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000178901 037__ $$aDKFZ-2022-00332
000178901 041__ $$aEnglish
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000178901 1001_ $$aObrecht, Denise$$b0
000178901 245__ $$aClinical and molecular characterization of isolated M1 disease in pediatric medulloblastoma: experience from the German HIT-MED studies.
000178901 260__ $$aDordrecht [u.a.]$$bSpringer Science + Business Media B.V$$c2022
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000178901 500__ $$a2022 Mar;157(1):37-48
000178901 520__ $$aTo evaluate the clinical impact of isolated spread of medulloblastoma cells into cerebrospinal fluid without additional macroscopic metastases (M1-only).The HIT-MED database was searched for pediatric patients with M1-only medulloblastoma diagnosed from 2000 to 2019. Corresponding clinical and molecular data was evaluated. Treatment was stratified by age and changed over time for older patients.70 patients with centrally reviewed M1-only disease were identified. Clinical data was available for all and molecular data for 45/70 cases. 91% were non-WNT/non-SHH medulloblastoma (Grp3/4). 5-year PFS for 52 patients ≥ 4 years was 59.4 (± 7.1) %, receiving either upfront craniospinal irradiation (CSI) or SKK-sandwich chemotherapy (CT). Outcomes did not differ between these strategies (5-year PFS: CSI 61.7 ± 9.9%, SKK-CT 56.7 ± 6.1%). For patients < 4 years (n = 18), 5-year PFS was 50.0 (± 13.2) %. M1-persistence occurred exclusively using postoperative CT and was a strong negative predictive factor (pPFS/OS < 0.01). Patients with additional clinical or molecular high-risk (HR) characteristics had worse outcomes (5-year PFS 42.7 ± 10.6% vs. 64.0 ± 7.0%, p = 0.03). In n = 22 patients ≥ 4 years with full molecular information and without additional HR characteristics, risk classification by molecular subtyping had an effect on 5-year PFS (HR 16.7 ± 15.2%, SR 77.8 ± 13.9%; p = 0.01).Our results confirm that M1-only is a high-risk condition, and further underline the importance of CSF staging. Specific risk stratification of affected patients needs attention in future discussions for trials and treatment recommendations. Future patients without contraindications may benefit from upfront CSI by sparing risks related to higher cumulative CT applied in sandwich regimen.
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000178901 650_7 $$2Other$$aCerebrospinal fluid
000178901 650_7 $$2Other$$aChildren
000178901 650_7 $$2Other$$aMedulloblastoma
000178901 650_7 $$2Other$$aMetastases
000178901 650_7 $$2Other$$aRadiotherapy
000178901 7001_ $$aMynarek, Martin$$b1
000178901 7001_ $$aHagel, Christian$$b2
000178901 7001_ $$aKwiecien, Robert$$b3
000178901 7001_ $$aSpohn, Michael$$b4
000178901 7001_ $$aBockmayr, Michael$$b5
000178901 7001_ $$aBison, Brigitte$$b6
000178901 7001_ $$0P:(DE-He78)f746aa965c4e1af518b016de3aaff5d9$$aPfister, Stefan M$$b7$$udkfz
000178901 7001_ $$0P:(DE-He78)551bb92841f634070997aa168d818492$$aJones, David T W$$b8$$udkfz
000178901 7001_ $$0P:(DE-He78)a46a5b2a871859c8e2d63d2f8c666807$$aSturm, Dominik$$b9$$udkfz
000178901 7001_ $$0P:(DE-He78)a8a10626a848d31e70cfd96a133cc144$$avon Deimling, Andreas$$b10$$udkfz
000178901 7001_ $$0P:(DE-He78)a1f4b408b9155beb2a8f7cba4d04fe88$$aSahm, Felix$$b11$$udkfz
000178901 7001_ $$avon Hoff, Katja$$b12
000178901 7001_ $$aJuhnke, B-Ole$$b13
000178901 7001_ $$aBenesch, Martin$$b14
000178901 7001_ $$aGerber, Nicolas U$$b15
000178901 7001_ $$aFriedrich, Carsten$$b16
000178901 7001_ $$avon Bueren, André O$$b17
000178901 7001_ $$aKortmann, Rolf-Dieter$$b18
000178901 7001_ $$aSchwarz, Rudolf$$b19
000178901 7001_ $$aPietsch, Torsten$$b20
000178901 7001_ $$aFleischhack, Gudrun$$b21
000178901 7001_ $$aSchüller, Ulrich$$b22
000178901 7001_ $$00000-0002-5446-9571$$aRutkowski, Stefan$$b23
000178901 773__ $$0PERI:(DE-600)2007293-4$$a10.1007/s11060-021-03913-5$$n1$$p37-48$$tJournal of neuro-oncology$$v157$$x0167-594X$$y2022
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