Ciliopathies

F Hildebrandt, T Benzing… - New England Journal of …, 2011 - Mass Medical Soc
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Ciliopathies: an expanding disease spectrum

AM Waters, PL Beales - Pediatric nephrology, 2011 - Springer
Ciliopathies comprise a group of disorders associated with genetic mutations encoding
defective proteins, which result in either abnormal formation or function of cilia. As cilia are a …

Development of a gene-editing approach to restore vision loss in Leber congenital amaurosis type 10

ML Maeder, M Stefanidakis, CJ Wilson, R Baral… - Nature medicine, 2019 - nature.com
Leber congenital amaurosis type 10 is a severe retinal dystrophy caused by mutations in the
CEP290 gene,. We developed EDIT-101, a candidate genome-editing therapeutic, to …

A platform for experimental precision medicine: The extended BXD mouse family

DG Ashbrook, D Arends, P Prins, MK Mulligan, S Roy… - Cell systems, 2021 - cell.com
The challenge of precision medicine is to model complex interactions among DNA variants,
phenotypes, development, environments, and treatments. We address this challenge by …

Identification and correction of mechanisms underlying inherited blindness in human iPSC-derived optic cups

DA Parfitt, A Lane, CM Ramsden, AJF Carr, PM Munro… - Cell stem cell, 2016 - cell.com
Leber congenital amaurosis (LCA) is an inherited retinal dystrophy that causes childhood
blindness. Photoreceptors are especially sensitive to an intronic mutation in the cilia-related …

CRISPR/Cas9-mediated genome editing as a therapeutic approach for Leber congenital amaurosis 10

GX Ruan, E Barry, D Yu, M Lukason, SH Cheng… - Molecular therapy, 2017 - cell.com
As the most common subtype of Leber congenital amaurosis (LCA), LCA10 is a severe
retinal dystrophy caused by mutations in the CEP290 gene. The most frequent mutation …

Effect of genome size on AAV vector packaging

Z Wu, H Yang, P Colosi - Molecular Therapy, 2010 - cell.com
Adeno-associated virus (AAV) vector genomes have been limited to 5 kilobases (kb) in
length because their packaging limit was thought to be similar to the size of the parent AAV …

When cilia go bad: cilia defects and ciliopathies

M Fliegauf, T Benzing, H Omran - Nature reviews Molecular cell biology, 2007 - nature.com
Defects in the function of cellular organelles such as peroxisomes, lysosomes and
mitochondria are well-known causes of human diseases. Recently, another organelle has …

Leber congenital amaurosis: genes, proteins and disease mechanisms

AI Den Hollander, R Roepman, RK Koenekoop… - Progress in retinal and …, 2008 - Elsevier
Leber congenital amaurosis (LCA) is the most severe retinal dystrophy causing blindness or
severe visual impairment before the age of 1 year. Linkage analysis, homozygosity mapping …

[HTML][HTML] Mapping the NPHP-JBTS-MKS protein network reveals ciliopathy disease genes and pathways

L Sang, JJ Miller, KC Corbit, RH Giles, MJ Brauer… - Cell, 2011 - cell.com
Summary Nephronophthisis (NPHP), Joubert (JBTS), and Meckel-Gruber (MKS) syndromes
are autosomal-recessive ciliopathies presenting with cystic kidneys, retinal degeneration …