The longitudinal evolution of cerebral blood flow in children with tuberous sclerosis assessed by arterial spin labeling magnetic resonance imaging may be related to cognitive performance.
Rutten 2023 The longitudinal evolution of cerebral blood flow in children with tuberous sclerosis assessed by arterial spin labeling magnetic resonance imaging may be related to cognitive performance.pdf
Arterial spin labeling; Child; Longitudinal studies; Magnetic resonance imaging; Tuberous sclerosis; Spin Labels; Humans; Cerebrovascular Circulation; Cognition; Longitudinal Studies; Magnetic Resonance Imaging; Retrospective Studies; Epilepsy; Tuberous Sclerosis/complications; Tuberous Sclerosis/diagnosis; Tuberous Sclerosis/pathology; Radiology, Nuclear Medicine and Imaging
Abstract :
[en] [en] OBJECTIVE: To study longitudinal changes in tuber and whole-brain perfusion in children with tuberous sclerosis complex (TSC) using arterial spin labeling (ASL) perfusion MRI and correlate them with pathological EEG slow wave activity and neurodevelopmental outcomes.
METHODS: Retrospective longitudinal cohort study of 13 children with TSC, 3 to 6 serial ASL-MRI scans between 2 months and 7 years of age (53 scans in total), and an EEG examination performed within 2 months of the last MRI. Tuber cerebral blood flow (CBF) values were calculated in tuber segmentation masks, and tuber:cortical CBF ratios were used to study tuber perfusion. Logistic regression analysis was performed to identify which initial tuber characteristics (CBF value, volume, location) in the first MRI predicted tubers subsequently associated with EEG slow waves. Whole-brain and lobar CBF values were extracted for all patient scans and age-matched controls. CBF ratios were compared in patients and controls to study longitudinal changes in whole-brain CBF.
RESULTS: Perfusion was reduced in tubers associated with EEG slow waves compared with other tubers. Low tuber CBF values around 6 months of age and large tuber volumes were predictive of tubers subsequently associated with EEG slow waves. Patients with severe developmental delay had more severe whole-brain hypoperfusion than those with no/mild delay, which became apparent after 2 years of age and were not associated with a higher tuber load.
CONCLUSIONS: Dynamic changes in tuber and brain perfusion occur over time. Perfusion is significantly reduced in tubers associated with EEG slow waves. Whole-brain perfusion is significantly reduced in patients with severe delay.
KEY POINTS: • Tubers associated with EEG slow wave activity were significantly more hypoperfused than other tubers, especially after 1 year of age. • Larger and more hypoperfused tubers at 6 months of age were more likely to subsequently be associated with pathological EEG slow wave activity. • Patients with severe developmental delay had more extensive and severe global hypoperfusion than those without developmental delay.
Disciplines :
Neurology
Author, co-author :
Rutten, Caroline; Department of Pediatric Radiology, Necker Children's Hospital, Assistance Publique-Hôpitaux de Paris, University of Paris cité, 149 rue de Sèvres, 75015, Paris, France ; Imagine Institute for Genetic Diseases, INSERM U1163, Paris, France
Fillon, Ludovic; Imagine Institute for Genetic Diseases, INSERM U1163, Paris, France
Kuchenbuch, Mathieu; Imagine Institute for Genetic Diseases, INSERM U1163, Paris, France ; Reference Centre for Rare Epilepsies, Department of Pediatric Neurology, Necker-Enfants Malades Hospital, Assistance Publique-Hôpitaux de Paris, University of Paris cité, 149 rue de Sèvres, 75015, Paris, France
Saitovitch, Ana; Imagine Institute for Genetic Diseases, INSERM U1163, Paris, France
Boisgontier, Jennifer; Imagine Institute for Genetic Diseases, INSERM U1163, Paris, France
Chemaly, Nicole; Imagine Institute for Genetic Diseases, INSERM U1163, Paris, France ; Reference Centre for Rare Epilepsies, Department of Pediatric Neurology, Necker-Enfants Malades Hospital, Assistance Publique-Hôpitaux de Paris, University of Paris cité, 149 rue de Sèvres, 75015, Paris, France
Breuillard, Delphine; Reference Centre for Rare Epilepsies, Department of Pediatric Neurology, Necker-Enfants Malades Hospital, Assistance Publique-Hôpitaux de Paris, University of Paris cité, 149 rue de Sèvres, 75015, Paris, France
Ouss, Lisa ; Université de Liège - ULiège > Département des sciences cliniques > Psychiatrie infanto-juvénile ; Department of Child Psychiatry, Necker Children's Hospital, Assistance Publique-Hôpitaux de Paris, University of Paris cité, 149 rue de Sèvres, 75015, Paris, France
Dangouloff-Ros, Volodia; Department of Pediatric Radiology, Necker Children's Hospital, Assistance Publique-Hôpitaux de Paris, University of Paris cité, 149 rue de Sèvres, 75015, Paris, France ; Imagine Institute for Genetic Diseases, INSERM U1163, Paris, France
Blauwblomme, Thomas; Imagine Institute for Genetic Diseases, INSERM U1163, Paris, France ; Department of Pediatric Neurosurgery, Necker Children's Hospital, Assistance Publique-Hôpitaux de Paris, University of Paris cité, 149 rue de Sèvres, 75015, Paris, France
Zilbovicius, Monica; Imagine Institute for Genetic Diseases, INSERM U1163, Paris, France
Nabbout, Rima; Imagine Institute for Genetic Diseases, INSERM U1163, Paris, France ; Reference Centre for Rare Epilepsies, Department of Pediatric Neurology, Necker-Enfants Malades Hospital, Assistance Publique-Hôpitaux de Paris, University of Paris cité, 149 rue de Sèvres, 75015, Paris, France
Boddaert, Nathalie; Department of Pediatric Radiology, Necker Children's Hospital, Assistance Publique-Hôpitaux de Paris, University of Paris cité, 149 rue de Sèvres, 75015, Paris, France. nathalie.boddaert@aphp.fr ; Imagine Institute for Genetic Diseases, INSERM U1163, Paris, France. nathalie.boddaert@aphp.fr
The longitudinal evolution of cerebral blood flow in children with tuberous sclerosis assessed by arterial spin labeling magnetic resonance imaging may be related to cognitive performance.
Publication date :
January 2023
Journal title :
European Radiology
ISSN :
0938-7994
eISSN :
1432-1084
Publisher :
Springer Science and Business Media Deutschland GmbH, Germany
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