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@ARTICLE{Filippidou:294066,
author = {M. Filippidou$^*$ and S. Glentis and I. Binenbaum and M.
Sill$^*$ and K. Roka and A. Vlachou and G. Avgerinou and J.
Ecker$^*$ and F. Selt$^*$ and M. Hasselblatt and M.
Blattner-Johnson$^*$ and K. Schramm$^*$ and C. Trougkou and
D. Doganis and N. Katzilakis and V. Ridola and E.
Papakonstantinou and V. Papadakis and E. Hatzipantelis and
E. Kokkinou and R. Pons and C. Kanaka-Gantenbein and D.
Sturm$^*$ and S. Hirsch$^*$ and N. Dikow and K. Pajtler$^*$
and C. M. van Tilburg$^*$ and M. C. Frühwald and T.
Milde$^*$ and O. Witt$^*$ and D. Jones$^*$ and A. von
Deimling$^*$ and F. Sahm$^*$ and K. Stefanaki and S.
Pfister$^*$ and A. Kattamis},
title = {{T}he impact of methylome analysis on the diagnosis and
treatment of {CNS} tumours in children and adolescents: {A}
population-based study in {G}reece},
journal = {EJC paediatric oncology},
volume = {4},
issn = {2772-610X},
address = {[Amsterdam]},
publisher = {Elsevier B.V.},
reportid = {DKFZ-2024-02087},
pages = {100198},
year = {2024},
note = {#EA:B062#LA:B062#},
abstract = {Background: The recently published WHO classification of
central nervous system (CNS) tumours recognizes
DNAmethylation profiling as a desirable and, for some
diagnoses, essential diagnostic tool adjunctive to
conventionalhistopathology. DNA methylation profiling is not
routinely available in many countries, including
Greece.Methods: In this collaborative study, we report the
DNA methylation results in a series of children and
adolescentswith CNS tumours in Greece (2018–2023). In
total, 130 tumour samples were analyzed using the latest
applicable version of the Heidelberg brain tumour
classifier.Results: Upon initial analysis, 80 $\%$ (104/130)
achieved calibrated scores (Cs) ≥ 0.9 and matched an
establishedmethylation class family/subclass. Among them,
methylation results confirmed (90/104, 86.5 $\%),$ refined
(50/104, 48 $\%)$ or changed (10/104, 9.6 $\%)$ the
histological diagnosis. Only four results were regarded as
noncontributing (4/104, 3.9 $\%).$ Twenty-six tumour samples
received Cs < 0.9. Despite low scores, methylationresults
supported the initial diagnosis with lower confidence in
38.5 $\%$ (10/26) and established the diagnosis intwo
tumours with non-conclusive histopathology. Additional
t-distributed stochastic neighbour embedding (tSNE) analysis
allowed the possible classification of twelve tumours. Nine
more samples reached high Cs using thenewer brain tumour
classifiers, since available. Samples co-tested in Greece
demonstrated excellent test reproducibility, supporting the
analysis’ local implementation. Methylome profiling
impacted the clinical management of 40 $\%$ of patients,
modifying stratification, prognosis, or treatment
approach.Conclusions: This study supports the need to
integrate methylome analysis into routine diagnostics in our
countryand highlights the importance of collaboration
between European pediatric oncology centres.},
cin = {B310 / HD01 / B062 / B360 / B300},
ddc = {610},
cid = {I:(DE-He78)B310-20160331 / I:(DE-He78)HD01-20160331 /
I:(DE-He78)B062-20160331 / I:(DE-He78)B360-20160331 /
I:(DE-He78)B300-20160331},
pnm = {312 - Funktionelle und strukturelle Genomforschung
(POF4-312)},
pid = {G:(DE-HGF)POF4-312},
typ = {PUB:(DE-HGF)16},
doi = {10.1016/j.ejcped.2024.100198},
url = {https://inrepo02.dkfz.de/record/294066},
}