Clifford},
title = {{S}econd-generation molecular subgrouping of
medulloblastoma: an international meta-analysis of {G}roup 3
and {G}roup 4 subtypes.},
journal = {Acta neuropathologica},
volume = {138},
number = {2},
issn = {1432-0533},
address = {Heidelberg},
publisher = {Springer},
reportid = {DKFZ-2019-01160},
pages = {309-326},
year = {2019},
abstract = {In 2012, an international consensus paper reported that
medulloblastoma comprises four molecular subgroups (WNT,
SHH, Group 3, and Group 4), each associated with distinct
genomic features and clinical behavior. Independently,
multiple recent reports have defined further intra-subgroup
heterogeneity in the form of biologically and clinically
relevant subtypes. However, owing to differences in patient
cohorts and analytical methods, estimates of subtype number
and definition have been inconsistent, especially within
Group 3 and Group 4. Herein, we aimed to reconcile the
definition of Group 3/Group 4 MB subtypes through the
analysis of a series of 1501 medulloblastomas with
DNA-methylation profiling data, including 852 with matched
transcriptome data. Using multiple complementary
bioinformatic approaches, we compared the concordance of
subtype calls between published cohorts and analytical
methods, including assessments of class-definition
confidence and reproducibility. While the lowest complexity
solutions continued to support the original consensus
subgroups of Group 3 and Group 4, our analysis most strongly
supported a definition comprising eight robust Group 3/Group
4 subtypes (types I-VIII). Subtype II was consistently
identified across all component studies, while all others
were supported by multiple class-definition methods.
Regardless of analytical technique, increasing cohort size
did not further increase the number of identified Group
3/Group 4 subtypes. Summarizing the molecular and
clinico-pathological features of these eight subtypes
indicated enrichment of specific driver gene alterations and
cytogenetic events amongst subtypes, and identified highly
disparate survival outcomes, further supporting their
biological and clinical relevance. Collectively, this study
provides continued support for consensus Groups 3 and 4
while enabling robust derivation of, and categorical
accounting for, the extensive intertumoral heterogeneity
within Groups 3 and 4, revealed by recent high-resolution
subclassification approaches. Furthermore, these findings
provide a basis for application of emerging methods (e.g.,
proteomics/single-cell approaches) which may additionally
inform medulloblastoma subclassification. Outputs from this
study will help shape definition of the next generation of
medulloblastoma clinical protocols and facilitate the
application of enhanced molecularly guided risk
stratification to improve outcomes and quality of life for
patients and their families.},
cin = {B062 / B300},
ddc = {610},
cid = {I:(DE-He78)B062-20160331 / I:(DE-He78)B300-20160331},
pnm = {312 - Functional and structural genomics (POF3-312)},
pid = {G:(DE-HGF)POF3-312},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:31076851},
doi = {10.1007/s00401-019-02020-0},
url = {https://inrepo02.dkfz.de/record/143580},
}