PXD030918

PXD030918 is an original dataset announced via ProteomeXchange.

Title	Effects of alpha-crystallin gene knockout on zebrafish lens development
Description	The alpha-crystallin small heat shock proteins contribute to the transparency and refractive properties of the vertebrate eye lens and prevent the protein aggregation that would otherwise produce lens cataracts, the leading cause of human blindness. There are conflicting data in the literature as to what role the alpha-crystallins may play in early lens development. In this study, we used CRISPR gene editing to produce zebrafish lines with null mutations for each of the three alpha-crystallin genes (cryaa, cryaba and cryabb). The absence of normal protein was confirmed by mass spectrometry, and lens phenotypes were assessed with differential interference contrast microscopy and histology. Loss of alpha A-crystallin produced a variety of lens defects with varying severity in larval lenses at 3 and 4 dpf but little substantial change in normal fiber cell denucleation. Loss of either alpha Ba- or full-length alpha Bb-crystallin produced no substantial lens defects. Mutation of each alpha-crystallin gene did not alter the expression levels of the remaining two, suggesting a lack of genetic compensation. These data confirm a developmental role for alphaA-crystallin in lens development, but the range of phenotype severity suggests that its loss simply increases the chance for defects and that the protein is not essential. Our finding that cryaba and cryabb mutants lack noticeable lens defects is congruent with insubstantial transcript levels in lens epithelial and fiber cells. Future experiments can explore the molecular consequences of cryaa mutation and causes of lens defects in this null mutant, as well as the roles of other genes in lens development and function.
HostingRepository	PanoramaPublic
AnnounceDate	2024-07-05
AnnouncementXML	Submission_2024-07-05_15:41:49.316.xml
DigitalObjectIdentifier
ReviewLevel	Peer-reviewed dataset
DatasetOrigin	Original dataset
RepositorySupport	Supported dataset by repository
PrimarySubmitter	Ashok Reddy
SpeciesList	scientific name: Danio rerio; NCBI TaxID: 7955;
ModificationList	Carbamidomethyl
Instrument	Q Exactive HF

Revision	Datetime	Status	ChangeLog Entry
0	2022-01-12 10:28:56	ID requested
1	2022-01-20 15:57:01	announced
⏵ 2	2024-07-05 15:41:50	announced	: Added PubMed Id

Posner M, Murray KL, Andrew B, Brdicka S, Roberts A, Franklin K, Hussen A, Kaye T, Kepp E, McDonald MS, Snodgrass T, Zientek K, David LL, -crystallin protein loss on zebrafish lens development. Exp Eye Res, 227():109358(2023) [pubmed]

submitter keyword: zebrafish, alpha-crystallin, lens, development, cataract, knockout

Ashok Reddy
contact affiliation	Oregon Health & Sciences University
contact email	reddyas@ohsu.edu
lab head
Ashok Reddy
contact affiliation	Oregon Health & Sciences University
contact email	reddyas@ohsu.edu
dataset submitter

Panorama Public dataset URI