Treatment of children under 4 years of age with medulloblastoma and ependymoma in the HIT2000/HIT-REZ 2005 trials
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Treatment of children under 4 years of age with medulloblastoma and ependymoma in the HIT2000/HIT-REZ 2005 trials : Neuropsychological outcome 5 years after treatment. / Ottensmeier, Holger; Schlegel, Paul G; Eyrich, Matthias; Wolff, Johannes E; Juhnke, Björn-Ole; von Hoff, Katja; Frahsek, Stefanie; Schmidt, Rene; Faldum, Andreas; Fleischhack, Gudrun; von Bueren, Andre; Friedrich, Carsten; Resch, Anika; Warmuth-Metz, Monika; Krauss, Jürgen; Kortmann, Rolf D; Bode, Udo; Kühl, Joachim; Rutkowski, Stefan.
In: PLOS ONE, Vol. 15, No. 1, 2020, p. e0227693.Research output: SCORING: Contribution to journal › SCORING: Journal article › Research › peer-review
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TY - JOUR
T1 - Treatment of children under 4 years of age with medulloblastoma and ependymoma in the HIT2000/HIT-REZ 2005 trials
T2 - Neuropsychological outcome 5 years after treatment
AU - Ottensmeier, Holger
AU - Schlegel, Paul G
AU - Eyrich, Matthias
AU - Wolff, Johannes E
AU - Juhnke, Björn-Ole
AU - von Hoff, Katja
AU - Frahsek, Stefanie
AU - Schmidt, Rene
AU - Faldum, Andreas
AU - Fleischhack, Gudrun
AU - von Bueren, Andre
AU - Friedrich, Carsten
AU - Resch, Anika
AU - Warmuth-Metz, Monika
AU - Krauss, Jürgen
AU - Kortmann, Rolf D
AU - Bode, Udo
AU - Kühl, Joachim
AU - Rutkowski, Stefan
PY - 2020
Y1 - 2020
N2 - Young children with brain tumours are at high risk of developing treatment-related sequelae. We aimed to assess neuropsychological outcomes 5 years after treatment. This cross-sectional study included children under 4 years of age with medulloblastoma (MB) or ependymoma (EP) enrolled in the German brain tumour trials HIT2000 and HIT-REZ2005. Testing was performed using the validated Wuerzburg Intelligence Diagnostics (WUEP-D), which includes Kaufman-Assessment-Battery, Coloured Progressive Matrices, Visual-Motor Integration, finger tapping "Speed", and the Continuous Performance Test. Of 104 patients in 47 centres, 72 were eligible for analyses. We assessed whether IQ was impacted by disease extent, disease location, patient age, gender, age at surgery, and treatment (chemotherapy with our without craniospinal irradiation [CSI] or local radiotherapy [LRT]). Median age at surgery was 2.3 years. Testing was performed at a median of 4.9 years after surgery. Patients with infratentorial EPs (treated with LRT) scored highest in fluid intelligence (CPM 100.9±16.9, mean±SD); second best scores were achieved by patients with MB without metastasis treated with chemotherapy alone (CPM 93.9±13.2), followed by patients with supratentorial EPs treated with LRT. In contrast, lowest scores were achieved by patients that received chemotherapy and CSI, which included children with metastasised MB and those with relapsed MB M0 (CPM 71.7±8.0 and 73.2±21.8, respectively). Fine motor skills were reduced in all groups. Multivariable analysis revealed that type of treatment had an impact on IQ, but essentially not age at surgery, time since surgery or gender. Our results confirm previous reports on the detrimental effects of CSI in a larger cohort of children. Comparable IQ scores in children with MB treated only with chemotherapy and in children with EP suggest that this treatment strategy represents an attractive option for children who have a high chance to avoid application of CSI. Longitudinal follow-up examinations are warranted to assess long-term neuropsychological outcomes.
AB - Young children with brain tumours are at high risk of developing treatment-related sequelae. We aimed to assess neuropsychological outcomes 5 years after treatment. This cross-sectional study included children under 4 years of age with medulloblastoma (MB) or ependymoma (EP) enrolled in the German brain tumour trials HIT2000 and HIT-REZ2005. Testing was performed using the validated Wuerzburg Intelligence Diagnostics (WUEP-D), which includes Kaufman-Assessment-Battery, Coloured Progressive Matrices, Visual-Motor Integration, finger tapping "Speed", and the Continuous Performance Test. Of 104 patients in 47 centres, 72 were eligible for analyses. We assessed whether IQ was impacted by disease extent, disease location, patient age, gender, age at surgery, and treatment (chemotherapy with our without craniospinal irradiation [CSI] or local radiotherapy [LRT]). Median age at surgery was 2.3 years. Testing was performed at a median of 4.9 years after surgery. Patients with infratentorial EPs (treated with LRT) scored highest in fluid intelligence (CPM 100.9±16.9, mean±SD); second best scores were achieved by patients with MB without metastasis treated with chemotherapy alone (CPM 93.9±13.2), followed by patients with supratentorial EPs treated with LRT. In contrast, lowest scores were achieved by patients that received chemotherapy and CSI, which included children with metastasised MB and those with relapsed MB M0 (CPM 71.7±8.0 and 73.2±21.8, respectively). Fine motor skills were reduced in all groups. Multivariable analysis revealed that type of treatment had an impact on IQ, but essentially not age at surgery, time since surgery or gender. Our results confirm previous reports on the detrimental effects of CSI in a larger cohort of children. Comparable IQ scores in children with MB treated only with chemotherapy and in children with EP suggest that this treatment strategy represents an attractive option for children who have a high chance to avoid application of CSI. Longitudinal follow-up examinations are warranted to assess long-term neuropsychological outcomes.
KW - Brain Neoplasms/pathology
KW - Child
KW - Child, Preschool
KW - Cohort Studies
KW - Combined Modality Therapy
KW - Craniospinal Irradiation/adverse effects
KW - Cross-Sectional Studies
KW - Ependymoma/pathology
KW - Female
KW - Follow-Up Studies
KW - Germany
KW - Humans
KW - Infant
KW - Intelligence
KW - Male
KW - Medulloblastoma/physiopathology
KW - Motor Skills
KW - Multivariate Analysis
KW - Neuropsychological Tests
KW - Treatment Outcome
U2 - 10.1371/journal.pone.0227693
DO - 10.1371/journal.pone.0227693
M3 - SCORING: Journal article
C2 - 31971950
VL - 15
SP - e0227693
JO - PLOS ONE
JF - PLOS ONE
SN - 1932-6203
IS - 1
ER -