ArticleInvestigative ophthalmology & visual science2019
Selective Ablation of Megalin in the Retinal Pigment Epithelium Results in Megaophthalmos, Macromelanosome Formation and Severe Retina Degeneration.
Article in Investigative ophthalmology & visual science, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 14 papers.
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Who cites it
14 citing papers in PubMed, 25 citations in OpenAlex.
- 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
- Specialised features of melanosomes in health and disease in the retinal pigment epithelium.Frontiers in cell and developmental biology · 2025Review
- The role of inflammation in myopic retinopathy.Frontiers in ophthalmology · 2025Review
- Internalization of Angiotensin-(1-12) in Adult Retinal Pigment Epithelial-19 Cells.Journal of ocular pharmacology and therapeutics : the official journal of the Association for Ocular Pharmacology and Therapeutics · 2023Article
- 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
- Histology and clinical imaging lifecycle of black pigment in fibrosis secondary to neovascular age-related macular degeneration.Experimental eye research · 2022Article
- Expanding the phenotypic spectrum of mutations in LRP2: a novel candidate gene of non-syndromic familial comitant strabismus.Journal of translational medicine · 2021Article
- Endocytic Protein Defects in the Neural Crest Cell Lineage and Its Pathway Are Associated with Congenital Heart Defects.International journal of molecular sciences · 2021Review
- IMI 2021 Yearly Digest.Investigative ophthalmology & visual science · 2021Review
- Immunoglobulin G Is a Novel Substrate for the Endocytic Protein Megalin.The AAPS journal · 2021Article
- Retinal pigment epithelium polarity in health and blinding diseases.Current opinion in cell biology · 2020Review
Corrections and comments
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Authors and funding
6 authors at 3 institutions in 3 countries.
Funding
Abstract
Purpose: Mutations in the megalin-encoding gene, LRP2, cause high myopia as seen in patients suffering from Donnai-Barrow/facio-oculo-acoustico-renal syndrome. Megalin is present in both the nonpigmented epithelium of the ciliary body and in the RPE. In this study, we set out to establish an animal model to study the mechanisms underlying the ocular phenotype and to establish if high myopia/megaophthalmos is induced by postnatal megalin-deficiency in the RPE. Methods: Postnatal RPE-specific deletion of megalin was generated by crossing mice bearing a homozygous loxP-flanked Lrp2 allele with transgenic mice expressing the Cre recombinase driven by the BEST1 promotor. The model was investigated by immunohistologic techniques, and transmission electron microscopy. Results: Mice with postnatal RPE-specific loss of megalin developed a megaophthalmos phenotype with dramatic increase in ocular size and severe retinal thinning associated with compromised vision. This phenotype was present at postnatal day 14, indicating rapid development in the period from onset of BEST1 promotor activity at postnatal day 10. Additionally, RPE melanosomes exhibited abnormal size and morphology, suggested by electron tomography to be caused by fusion events between multiple melanosomes. Conclusions: Postnatal loss of megalin in the RPE induces dramatic and rapid ocular growth and retinal degeneration compatible with the high myopia observed in Donnai-Barrow patients. The morphologic changes of RPE melanosomes, believed to be largely inert and fully differentiated at birth, suggested a continued plasticity of mature melanosomes and a requirement for megalin to maintain their number and morphology.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.