Understanding the Ultra-Rare Disease Autosomal Dominant Leukodystrophy: an Updated Review on Morpho-Functional Alterations Found in Experimental Models.


Journal

Molecular neurobiology
ISSN: 1559-1182
Titre abrégé: Mol Neurobiol
Pays: United States
ID NLM: 8900963

Informations de publication

Date de publication:
Nov 2023
Historique:
received: 19 12 2022
accepted: 22 06 2023
medline: 28 9 2023
pubmed: 14 7 2023
entrez: 14 7 2023
Statut: ppublish

Résumé

Autosomal dominant leukodystrophy (ADLD) is an ultra-rare, slowly progressive, and fatal neurodegenerative disorder associated with the loss of white matter in the central nervous system (CNS). Several years after its first clinical description, ADLD was found to be caused by coding and non-coding variants in the LMNB1 gene that cause its overexpression in at least the brain of patients. LMNB1 encodes for Lamin B1, a protein of the nuclear lamina. Lamin B1 regulates many cellular processes such as DNA replication, chromatin organization, and senescence. However, its functions have not been fully characterized yet. Nevertheless, Lamin B1 together with the other lamins that constitute the nuclear lamina has firstly the key role of maintaining the nuclear structure. Being the nucleus a dynamic system subject to both biochemical and mechanical regulation, it is conceivable that changes to its structural homeostasis might translate into functional alterations. Under this light, this review aims at describing the pieces of evidence that to date have been obtained regarding the effects of LMNB1 overexpression on cellular morphology and functionality. Moreover, we suggest that further investigation on ADLD morpho-functional consequences is essential to better understand this complex disease and, possibly, other neurological disorders affecting CNS myelination.

Identifiants

pubmed: 37450245
doi: 10.1007/s12035-023-03461-1
pii: 10.1007/s12035-023-03461-1
pmc: PMC10533580
doi:

Types de publication

Journal Article Review

Langues

eng

Sous-ensembles de citation

IM

Pagination

6362-6372

Informations de copyright

© 2023. The Author(s).

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Auteurs

Irene Neri (I)

Cellular Signalling Laboratory, Anatomy Centre, Department of Biomedical and Neuromotor Sciences (DIBINEM), University of Bologna, 40126, Bologna, Italy.

Giulia Ramazzotti (G)

Cellular Signalling Laboratory, Anatomy Centre, Department of Biomedical and Neuromotor Sciences (DIBINEM), University of Bologna, 40126, Bologna, Italy.

Sara Mongiorgi (S)

Cellular Signalling Laboratory, Anatomy Centre, Department of Biomedical and Neuromotor Sciences (DIBINEM), University of Bologna, 40126, Bologna, Italy.

Isabella Rusciano (I)

Cellular Signalling Laboratory, Anatomy Centre, Department of Biomedical and Neuromotor Sciences (DIBINEM), University of Bologna, 40126, Bologna, Italy.

Marianna Bugiani (M)

Department of Pathology, Amsterdam University Medical Centers, Vrije Universiteit and Amsterdam Neuroscience, 1105, Amsterdam, The Netherlands.

Luciano Conti (L)

Department of Cellular, Computational, and Integrative Biology (CIBIO), Università Degli Studi Di Trento, 38123, Povo-Trento, Italy.

Margot Cousin (M)

Center for Individualized Medicine and Department of Clinical Genomics, Mayo Clinic, Rochester, MN, 55905, USA.

Elisa Giorgio (E)

Department of Molecular Medicine, University of Pavia, 27100, Pavia, Italy.
Medical Genetics Unit, IRCCS Mondino Foundation, 27100, Pavia, Italy.

Quasar S Padiath (QS)

Department of Human Genetics, Graduate School of Public Health, University of Pittsburgh, Pittsburgh, PA, 15261, USA.

Giovanna Vaula (G)

Department of Neuroscience, Azienda Ospedaliera-Universitaria Città della Salute e della Scienza, 10126, Turin, Italy.

Pietro Cortelli (P)

IRCCS, Istituto Di Scienze Neurologiche Di Bologna, 40139, Bologna, Italy.
Department of Biomedical and Neuromotor Sciences (DIBINEM), University of Bologna, 40126 , Bologna, Italy.

Lucia Manzoli (L)

Cellular Signalling Laboratory, Anatomy Centre, Department of Biomedical and Neuromotor Sciences (DIBINEM), University of Bologna, 40126, Bologna, Italy.

Stefano Ratti (S)

Cellular Signalling Laboratory, Anatomy Centre, Department of Biomedical and Neuromotor Sciences (DIBINEM), University of Bologna, 40126, Bologna, Italy. stefano.ratti@unibo.it.

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Classifications MeSH