Inherited retinal diseases: linking genes, disease-causing variants, and relevant therapeutic modalities

N Schneider, Y Sundaresan, P Gopalakrishnan… - Progress in retinal and …, 2022 - Elsevier
Inherited retinal diseases (IRDs) are a clinically complex and heterogenous group of visual
impairment phenotypes caused by pathogenic variants in at least 277 nuclear and …

The essential role of docosahexaenoic acid and its derivatives for retinal integrity

D Swinkels, M Baes - Pharmacology & Therapeutics, 2023 - Elsevier
The fatty acid composition of photoreceptor outer segment (POS) phospholipids diverges
from other membranes, being highly enriched in polyunsaturated fatty acids (PUFAs). The …

Causes and consequences of inherited cone disorders

S Roosing, AAHJ Thiadens, CB Hoyng… - Progress in retinal and …, 2014 - Elsevier
Hereditary cone disorders (CDs) are characterized by defects of the cone photoreceptors or
retinal pigment epithelium underlying the macula, and include achromatopsia (ACHM), cone …

Understanding the roles of very-long-chain polyunsaturated fatty acids (VLC-PUFAs) in eye health

U Nwagbo, PS Bernstein - Nutrients, 2023 - mdpi.com
Lipids serve many roles in the neural system, from synaptic stabilization and signaling to
DNA regulation and neuroprotection. They also regulate inflammatory responses, maintain …

Adiponectin receptor 1 conserves docosahexaenoic acid and promotes photoreceptor cell survival

DS Rice, JM Calandria, WC Gordon, B Jun… - Nature …, 2015 - nature.com
The identification of pathways necessary for photoreceptor and retinal pigment epithelium
(RPE) function is critical to uncover therapies for blindness. Here we report the discovery of …

ELOVL5 mutations cause spinocerebellar ataxia 38

E Di Gregorio, B Borroni, E Giorgio, D Lacerenza… - The American Journal of …, 2014 - cell.com
Spinocerebellar ataxias (SCAs) are a heterogeneous group of autosomal-dominant
neurodegenerative disorders involving the cerebellum and 23 different genes. We mapped …

Photoreceptor metabolic reprogramming: current understanding and therapeutic implications

WW Pan, TJ Wubben, CG Besirli - Communications biology, 2021 - nature.com
Acquired and inherited retinal disorders are responsible for vision loss in an increasing
proportion of individuals worldwide. Photoreceptor (PR) death is central to the vision loss …

Expanding the clinical phenotype associated with ELOVL4 mutation: study of a large French-Canadian family with autosomal dominant spinocerebellar ataxia and …

M Cadieux-Dion, M Turcotte-Gauthier, A Noreau… - JAMA …, 2014 - jamanetwork.com
Importance The autosomal dominant spinocerebellar ataxias (SCAs) are a complex group of
neurodegenerative disorders with significant genetic heterogeneity. Despite the …

Palmitic acid lipotoxicity in microglia cells is ameliorated by unsaturated fatty acids

CJ Urso, H Zhou - International Journal of Molecular Sciences, 2021 - mdpi.com
Obesity and metabolic syndrome are associated with cognitive decline and dementia.
Palmitic acid (PA) is increased in the cerebrospinal fluid of obese patients with cognitive …

[HTML][HTML] ELOVL4: Very long-chain fatty acids serve an eclectic role in mammalian health and function

BR Hopiavuori, RE Anderson, MP Agbaga - Progress in retinal and eye …, 2019 - Elsevier
Abstract ELOngation of Very Long chain fatty acids-4 (ELOVL4) is an elongase responsible
for the biosynthesis of very long chain (VLC,≥ C28) saturated (VLC-SFA) and …