Patient-derived iPSC modeling of rare neurodevelopmental disorders: Molecular pathophysiology and prospective therapies

KR Sabitha, AK Shetty, D Upadhya - Neuroscience & Biobehavioral …, 2021 - Elsevier
The pathological alterations that manifest during the early embryonic development due to
inherited and acquired factors trigger various neurodevelopmental disorders (NDDs) …

Drug screening and drug repositioning as promising therapeutic approaches for spinal muscular atrophy treatment

G Menduti, DM Rasà, S Stanga, M Boido - Frontiers in pharmacology, 2020 - frontiersin.org
Spinal muscular atrophy (SMA) is the most common genetic disease affecting infants and
young adults. Due to mutation/deletion of the survival motor neuron (SMN) gene, SMA is …

Maturation of human intestinal organoids in vitro facilitates colonization by commensal lactobacilli by reinforcing the mucus layer

YS Son, SJ Ki, R Thanavel, JJ Kim, MO Lee… - The FASEB …, 2020 - Wiley Online Library
Lactobacilli, which are probiotic commensal bacteria that mainly reside in the human small
intestine, have attracted attention for their ability to exert health‐promoting effects and …

RNA-targeted therapies and high-throughput screening methods

S Zhu, S Rooney, G Michlewski - International Journal of Molecular …, 2020 - mdpi.com
RNA-binding proteins (RBPs) are involved in regulating all aspects of RNA metabolism,
including processing, transport, translation, and degradation. Dysregulation of RNA …

Therapeutic interventions for spinal muscular atrophy: preclinical and early clinical development opportunities

L Servais, G Baranello, M Scoto, A Daron… - Expert Opinion on …, 2021 - Taylor & Francis
Introduction Spinal muscular atrophy (SMA) is an autosomal recessive neurodegenerative
neuromuscular disease that presents primarily in children. Abnormalities in the SMN1 gene …

Exploring motor neuron diseases using iPSC platforms

AE Johns, NJ Maragakis - Stem Cells, 2022 - academic.oup.com
The degeneration of motor neurons is a pathological hallmark of motor neuron diseases
(MNDs), but emerging evidence suggests that neuronal vulnerability extends well beyond …

In vitro modeling as a tool for testing therapeutics for spinal muscular atrophy and IGHMBP2-related disorders

JA Sierra-Delgado, S Sinha-Ray, A Kaleem… - Biology, 2023 - mdpi.com
Simple Summary Spinal Muscular Atrophy (SMA) is a genetic disease that can cause infant
mortality. It is typically caused by mutations in the SMN1 gene. On the other hand, mutations …

Stem cell models and gene targeting for human motor neuron diseases

Y Karpe, Z Chen, XJ Li - Pharmaceuticals, 2021 - mdpi.com
Motor neurons are large projection neurons classified into upper and lower motor neurons
responsible for controlling the movement of muscles. Degeneration of motor neurons results …

Spinal muscular atrophy modeling and treatment advances by induced pluripotent stem cells studies

R Adami, D Bottai - Stem Cell Reviews and Reports, 2019 - Springer
Abstract Spinal Muscular Atrophy (SMA) is a neurodegenerative disease characterized by
specific and predominantly lower motor neuron (MN) loss. SMA is the main reason for infant …

Primary Coenzyme Q10 Deficiency-7 and Pathogenic COQ4 Variants: Clinical Presentation, Biochemical Analyses, and Treatment

J **e, J Jiang, Q Guo - Frontiers in Genetics, 2022 - frontiersin.org
Primary Coenzyme Q10 Deficiency-7 (COQ10D7) is a rare mitochondrial disorder caused by
pathogenic COQ4 variants. In this review, we discuss the correlation of COQ4 genotypes …