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      Quantitative magnetic resonance imaging evidence for altered structural remodeling of the temporal lobe in West syndrome

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          Summary

          Objective

          To explore the structure–function relation of the temporal lobe in newly diagnosed West syndrome of unknown cause ( uWS).

          Methods

          Quantitative magnetic resonance imaging (three‐dimensional [3D] structural MRI and diffusion tensor imaging [DTI]) was analyzed using voxel‐based morphometry ( VBM) and tract‐based spatial statistics ( TBSS) in 22 patients and healthy age‐matched controls. The electrophysiologic responsiveness of the temporal lobe was measured using the N100 auditory event‐related potential ( aERP) to a repeated 1,000 Hz tone. Neurocognitive function was assessed using the Bayley Scales of Infant Development, Second Edition ( BSID‐ II). Tests followed first‐line treatment with vigabatrin (17 patients) or high‐dose oral prednisolone (5 patients).

          Results

          Total temporal lobe volume was similar in patients and controls. Patients had a smaller temporal stem ( TS) (p < 0.0001) and planum temporale ( PT) (p = 0.029) bilaterally. TS width asymmetry with a larger right‐sided width in controls was absent in patients (p = 0.033). PT asymmetry was present in both groups, being larger on the right (p = 0.048). VBM gray matter volume was increased at the left temporal lobe (superior and middle temporal gyri, the peri‐rhinal cortex, and medial temporal lobe) (p < 0.005, family wise error‐corrected). VBM gray matter volume correlated with the duration of infantile spasms (Pearson's r = −0.630, p = 0.009). DTI metrics did not differ between patients and controls on TBSS. Mean BSID‐ II scores were lower (p < 0.001) and auditory N100 ERP attenuated less in patients than in controls (p = 0.002).

          Significance

          The functional networking and white matter development of the temporal lobe are impaired following infantile spasms. Treatment may promote structural plasticity within the temporal lobe following infantile spasms, manifest as increased gray matter volume on VBM. It remains to be investigated further whether this predicts patients' long‐term cognitive difficulties.

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          Most cited references51

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          Brain mechanisms in early language acquisition.

          Patricia K. Kuhl (2010)
          The last decade has produced an explosion in neuroscience research examining young children's early processing of language. Noninvasive, safe functional brain measurements have now been proven feasible for use with children starting at birth. The phonetic level of language is especially accessible to experimental studies that document the innate state and the effect of learning on the brain. The neural signatures of learning at the phonetic level can be documented at a remarkably early point in development. Continuity in linguistic development from infants' earliest brain responses to phonetic stimuli is reflected in their language and prereading abilities in the second, third, and fifth year of life, a finding with theoretical and clinical impact. There is evidence that early mastery of the phonetic units of language requires learning in a social context. Neuroscience on early language learning is beginning to reveal the multiple brain systems that underlie the human language faculty. 2010 Elsevier Inc. All rights reserved.
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            A proposal for case definitions and outcome measures in studies of infantile spasms and West syndrome: consensus statement of the West Delphi group.

            Andrew Lux, Colin Osborne (2004)
            To reach a broad consensus on case definitions, outcomes, and outcome measures that will ease future study design and facilitate comparison of data from different studies of infantile spasms and West syndrome. Persons who had recently presented or published first-author original research in this field were invited to participate in an e-mail Delphi process and to invite other investigators or clinicians who they thought might participate. The process consisted of six rounds, anonymous except to the facilitator. In total, responses were received from 46 participants. The final statement was approved by 31 participants from 15 countries. It concluded that the primary clinical outcome, cessation of spasms, should denote absence of witnessed spasms from within 14 days of commencement of treatment, and for > or =28 consecutive days from the last witnessed spasm. Primary electroclinical outcome denotes cessation of spasms with resolution of hypsarrhythmia. West syndrome should be a defined subset of the syndrome of infantile spasms. An infantile spasms single-spasm variant should be recognized. Ways are suggested of handling subtle spasms in the context of clinical studies. It proposes a standard for reporting modifying and atypical features of hypsarrhythmia, a minimal set of baseline characteristics and outcomes that should be reported in trials of infantile spasms, and suggests a standard definition of relapse. Consensus was not reached on a definition of hypsarrhythmia. We reached a clear consensus on many aspects of study design for the investigation of infantile spasms, although incomplete consensus was found on how to define EEG criteria.
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              Characterization of atypical language activation patterns in focal epilepsy.

