Restoration of functional PAX6 in aniridia patient iPSC-derived ocular tissue models using repurposed nonsense suppression drugs

DL Cunha, H Sarkar, J Eintracht, P Harding… - … Therapy-Nucleic Acids, 2023 - cell.com
Congenital aniridia is a rare, pan-ocular disease causing severe sight loss, with only
symptomatic intervention offered to patients. Approximately 40% of aniridia patients present …

Loss of the crumbs cell polarity complex disrupts epigenetic transcriptional control and cell cycle progression in the developing retina

N Owen, M Toms, Y Tian, L Toualbi… - The Journal of …, 2023 - Wiley Online Library
The crumbs cell polarity complex plays a crucial role in apical–basal epithelial polarity,
cellular adhesion, and morphogenesis. Homozygous variants in human CRB1 result in …

Disruption of common ocular developmental pathways in patient-derived optic vesicle models of microphthalmia

J Eintracht, N Owen, P Harding, M Moosajee - Stem Cell Reports, 2024 - cell.com
Genetic perturbations influencing early eye development can result in microphthalmia,
anophthalmia, and coloboma (MAC). Over 100 genes are associated with MAC, but little is …

[HTML][HTML] Efficient embryoid-based method to improve generation of optic vesicles from human induced pluripotent stem cells

J Eintracht, P Harding, DL Cunha, M Moosajee - F1000Research, 2022 - ncbi.nlm.nih.gov
Animal models have provided many insights into ocular development and disease, but they
remain suboptimal for understanding human oculogenesis. Eye development requires …

hiPSC-derived epidermal keratinocytes from ichthyosis patients show altered expression of cornification markers

D Lima Cunha, A Oram, R Gruber, R Plank… - International Journal of …, 2021 - mdpi.com
Inherited ichthyoses represent a large heterogeneous group of skin disorders characterised
by impaired epidermal barrier function and disturbed cornification. Current knowledge about …

Dominant RDH12-retinitis pigmentosa impairs photoreceptor development and cone function in retinal organoids

C Mejecase, Y Zhou, N Owen, P Soro-Barrio… - bioRxiv, 2024 - biorxiv.org
Retinal dehydrogenase 12 (RDH12) is a photoreceptor NADPH-dependent retinal
reductase enzyme, converting all-trans-retinal to all-trans-retinol. Heterozygous variants in …

[PDF][PDF] Restoration of functional PAX6 in aniridia patient iPSC-derived ocular tissue models using repurposed nonsense suppression drugs

DM de Lima Cunha, H Sarkar, J Eintracht, P Harding… - 2023 - repository.ubn.ru.nl
Congenital aniridia is a rare, pan-ocular disease causing severe sight loss, with only
symptomatic intervention offered to patients. Approximately 40% of aniridia patients present …

Patient-specific stem cell-derived optic vesicles reveal novel pathogenic variants and common disease mechanisms in microphthalmia

J Eintracht - 2023 - discovery.ucl.ac.uk
Human eye development is tightly regulated by gene regulatory networks (GRNs) that guide
early eye structures through morphogenetic events from optic vesicle formation to the …