ArticleInvestigative ophthalmology & visual science2016
Loss of Zebrafish Mfrp Causes Nanophthalmia, Hyperopia, and Accumulation of Subretinal Macrophages.
Article in Investigative ophthalmology & visual science, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 20 papers.
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Who cites it
20 citing papers in PubMed, 39 citations in OpenAlex.
- Environmental context modulates sociability in ube3a zebrafish mutants via alterations in sensory pathways.Molecular psychiatry · 2026Article
- The Zebrafish as a Model for Ocular Translational Research: From Retinal Repair to Regeneration.Cells · 2025Review
- Germline Disruption of Retinal Pigment Epithelium-Expressed Zebrafish rlbp1bFASEB journal : official publication of the Federation of American Societies for Experimental Biology · 2025Article
- Optical Coherence Tomography Is a Promising Tool for Zebrafish-Based Research-A Review.Bioengineering (Basel, Switzerland) · 2022Review
- Genetic and Neurological Deficiencies in the Visual System ofInternational journal of molecular sciences · 2022Article
- Genetic Interaction betweenInternational journal of molecular sciences · 2022Article
- A Novel Mutation in the Membrane Frizzled-Related Protein Gene for Posterior Microphthalmia, Non-pigmented Retinitis Pigmentosa, Optic Nerve Drusen, and Retinoschisis in a Consanguineous Family.Frontiers in medicine · 2022Article
- Ablation of mpeg+ Macrophages Exacerbates mfrp-Related Hyperopia.Investigative ophthalmology & visual science · 2021Article
- Retinal Stem Cell 'Retirement Plans': Growth, Regulation and Species Adaptations in the Retinal Ciliary Marginal Zone.International journal of molecular sciences · 2021Review
- Identification of MFRP and the secreted serine proteases PRSS56 and ADAMTS19 as part of a molecular network involved in ocular growth regulation.PLoS genetics · 2021Article
- Membrane-type frizzled-related protein regulates lipidome and transcription for photoreceptor function.FASEB journal : official publication of the Federation of American Societies for Experimental Biology · 2020Article
- Genetics of anophthalmia and microphthalmia. Part 1: Non-syndromic anophthalmia/microphthalmia.Human genetics · 2019Review
- Long-Term Effects of Gene Therapy in a Novel Mouse Model of HumanHuman gene therapy · 2019Article
- Regeneration of the zebrafish retinal pigment epithelium after widespread genetic ablation.PLoS genetics · 2019Article
- CRISPR GENOME SURGERY IN THE RETINA IN LIGHT OF OFF-TARGETING.Retina (Philadelphia, Pa.) · 2018Review
- Versatile Genome Engineering Techniques Advance Human Ocular Disease Researches in Zebrafish.Frontiers in cell and developmental biology · 2018Review
- Translation of CRISPR Genome Surgery to the Bedside for Retinal Diseases.Frontiers in cell and developmental biology · 2018Review
- Nanophthalmos: A Review of the Clinical Spectrum and Genetics.Journal of ophthalmology · 2018Review
- Molecular genetics and targeted therapy of WNT-related human diseases (Review).International journal of molecular medicine · 2017Article
- CRISPR-Cas Genome Surgery in Ophthalmology.Translational vision science & technology · 2017Review
Corrections and comments
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Authors and funding
5 authors at 1 institution in 1 country.
Funding
Abstract
Purpose: Mutations in membrane frizzled-related protein (MFRP) are associated with nanophthalmia, hyperopia, foveoschisis, irregular patches of RPE atrophy, and optic disc drusen in humans. Mouse mfrp mutants show retinal degeneration but no change in eye size or refractive state. The goal of this work was to generate zebrafish mutants to investigate the loss of Mfrp on eye size and refractive state, and to characterize other phenotypes observed. Methods: Clustered regularly interspaced short palindromic repeats (CRISPR)/Cas9 methods were used to generate multiple frameshift mutations in zebrafish mfrp causing premature translational stops in Mfrp. Spectral-domain optical coherence tomography (SD-OCT) was used to measure eye metrics and refractive state, and immunohistochemistry was used to study adult eyes. Gene expression levels were measured using quantitative PCR. Results: Zebrafish Mfrp was shown to localize to apical and basal regions of RPE cells, as well as the ciliary marginal zone. Loss of Mfrp in mutant zebrafish was verified histologically. Zebrafish eyes that were mfrp mutant showed reduced axial length causing hyperopia, RPE folding, and macrophages were observed subretinally. Visual acuity was reduced in mfrp mutant animals. Conclusions: Mutation of zebrafish mfrp results in hyperopia with subretinal macrophage infiltration, phenocopying aspects of human and mouse Mfrp deficiency. These mutant zebrafish will be useful in studying the onset and progression of Mfrp-related nanophthalmia, the cues that initiate the recruitment of macrophages, and the mechanisms of Mfrp function.
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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.