              Madison M. Berl, William Theodore, Lauren Zimmaro … (2013)
              Functional magnetic resonance imaging is sensitive to the variation in language network patterns. Large populations are needed to rigorously assess atypical patterns, which, even in neurological populations, are a minority. We studied 220 patients with focal epilepsy and 118 healthy volunteers who performed an auditory description decision task. We compared a data-driven hierarchical clustering approach to the commonly used a priori laterality index (LI) threshold (LI < 0.20 as atypical) to classify language patterns within frontal and temporal regions of interest. We explored (n = 128) whether IQ varied with different language activation patterns. The rate of atypical language among healthy volunteers (2.5%) and patients (24.5%) agreed with previous studies; however, we found 6 patterns of atypical language: a symmetrically bilateral, 2 unilaterally crossed, and 3 right dominant patterns. There was high agreement between classification methods, yet the cluster analysis revealed novel correlations with clinical features. Beyond the established association of left-handedness, early seizure onset, and vascular pathology with atypical language, cluster analysis identified an association of handedness with frontal lateralization, early seizure onset with temporal lateralization, and left hemisphere focus with a unilateral right pattern. Intelligence quotient was not significantly different among patterns. Language dominance is a continuum; however, our results demonstrate meaningful thresholds in classifying laterality. Atypical language patterns are less frequent but more variable than typical language patterns, posing challenges for accurate presurgical planning. Language dominance should be assessed on a regional rather than hemispheric basis, and clinical characteristics should inform evaluation of atypical language dominance. Reorganization of language is not uniformly detrimental to language functioning. © 2014 American Neurological Association.
              Article 0 comments Cited 65 times – based on 0 reviews     Review now
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                Author and article information

                Journal
                Journal ID (nlm-ta): Epilepsia
                Journal ID (iso-abbrev): Epilepsia
                Journal ID (doi): 10.1111/(ISSN)1528-1167
                Journal ID (publisher-id): EPI
                Title: Epilepsia
                Publisher: John Wiley and Sons Inc. (Hoboken )
                ISSN (Print): 0013-9580
                ISSN (Electronic): 1528-1167
                Publication date (Electronic): 02 March 2015
                Publication date (Print): April 2015
                Volume: 56
                Issue: 4 ( doiID: 10.1111/epi.2015.56.issue-4 )
                Pages: 608-616
                Affiliations
                [ 1 ]Young Epilepsy SurreyUnited Kingdom
                [ 2 ] Department of Clinical NeurophysiologyGreat Ormond Street Hospital for Children NHS Trust LondonUnited Kingdom
                [ 3 ] Neurosciences UnitUniversity College London Institute of Child Health LondonUnited Kingdom
                [ 4 ] The Functional Imaging LaboratoryUniversity College London LondonUnited Kingdom
                [ 5 ] Department of NeuroradiologyGreat Ormond Street Hospital for Children NHS Trust LondonUnited Kingdom
                [ 6 ] RCS Unit of BiophysicsUniversity College London Institute of Child Health LondonUnited Kingdom
                [ 7 ] Department of NeurologyGreat Ormond Street Hospital for Children NHS Trust LondonUnited Kingdom
                [ 8 ] Department of Neurological SciencesUniversity of Vermont Burlington VermontU.S.A
                [ 9 ] Centre for Developmental Cognitive NeurosciencesUniversity College London LondonUnited Kingdom
                Author notes
                [*] [* ]Address correspondence to Tangunu Fosi, UCL Institute of Child Health, 4‐5 Long Yard, London WC1N 3LU, U.K. E‐mail: sejjtfo@ 123456ucl.ac.uk
                Article
                Publisher ID: EPI12907
                DOI: 10.1111/epi.12907
                PMC ID: 5006860
                PubMed ID: 25802930
                SO-VID: 790c0a6c-136c-4aa2-9cf1-0dac805126d7
                Copyright © © 2015 The Authors. Epilepsia published by Wiley Periodicals, Inc. on behalf of International League Against Epilepsy.
                License:

                This is an open access article under the terms of the Creative Commons Attribution‐NonCommercial‐NoDerivs License, which permits use and distribution in any medium, provided the original work is properly cited, the use is non‐commercial and no modifications or adaptations are made.

                History
                Date accepted : 30 November 2014
                Page count
                Pages: 9
                Funding
                Funded by: Department of Health's NIHR Biomedical Research Centre
                Categories
                Subject: Full‐Length Original Research
                Subject: Full‐length Original Research
                Custom metadata
                source-schema-version-number 2.0
                component-id epi12907
                cover-date April 2015
                details-of-publishers-convertor Converter:WILEY_ML3GV2_TO_NLMPMC version:4.9.4 mode:remove_FC converted:31.08.2016

                ScienceOpen disciplines: Neurology
                Keywords: west syndrome,temporal lobe,auditory event–related potential,quantitative magnetic resonance imaging
                Data availability:
                ScienceOpen disciplines: Neurology
                Keywords: west syndrome, temporal lobe, auditory event–related potential, quantitative magnetic resonance imaging

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