title: Multimodal Assessment Reveals Late-Onset Hemispheric Shift of Language in a Child with Meningocerebral Dysplasia.
abstract: We report on a girl with progressive left frontal tissue destruction starting at the age of almost 8 years. She manifested acutely with epileptic seizures accompanied by Broca aphasia as well as transient right hemiparesis. Due to refractory epilepsy developing over the next years, which originated from the left frontal lobe, the decision was made to proceed to epilepsy surgery. By then, her language functions had recovered despite progressive left frontal tissue-destruction, raising the possibility of a hemispheric shift of language. Clinical functional magnetic resonance imaging (fMRI) was conducted to localize brain regions involved in language production. A complex pattern of clear right-hemispheric dominance, but with some left-sided contribution was found. However, a Wada test suggested the left hemisphere to be critical, seemingly contradicting fMRI. Invasive electroencephalogram recordings could reconcile these results by identifying the fMRI-detected, residual left-sided activation as being relevant for speech production. Only by combining the localizing information from fMRI with the information obtained by two invasive procedures could the unusual pattern of late-onset language reorganization be uncovered. This allowed for extensive left frontal resection, with histology confirming meningocerebral angiodysplasia. Postoperatively, language functions were preserved and seizure outcome was excellent. The implications of our findings for presurgical assessments in children are discussed.
authors.affiliation: Department of Pediatric Neurology and Developmental Medicine, University of Tübingen, Tübingen, Germany.
authors.forename: Anna
authors.initials: A
authors.lastname: Lorenzen
authors.affiliation: Department of Pediatric Neurology and Developmental Medicine, University of Tübingen, Tübingen, Germany.
authors.forename: Marko
authors.initials: M
authors.lastname: Wilke
authors.affiliation: Department of Pediatric Neurology and Developmental Medicine, University of Tübingen, Tübingen, Germany.
authors.forename: Michael
authors.initials: M
authors.lastname: Alber
authors.affiliation: Department of Neurosurgery, University of Tübingen, Tübingen, Germany.
authors.forename: Monika
authors.initials: M
authors.lastname: Milian
authors.affiliation: Division of Neuropathology, Department of Pathology, University of Tübingen, Tübingen, Germany.
authors.forename: Antje
authors.initials: A
authors.lastname: Bornemann
authors.affiliation: Department of Diagnostic and Interventional Neuroradiology, University of Tübingen, Tübingen, Germany.
authors.forename: Ulrike
authors.initials: U
authors.lastname: Ernemann
authors.affiliation: Department of Neurosurgery, University of Tübingen, Tübingen, Germany.
authors.forename: Sabine
authors.initials: S
authors.lastname: Rona
mesh_terms: D016888:Angiodysplasia / Q000150:complications / Q000473:pathology / Q000503:physiopathology / Q000601:surgery*; D001039:Aphasia, Broca / Q000209:etiology / Q000503:physiopathology*; D001927:Brain Diseases / Q000150:complications / Q000473:pathology / Q000503:physiopathology / Q000601:surgery*; D001931:Brain Mapping; D002648:Child; D004569:Electroencephalography; D017034:Epilepsy, Frontal Lobe / Q000209:etiology / Q000503:physiopathology / Q000601:surgery*; D005260:Female; D007839:Functional Laterality; D059907:Functional Neuroimaging; D006801:Humans; D007802:Language; D008279:Magnetic Resonance Imaging; D009473:Neuronal Plasticity; D010291:Paresis / Q000209:etiology