Early-Onset Glaucoma in egl1 Mice Homozygous for Pitx2 Mutation

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dc.creatorKodati, Bindu
dc.creatorMerchant, Shawn A.
dc.creatorMillar, J. Cameron
dc.creatorLiu, Yang
dc.date.accessioned2022-09-30T17:34:19Z
dc.date.available2022-09-30T17:34:19Z
dc.date.issued2022-02-22
dc.description.abstractMutations in PITX2 cause Axenfeld-Rieger syndrome, with congenital glaucoma as an ocular feature. The egl1 mouse strain carries a chemically induced Pitx2 mutation and develops early-onset glaucoma. In this study, we characterized the glaucomatous features in egl1 mice. The eyes of egl1 and C57BL/6J control mice were assessed by slit lamp examination, total aqueous humor outflow facility, intraocular pressure (IOP) measurement, pattern electroretinography (PERG) recording, and histologic and immunohistochemistry assessment beginning at 3 weeks and up to 12 months of age. The egl1 mice developed elevated IOP as early as 4 weeks old. The IOP elevation was variable and asymmetric within and between the animals. The aqueous humor outflow facility was significantly reduced in 12-month-old animals. PERG detected a decreased response at 2 weeks after the development of IOP elevation. Retinal ganglion cell (RGC) loss was detected after 8 weeks of IOP elevation. Slit lamp and histologic evaluation revealed corneal opacity, iridocorneal adhesions (anterior synechiae), and ciliary body atrophy in egl1 mice. Immunohistochemistry assessment demonstrated glial cell activation and RGC axonal injury in response to IOP elevation. These results show that the eyes of egl1 mice exhibit anterior segment dysgenesis and early-onset glaucoma. The egl1 mouse strain may represent a useful model for the study of congenital glaucoma.
dc.description.sponsorshipThis research was funded by the Knights Templar Eye Foundation grant number [2021-7], and the APC was funded by Department of Pharmacology and Neuroscience, University of North Texas Health Science Center.
dc.identifier.citationKodati, B., Merchant, S. A., Millar, J. C., & Liu, Y. (2022). Early-Onset Glaucoma in egl1 Mice Homozygous for Pitx2 Mutation. Biomedicines, 10(3), 516. https://doi.org/10.3390/biomedicines10030516
dc.identifier.issn2227-9059
dc.identifier.issue3
dc.identifier.urihttps://hdl.handle.net/20.500.12503/31827
dc.identifier.volume10
dc.publisherMDPI
dc.relation.urihttps://doi.org/10.3390/biomedicines10030516
dc.rights.holder© 2022 by the authors.
dc.rights.licenseAttribution 4.0 International (CC BY 4.0)
dc.rights.urihttp://creativecommons.org/licenses/by/4.0/
dc.sourceBiomedicines
dc.subjectPitx2
dc.subjectglaucoma
dc.subjectmouse model
dc.titleEarly-Onset Glaucoma in egl1 Mice Homozygous for Pitx2 Mutation
dc.typeArticle
dc.type.materialtext

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