Implications of circulating neurofilaments for spinal muscular atrophy treatment early in life: A case series.

Biomarkers Gene Therapy Neurogenetics Neuromuscular diseases SMA

Journal

Molecular therapy. Methods & clinical development
ISSN: 2329-0501
Titre abrégé: Mol Ther Methods Clin Dev
Pays: United States
ID NLM: 101624857

Informations de publication

Date de publication:
10 Dec 2021
Historique:
received: 22 08 2021
revised: 06 10 2021
accepted: 26 10 2021
pubmed: 3 12 2021
medline: 3 12 2021
entrez: 2 12 2021
Statut: epublish

Résumé

This longitudinal cohort study aimed to determine whether circulating neurofilaments (NFs) can monitor response to molecular therapies in newborns with spinal muscular atrophy (SMA; NCT02831296). We applied a mixed-effect model to examine differences in serum NF levels among healthy control infants (n = 13), untreated SMA infants (n = 68), and SMA infants who received the genetic therapies nusinersen and/or onasemnogene abeparvovec (n = 22). Increased NF levels were inversely associated with

Identifiants

pubmed: 34853799
doi: 10.1016/j.omtm.2021.10.011
pii: S2329-0501(21)00168-6
pmc: PMC8605296
doi:

Types de publication

Journal Article

Langues

eng

Pagination

524-538

Subventions

Organisme : NICHD NIH HHS
ID : R01 HD054599
Pays : United States
Organisme : NINDS NIH HHS
ID : R21 NS108015
Pays : United States

Informations de copyright

© 2021 The Authors.

Déclaration de conflit d'intérêts

C.R.R.A. and K.J.S. are inventors on a patent filed by Mass General Brigham that describes genome engineering technologies to treat SMA. C.R.R.A. holds stocks in CRISPR Therapeutics. K.J.S. is the recipient of a grant from Biogen and receives clinical trial funding from AveXis and Biogen. E.L.T. serves on a physical therapy advisory board for Biogen. M.P., J.S., and W.F. are current or former employees of Biogen and hold stock/stock options in Biogen. R.S., R.G., R.Z., E.J.E., E.R., E.A.A., S.S., and M.K. report no conflict of interest.

Auteurs

Christiano R R Alves (CRR)

Department of Neurology, Center for Genomic Medicine, Massachusetts General Hospital, Boston, MA, USA.

Marco Petrillo (M)

Biogen, Cambridge, MA, USA.
Takeda Pharmaceuticals, Lexington, MA, USA.

Rebecca Spellman (R)

Department of Neurology, Center for Genomic Medicine, Massachusetts General Hospital, Boston, MA, USA.

Reid Garner (R)

Department of Neurology, Center for Genomic Medicine, Massachusetts General Hospital, Boston, MA, USA.

Ren Zhang (R)

Department of Neurology, Center for Genomic Medicine, Massachusetts General Hospital, Boston, MA, USA.

Michael Kiefer (M)

Department of Neurology, Center for Genomic Medicine, Massachusetts General Hospital, Boston, MA, USA.
Department of Physical Therapy, School of Health and Rehabilitation Sciences, MGH Institute of Health Professions, Boston, MA, USA.

Sarah Simeone (S)

Department of Neurology, Center for Genomic Medicine, Massachusetts General Hospital, Boston, MA, USA.

Jihee Sohn (J)

Biogen, Cambridge, MA, USA.

Eric J Eichelberger (EJ)

Department of Neurology, Center for Genomic Medicine, Massachusetts General Hospital, Boston, MA, USA.

Emma Rodrigues (E)

Department of Neurology, Center for Genomic Medicine, Massachusetts General Hospital, Boston, MA, USA.

Elizabeth A Arruda (EA)

Department of Neurology, Center for Genomic Medicine, Massachusetts General Hospital, Boston, MA, USA.

Elise L Townsend (EL)

Department of Neurology, Center for Genomic Medicine, Massachusetts General Hospital, Boston, MA, USA.
Department of Physical Therapy, School of Health and Rehabilitation Sciences, MGH Institute of Health Professions, Boston, MA, USA.

Wildon Farwell (W)

Biogen, Cambridge, MA, USA.

Kathryn J Swoboda (KJ)

Department of Neurology, Center for Genomic Medicine, Massachusetts General Hospital, Boston, MA, USA.

Classifications MeSH