Can Disruption of Basal Ganglia-Thalamocortical Circuit in Wilson Disease Be Associated with Juvenile Myoclonic Epilepsy Phenotype?

Wilson disease (WD) fMRI globus pallidus internus (GPi) myoclonic epilepsy thalamocortical circuit

Journal

Brain sciences
ISSN: 2076-3425
Titre abrégé: Brain Sci
Pays: Switzerland
ID NLM: 101598646

Informations de publication

Date de publication:
26 Apr 2022
Historique:
received: 30 03 2022
revised: 20 04 2022
accepted: 22 04 2022
entrez: 28 5 2022
pubmed: 29 5 2022
medline: 29 5 2022
Statut: epublish

Résumé

In this paper, we describe the multimodal MRI findings in a patient with Wilson disease and a seizure disorder, characterized by an electroclinical picture resembling juvenile myoclonic epilepsy. The brain structural MRI showed a deposition of ferromagnetic materials in the basal ganglia, with marked hypointensities in T2-weighted images of globus pallidus internus bilaterally. A resting-state fMRI study revealed increased functional connectivity in the patient, compared to control subjects, in the following networks: (1) between the primary motor cortex and several cortical regions, including the secondary somatosensory cortex and (2) between the globus pallidus and the thalamo-frontal network. These findings suggest that globus pallidus alterations, due to metal accumulation, can lead to a reduction in the normal globus pallidus inhibitory tone on the thalamo-(motor)-cortical pathway. This, in turn, can result in hyperconnectivity in the motor cortex circuitry, leading to myoclonus and tonic-clonic seizures. We suppose that, in this patient, Wilson disease generated a 'lesion model' of myoclonic epilepsy.

Identifiants

pubmed: 35624941
pii: brainsci12050553
doi: 10.3390/brainsci12050553
pmc: PMC9138607
pii:
doi:

Types de publication

Case Reports

Langues

eng

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Auteurs

Jessica Rossi (J)

Clinical and Experimental Medicine PhD Program, University of Modena and Reggio Emilia, 41121 Modena, Italy.

Francesco Cavallieri (F)

Neurology Unit, Neuromotor & Rehabilitation Department, Azienda USL-IRCCS of Reggio Emilia, 42123 Reggio Emilia, Italy.

Giada Giovannini (G)

Clinical and Experimental Medicine PhD Program, University of Modena and Reggio Emilia, 41121 Modena, Italy.
Unit of Neurology, OCB, Azienda Ospedaliero Universitaria di Modena, 41126 Modena, Italy.

Francesca Benuzzi (F)

Department of Biomedical, Metabolic and Neural Sciences, University of Modena and Reggio Emilia, 41125 Modena, Italy.

Daniela Ballotta (D)

Department of Biomedical, Metabolic and Neural Sciences, University of Modena and Reggio Emilia, 41125 Modena, Italy.

Anna Elisabetta Vaudano (AE)

Unit of Neurology, OCB, Azienda Ospedaliero Universitaria di Modena, 41126 Modena, Italy.
Department of Biomedical, Metabolic and Neural Sciences, University of Modena and Reggio Emilia, 41125 Modena, Italy.

Francesca Ferrara (F)

Internal Medicine Unit, Centre for Hemochromatosis and Heredometabolic Liver Diseases, Policlinico, Azienda Ospedaliero Universitaria di Modena, 41124 Modena, Italy.

Sara Contardi (S)

IRCCS Istituto delle Scienze Neurologiche di Bologna, UOC Neurologia e Rete Stroke Metropolitana, Ospedale Maggiore, 40133 Bologna, Italy.

Antonello Pietrangelo (A)

Internal Medicine Unit, Centre for Hemochromatosis and Heredometabolic Liver Diseases, Policlinico, Azienda Ospedaliero Universitaria di Modena, 41124 Modena, Italy.
Department of Medical and Surgical Sciences, University of Modena and Reggio Emilia, 41125 Modena, Italy.

Elena Corradini (E)

Internal Medicine Unit, Centre for Hemochromatosis and Heredometabolic Liver Diseases, Policlinico, Azienda Ospedaliero Universitaria di Modena, 41124 Modena, Italy.
Department of Medical and Surgical Sciences, University of Modena and Reggio Emilia, 41125 Modena, Italy.

Fausta Lui (F)

Department of Biomedical, Metabolic and Neural Sciences, University of Modena and Reggio Emilia, 41125 Modena, Italy.

Stefano Meletti (S)

Unit of Neurology, OCB, Azienda Ospedaliero Universitaria di Modena, 41126 Modena, Italy.
Department of Biomedical, Metabolic and Neural Sciences, University of Modena and Reggio Emilia, 41125 Modena, Italy.

Classifications MeSH