Survivors of infant atypical teratoid/rhabdoid tumors present with severely impaired cognitive functions especially for fluid intelligence and visual processing: data from the German brain tumor studies
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Survivors of infant atypical teratoid/rhabdoid tumors present with severely impaired cognitive functions especially for fluid intelligence and visual processing: data from the German brain tumor studies. / Traunwieser, Thomas; Loos, Elena; Ottensmeier, Holger; Gastberger, Katharina; Nemes, Karolina; Mynarek, Martin; Bison, Brigitte; Kandels, Daniela; Neumayer, Petra; Neumann-Holbeck, Anne; Lüttich, Peggy; Baust, Katja; Faulstich-Ritter, Kristin; John, Rainer; Kreisch, Andrea; Landmann, Judyta; Manteufel, Eva; Nest, Alexandra; Prüfe, Jenny; Schubert, Lisa; Stamm, Walther; Timmermann, Beate; Gerss, Joachim; Rutkowski, Stefan; Schlegel, Paul-Gerhardt; Eyrich, Matthias; Gnekow, Astrid K; Frühwald, Michael C.
In: PEDIATR BLOOD CANCER, Vol. 71, No. 5, 05.2024, p. e30910.Research output: SCORING: Contribution to journal › SCORING: Journal article › Research › peer-review
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TY - JOUR
T1 - Survivors of infant atypical teratoid/rhabdoid tumors present with severely impaired cognitive functions especially for fluid intelligence and visual processing: data from the German brain tumor studies
AU - Traunwieser, Thomas
AU - Loos, Elena
AU - Ottensmeier, Holger
AU - Gastberger, Katharina
AU - Nemes, Karolina
AU - Mynarek, Martin
AU - Bison, Brigitte
AU - Kandels, Daniela
AU - Neumayer, Petra
AU - Neumann-Holbeck, Anne
AU - Lüttich, Peggy
AU - Baust, Katja
AU - Faulstich-Ritter, Kristin
AU - John, Rainer
AU - Kreisch, Andrea
AU - Landmann, Judyta
AU - Manteufel, Eva
AU - Nest, Alexandra
AU - Prüfe, Jenny
AU - Schubert, Lisa
AU - Stamm, Walther
AU - Timmermann, Beate
AU - Gerss, Joachim
AU - Rutkowski, Stefan
AU - Schlegel, Paul-Gerhardt
AU - Eyrich, Matthias
AU - Gnekow, Astrid K
AU - Frühwald, Michael C
N1 - © 2024 The Authors. Pediatric Blood & Cancer published by Wiley Periodicals LLC.
PY - 2024/5
Y1 - 2024/5
N2 - BACKGROUND: The contribution of tumor type, multimodal treatment, and other patient-related factors upon long-term cognitive sequelae in infant brain tumor survivors remains undefined. We add our retrospective analysis of neuropsychological and quality of survival (QoS) outcome data of survivors of atypical teratoid/rhabdoid tumors (ATRT) and extracranial malignant rhabdoid tumors of the soft tissues (eMRT) and kidneys (RTK) treated within the same framework. Neuropsychological data from children with ATRT were compared to data from children with non-irradiated low-grade glioma (LGG).PATIENTS AND METHODS: Following surgery, patients (0-36 months at diagnosis) had received radio-chemotherapy (up to 54 Gy; ATRT: n = 13; eMRT/RTK: n = 7), chemotherapy only (LGG: n = 4; eMRT/RTK: n = 1) or had been observed (LGG: n = 11). Neuropsychological evaluation employing comparable tests was performed at median 6.8 years (ATRT), 6.6 years (eMRT/RTK), and 5.2 years (LGG) post diagnosis.RESULTS: We detected sequelae in various domains for all tumor types. Group comparison showed impairments, specifically in fluid intelligence (p = .041; d = 1.11) and visual processing (p = .001; d = 2.09) in ATRT patients when compared to LGG patients. Results for psychomotor speed and attention abilities were significantly below the norm for both groups (p < .001-.019; d = 0.79-1.90). Diagnosis predicted impairments of cognitive outcome, while sex- and age-related variables did not. QoS outcome for all rhabdoid patients displayed impairments mainly in social (p = .008; d = 0.74) and school functioning (p = .048; d = 0.67), as well as lower overall scores in psychosocial functioning (p = .023; d = 0.78) and quality of life (p = .006; d = 0.79) compared to healthy controls.CONCLUSION: Survivors of infant ATRT experience various late effects in cognition and QoS following multimodal treatment, while infant LGG patients without radiotherapy demonstrated comparable impairments in psychomotor and attention abilities. Early onset and multimodal treatment of rhabdoid tumors require close monitoring of neuropsychological and QoS sequelae.
AB - BACKGROUND: The contribution of tumor type, multimodal treatment, and other patient-related factors upon long-term cognitive sequelae in infant brain tumor survivors remains undefined. We add our retrospective analysis of neuropsychological and quality of survival (QoS) outcome data of survivors of atypical teratoid/rhabdoid tumors (ATRT) and extracranial malignant rhabdoid tumors of the soft tissues (eMRT) and kidneys (RTK) treated within the same framework. Neuropsychological data from children with ATRT were compared to data from children with non-irradiated low-grade glioma (LGG).PATIENTS AND METHODS: Following surgery, patients (0-36 months at diagnosis) had received radio-chemotherapy (up to 54 Gy; ATRT: n = 13; eMRT/RTK: n = 7), chemotherapy only (LGG: n = 4; eMRT/RTK: n = 1) or had been observed (LGG: n = 11). Neuropsychological evaluation employing comparable tests was performed at median 6.8 years (ATRT), 6.6 years (eMRT/RTK), and 5.2 years (LGG) post diagnosis.RESULTS: We detected sequelae in various domains for all tumor types. Group comparison showed impairments, specifically in fluid intelligence (p = .041; d = 1.11) and visual processing (p = .001; d = 2.09) in ATRT patients when compared to LGG patients. Results for psychomotor speed and attention abilities were significantly below the norm for both groups (p < .001-.019; d = 0.79-1.90). Diagnosis predicted impairments of cognitive outcome, while sex- and age-related variables did not. QoS outcome for all rhabdoid patients displayed impairments mainly in social (p = .008; d = 0.74) and school functioning (p = .048; d = 0.67), as well as lower overall scores in psychosocial functioning (p = .023; d = 0.78) and quality of life (p = .006; d = 0.79) compared to healthy controls.CONCLUSION: Survivors of infant ATRT experience various late effects in cognition and QoS following multimodal treatment, while infant LGG patients without radiotherapy demonstrated comparable impairments in psychomotor and attention abilities. Early onset and multimodal treatment of rhabdoid tumors require close monitoring of neuropsychological and QoS sequelae.
KW - Child
KW - Infant
KW - Humans
KW - Rhabdoid Tumor/complications
KW - Retrospective Studies
KW - Quality of Life
KW - Teratoma/complications
KW - Brain Neoplasms/complications
KW - Central Nervous System Neoplasms/pathology
KW - Glioma
KW - Disease Progression
KW - Visual Perception
KW - Cognition
KW - Neoplasms, Neuroepithelial
KW - Survivors
U2 - 10.1002/pbc.30910
DO - 10.1002/pbc.30910
M3 - SCORING: Journal article
C2 - 38342954
VL - 71
SP - e30910
JO - PEDIATR BLOOD CANCER
JF - PEDIATR BLOOD CANCER
SN - 1545-5009
IS - 5
ER -