Evidence map›Paper›PMID 40986557›Full record

ArticlePloS one2025

Identification of coexisting Mfrprd6 and Pde6brd10 mutations causing spontaneous retinal detachment in commercially available rd6 mice.

Asaka Lee Shiozawa, Maika Hosoi Kobayashi, Yusuke Shiozawa, Yasuhiro Ikeda, Yoshitaka Miyagawa, Fumiki Okamoto, Mashito Sakai, Takashi Okada, Tsutomu Igarashi

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Article in PloS one, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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1 · What the graph read from it

What it found

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2 · The registry

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3 · Its place in the literature

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4 · The record

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5 · Who and what money

Authors and funding

9 authors.

Asaka Lee ShiozawaDepartment of Ophthalmology, Nippon Medical School, Sendagi, Bunkyo-ku, Tokyo, Japan.
Maika Hosoi KobayashiDepartment of Ophthalmology, Nippon Medical School, Sendagi, Bunkyo-ku, Tokyo, Japan.
Yusuke ShiozawaLaboratory of Molecular Analysis, Nippon Medical School, Sendagi, Bunkyo-ku, Tokyo, Japan.ORCID https://orcid.org/0000-0001-8673-677X
Yasuhiro IkedaFaculty of Medicine, University of Miyazaki, Kihara, Kiyotake, Miyazaki City, Miyazaki, Japan.
Yoshitaka MiyagawaDepartment of Biochemistry and Molecular Biology, Nippon Medical School, Sendagi, Bunkyo-ku, Tokyo, Japan.
Fumiki OkamotoDepartment of Ophthalmology, Nippon Medical School, Sendagi, Bunkyo-ku, Tokyo, Japan.
Mashito SakaiDepartment of Biochemistry and Molecular Biology, Nippon Medical School, Sendagi, Bunkyo-ku, Tokyo, Japan.
Takashi OkadaDivision of Molecular and Medical Genetics, Center for Gene and Cell Therapy, Institute of Medical Science, University of Tokyo, Shirokanedai, Minato-ku, Tokyo, Japan.ORCID https://orcid.org/0000-0002-2910-3797
Tsutomu IgarashiDepartment of Biochemistry and Molecular Biology, Nippon Medical School, Sendagi, Bunkyo-ku, Tokyo, Japan.ORCID https://orcid.org/0000-0002-7467-6746

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

purposeThe rd6 mouse model, characterized by retinal degeneration due to an Mfrp mutation, has been widely studied. However, we identified a subset of rd6 mice that developed severe non-rhegmatogenous retinal detachment (rd6-RD), suggesting the presence of additional genetic factors. This study aimed to characterize the retinal phenotype of rd6-RD mice and identify potential causative genetic mutations.

methodsWe performed optical coherence tomography, fundus imaging, electroretinography, and histological analysis to compare retinal structures and functions between rd6, rd6-RD, and C57BL/6J mice. Whole-genome sequencing was conducted to identify potential mutations associated with the retinal detachment phenotype.

resultsOptical coherence tomography revealed retinal detachment in rd6-RD mice as early as 4 weeks old, with complete loss of the outer nuclear layer by 6 weeks. Fundus examination at 11 weeks showed pale fundi and narrowed, whitened retinal vessels in rd6-RD mice, distinct from rd6 mice. On electroretinography, rd6-RD mice displayed significantly diminished a- and b-wave amplitudes, with no detectable responses by 10 weeks. Histological analysis confirmed severe outer retinal degeneration and disappearance of the outer layers in rd6-RD mice. Whole-genome sequencing identified a missense R560C mutation in Pde6b, corresponding to the Pde6brd10 mutation, in rd6-RD mice.

conclusionsA subset of rd6 mice exhibited severe retinal detachment and outer retinal degeneration, distinct from the previously characterized Mfrp-related phenotype. The identification of the Pde6brd10 mutation suggests that these mice possess a dual-mutant genotype (Mfrprd6 and Pde6brd10), exacerbating retinal degeneration. These findings highlight the importance of genetic verification in commercially available mouse models and provide new insights into the genetic complexity of inherited retinal degenerations.

Indexed as

Cyclic Nucleotide Phosphodiesterases, Type 6Membrane ProteinsMutationRetinal DetachmentAnimalsDisease Models, AnimalElectroretinographyMiceMice, Inbred C57BLPhenotypeRetinaRetinal DegenerationTomography, Optical CoherenceWhole Genome SequencingCyclic Nucleotide Phosphodiesterases, Type 6Membrane Proteins

Identifiers

PMID40986557
PMCPMC12456819

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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 Socratic graph.