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@ARTICLE{ObrechtSturm:271966,
author = {Obrecht-Sturm, Denise and Schömig, Lena and Mynarek,
Martin and Bison, Brigitte and Schwarz, Rudolf and Pietsch,
Torsten and Pfister, Stefan M and Sill, Martin and Sturm,
Dominik and Sahm, Felix and Kortmann, Rolf-Dieter and
Gerber, Nicolas U and von Bueren, André O and Fleischhack,
Gudrun and Schüller, Ulrich and Nussbaumer, Gunther and
Benesch, Martin and Rutkowski, Stefan},
title = {{T}reatment response as surrogate to predict risk for
disease progression in pediatric medulloblastoma with
persistent magnetic resonance imaging lesions after
first-line treatment.},
journal = {Neuro-Oncology},
volume = {26},
number = {9},
issn = {1522-8517},
address = {Oxford},
publisher = {Oxford Univ. Press},
reportid = {DZNE-2024-01108},
pages = {1712 - 1722},
year = {2024},
abstract = {This study aims at clarifying the impact of persistent
residual lesions following first-line treatment for
pediatric medulloblastoma.Data on 84 pediatric patients with
medulloblastoma and persistent residual lesions on centrally
reviewed magnetic resonance imaging (MRI) at the end of
first-line therapy were analyzed.Twenty patients $(23.8\%)$
had residual lesions in the tumor bed (R+/M0), 51 $(60.7\%)$
had distant lesions (R0/M+) and 13 $(15.5\%)$ had both
(R+/M+). Overall response to first-line therapy was minor or
partial (≥ $25\%$ reduction, minor response [MR]/PR) for
64 $(76.2\%)$ and stable disease (SD) for 20 patients
$(23.8\%).$ Five-year post-primary-treatment
progression-free (pptPFS) and overall survival (pptOS) were
superior after MR/PR (pptPFS: 62.5 ± $7.0\%[MR/PR]$ vs.
35.9 ± $12.8\%[SD],$ P = .03; pptOS: 79.7 ± 5.9[MR/PR] vs.
55.5 ± 13.9[SD], P = .04). Furthermore, R+/M + was
associated with a higher risk for progression (5-year
pptPFS: 22.9 ± $17.9\%[R+,$ M+] vs. 72.4 ± $12.0\%[R+,$
M0]; P = .03). Watch-and-wait was pursued in 58 patients,
while n = 26 received additional treatments (chemotherapy
only, n = 19; surgery only, n = 2; combined, n = 3; valproic
acid, n = 2), and their outcomes were not superior to
watch-and-wait (5-year pptPFS: 58.5 ± $7.7\%$ vs. 51.6 ±
$10.7\%$ P = .71; 5-year pptOS: 76.3 ± $6.9\%$ vs. 69.8 ±
$9.7\%,$ P = .74). For the whole cohort, 5-year pptPFS by
molecular subgroup (58 cases) were WNT: $100\%,$ SHH: 50.0
± $35.4\%,$ group-4, 52.5 ± 10.5, group-3 54.2 ±
$13.8\%;$ (P = .08).Overall response and extent of lesions
can function as surrogate parameters to predict outcomes in
pediatric MB patients with persistent lesions after
first-line therapy. Especially in the case of solitary
persistent medulloblastoma MRI lesions, additional therapy
was not beneficial. Therefore, treatment response,
extent/kind of residual lesions and further diagnostic
information need consideration for indication of additional
treatments for persisting lesions.},
keywords = {Humans / Medulloblastoma: diagnostic imaging /
Medulloblastoma: pathology / Male / Child / Female /
Magnetic Resonance Imaging: methods / Cerebellar Neoplasms:
diagnostic imaging / Cerebellar Neoplasms: pathology /
Disease Progression / Child, Preschool / Adolescent /
Follow-Up Studies / Prognosis / Retrospective Studies /
Survival Rate / Infant / Neoplasm, Residual: diagnostic
imaging / Neoplasm, Residual: pathology / MRI (Other) /
children (Other) / medulloblastoma (Other) / persistent
residual disease (Other)},
cin = {Brainbank (Bonn)},
ddc = {610},
cid = {I:(DE-2719)1011009},
pnm = {353 - Clinical and Health Care Research (POF4-353)},
pid = {G:(DE-HGF)POF4-353},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:38578306},
pmc = {pmc:PMC11376455},
doi = {10.1093/neuonc/noae071},
url = {https://pub.dzne.de/record/271966},
}