ArticlePloS one2015
Foxg1-Cre Mediated Lrp2 Inactivation in the Developing Mouse Neural Retina, Ciliary and Retinal Pigment Epithelia Models Congenital High Myopia.
Article in PloS one, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 30 papers.
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Who cites it
30 citing papers in PubMed, 54 citations in OpenAlex.
- Multi-omics Mendelian randomisation reveals distinct molecular drivers of pathological myopia complications vs simple myopia.Journal of global health · 2026Article
- Photobiology in ophthalmology: from evolutionary adaptation to translational therapeutics.Japanese journal of ophthalmology · 2026Review
- Spontaneous Pathologic Myopia and Contributing Factors in Nonhuman Primate Models.Translational vision science & technology · 2026Article
- Degradable Suture-Free Scaffold to Induce Therapeutic Ridge Formation and to Control Eye Elongation: A Potential Treatment for Pathological Myopia.Translational vision science & technology · 2026Article
- Selective retinal neuron loss and impaired neurovascular support underlie myopic retinopathy in RPE-specific Lrp2-deficient mice.Frontiers in cellular neuroscience · 2026Article
- Independent Mutations in theInternational journal of molecular sciences · 2025Article
- Article
- The role of inflammation in myopic retinopathy.Frontiers in ophthalmology · 2025Review
- Article
- Spontaneously Myopic Guinea Pig: Model of Early Pathologic Myopia.Investigative ophthalmology & visual science · 2023Article
- Whole-Exome Sequencing of 21 Families: Candidate Genes for Early-Onset High Myopia.International journal of molecular sciences · 2023Article
- Analysis of Bilaterality and Symmetry of Posterior Staphyloma in High Myopia.Diagnostics (Basel, Switzerland) · 2023Article
- Myopia control in Mendelian forms of myopia.Ophthalmic & physiological optics : the journal of the British College of Ophthalmic Opticians (Optometrists) · 2023Article
- Two novel variations inFrontiers in genetics · 2023Article
- Lhx2 is a progenitor-intrinsic modulator of Sonic Hedgehog signaling during early retinal neurogenesis.eLife · 2022Article
- Vascular endothelial growth factor from retinal pigment epithelium is essential in choriocapillaris and axial length maintenance.PNAS nexus · 2022Article
- Postnatal eye size in mice is controlled by SREBP2-mediated transcriptional repression of Lrp2 and Bmp2.Development (Cambridge, England) · 2022Article
- Quantitative proteomics analysis of human vitreous in rhegmatogenous retinal detachment associated with choroidal detachment by data-independent acquisition mass spectrometry.Molecular and cellular biochemistry · 2022Article
- Expanding the phenotypic spectrum of mutations in LRP2: a novel candidate gene of non-syndromic familial comitant strabismus.Journal of translational medicine · 2021Article
- IMI Pathologic Myopia.Investigative ophthalmology & visual science · 2021Review
Corrections and comments
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Authors and funding
17 authors at 9 institutions in 3 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Myopia is a common ocular disorder generally due to increased axial length of the eye-globe. Its extreme form high myopia (HM) is a multifactorial disease leading to retinal and scleral damage, visual impairment or loss and is an important health issue. Mutations in the endocytic receptor LRP2 gene result in Donnai-Barrow (DBS) and Stickler syndromes, both characterized by HM. To clearly establish the link between Lrp2 and congenital HM we inactivated Lrp2 in the mouse forebrain including the neural retina and the retinal and ciliary pigment epithelia. High resolution in vivo MRI imaging and ophthalmological analyses showed that the adult Lrp2-deficient eyes were 40% longer than the control ones mainly due to an excessive elongation of the vitreal chamber. They had an apparently normal intraocular pressure and developed chorioretinal atrophy and posterior scleral staphyloma features reminiscent of human myopic retinopathy. Immunomorphological and ultrastructural analyses showed that increased eye lengthening was first observed by post-natal day 5 (P5) and that it was accompanied by a rapid decrease of the bipolar, photoreceptor and retinal ganglion cells, and eventually the optic nerve axons. It was followed by scleral thinning and collagen fiber disorganization, essentially in the posterior pole. We conclude that the function of LRP2 in the ocular tissues is necessary for normal eye growth and that the Lrp2-deficient eyes provide a unique tool to further study human HM.
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