% IMPORTANT: The following is UTF-8 encoded. This means that in the presence
% of non-ASCII characters, it will not work with BibTeX 0.99 or older.
% Instead, you should use an up-to-date BibTeX implementation like “bibtex8” or
% “biber”.
@ARTICLE{Hang:279369,
author = {Hang, Abraham and Shao, Andy and Shea, Michael and Roux,
Michel J and Imai-Leonard, Denise M and Adams, David J and
Amano, Takanori and Amarie, Oana V and Berberovic, Zorana
and Bour, Raphaël and Bower, Lynette and Leonard, Brian C
and Brown, Steve D and Cho, Soo Young and Clementson-Mobbs,
Sharon and D'Souza, Abigail J and Dickinson, Mary and
Eskandarian, Mohammad and Flenniken, Ann M and Fuchs, Helmut
and Gailus-Durner, Valerie and Heaney, Jason and Hérault,
Yann and Hrabe de Angelis, Martin and Hsu, Chih-Wei and Jin,
Shundan and Joynson, Russell and Kang, Yeon Kyung and Kim,
Haerim and Masuya, Hiroshi and Nam, Ki-Hoan and Noh, Hyuna
and Nutter, Lauryl M J and Palkova, Marcela and Prochazka,
Jan and Raishbrook, Miles Joseph and Riet, Fabrice and
Salazar, Jason and Seavitt, John Richard and Sedlacek,
Radislav and Selloum, Mohammed and Seo, Kyoung Yul and
Seong, Je Kyung and Shin, Hae-Sol and Shiroishi, Toshihiko
and Sorg, Tania and Stewart, Michelle and Tamura, Masaru and
Tolentino, Heather and Udensi, Uchechukwu and Wells, Sara
and Wurst, Wolfgang and Yoshiki, Atsushi and Meziane, Hamid
and Yiu, Glenn C and Sieving, Paul A and Lanoue, Louise and
Lloyd, K C Kent and McKerlie, Colin and Moshiri, Ala},
collaboration = {Consortium, International Mouse Phenotyping},
title = {{O}cular {P}henotyping of {K}nockout {M}ice {I}dentifies
{G}enes {A}ssociated {W}ith {L}ate {A}dult {R}etinal
{P}henotypes.},
journal = {Investigative ophthalmology $\&$ visual science},
volume = {66},
number = {6},
issn = {0146-0404},
address = {Rockville, Md.},
publisher = {ARVO},
reportid = {DZNE-2025-00746},
pages = {64},
year = {2025},
abstract = {Analyze phenotypic data from knockout mice with late-adult
retinal pathologic phenotypes to identify genes associated
with development of adult-onset retinal diseases.The
International Mouse Phenotyping Consortium (IMPC) database
was queried for genes associated with abnormal retinal
phenotypes in the late-adult knockout mouse pipeline (49-80
weeks postnatal age). We identified human orthologs and
performed protein-protein analysis and biological pathways
analysis with known inherited retinal disease (IRD) and
age-related macular degeneration (AMD) genes using Search
Tool for the Retrieval of Interacting Genes/Proteins
(STRING), PLatform for Analysis of single cell Eye in a Disk
(PLAE), Protein Analysis Through Evolutionary Relationships
(PANTHER), and Kyoto Encyclopedia of Genes and Genomes
(KEGG).Screening of 587 late-adult mouse genes yielded 12
with abnormal retinal phenotypes, which corresponded to 20
human orthologs. Three of the 12 mouse genes and two of the
20 human orthologs were previously implicated in retinal
pathology or physiology in a literature review. Although all
of the genes demonstrated retinal pathology when deleted
from the mouse genome, most do not have established roles in
human retinal disease. Furthermore, human protein-protein
analysis and biological pathway analysis yielded only a few
relationships between the candidate gene list and that of
known IRD and AMD genes, suggesting they may represent novel
retinal functions.We identified 12 mouse genes with
significant late-adult abnormal retinal pathology, eight of
which have not been previously implicated in either mouse or
human retinal physiology or pathology. These serve as novel
retinal disease gene candidates for late-onset retinal
disease.},
keywords = {Animals / Mice, Knockout / Mice / Phenotype / Disease
Models, Animal / Humans / Retina: pathology / Retina:
metabolism / Macular Degeneration: genetics / Eye Proteins:
genetics / Eye Proteins (NLM Chemicals)},
cin = {AG Wurst},
ddc = {610},
cid = {I:(DE-2719)1140001},
pnm = {352 - Disease Mechanisms (POF4-352)},
pid = {G:(DE-HGF)POF4-352},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:40548636},
pmc = {pmc:PMC12186831},
doi = {10.1167/iovs.66.6.64},
url = {https://pub.dzne.de/record/279369},
}