ArticleInvestigative ophthalmology & visual science2012
Meckelin is necessary for photoreceptor intraciliary transport and outer segment morphogenesis.
Article in Investigative ophthalmology & visual science, 2012. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 25 papers.
What it found
Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.
The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.
Who cites it
25 citing papers in PubMed, 37 citations in OpenAlex.
- Cis-regulatory evolution reveals sensory trade-offs as a genetic basis for temporal niche evolution in tapirs.Science advances · 2026Article
- Glucose-Regulated Protein 78, via Releasing β-Catenin from Adherens Junctions, Facilitates Its Interaction with STAT3 in Mediating Retinal Neovascularization.The American journal of pathology · 2024Article
- The endoplasmic reticulum membrane protein complex subunitGenes & diseases · 2024Article
- Requirement of Site-Specific Tyrosine Phosphorylation of Cortactin in Retinal Neovascularization and Vascular Leakage.Arteriosclerosis, thrombosis, and vascular biology · 2024Article
- Systematic characterization of gene function in the photosynthetic alga Chlamydomonas reinhardtii.Nature genetics · 2022Article
- Tmem138 is localized to the connecting cilium essential for rhodopsin localization and outer segment biogenesis.Proceedings of the National Academy of Sciences of the United States of America · 2022Article
- On the Wrong Track: Alterations of Ciliary Transport in Inherited Retinal Dystrophies.Frontiers in cell and developmental biology · 2021Review
- Functional analysis of new human Bardet-Biedl syndrome loci specific variants in the zebrafish model.Scientific reports · 2019Article
- Clinical characteristics of individual organ system disease in non-motile ciliopathies.Translational science of rare diseases · 2019Article
- Cell type-specific regulation of ciliary transition zone assembly in vertebrates.The EMBO journal · 2018Article
- Rescue of cone function in cone-onlyMolecular vision · 2018Article
- Photoreceptor outer segment as a sink for membrane proteins: hypothesis and implications in retinal ciliopathies.Human molecular genetics · 2017Review
- The Biology of Ciliary Dynamics.Cold Spring Harbor perspectives in biology · 2017Review
- Article
- Ciliopathy-associated IQCB1/NPHP5 protein is required for mouse photoreceptor outer segment formation.FASEB journal : official publication of the Federation of American Societies for Experimental Biology · 2016Article
- Aberrant protein trafficking in retinal degenerations: The initial phase of retinal remodeling.Experimental eye research · 2016Review
- Nephronophthisis and related syndromes.Current opinion in pediatrics · 2015Review
- Ciliopathies: the trafficking connection.Traffic (Copenhagen, Denmark) · 2014Review
- Article
- The role of primary cilia in the development and disease of the retina.Organogenesis · 2014Review
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
6 authors at 1 institution in 1 country.
Funding
Abstract
purposeCilia, complex structures found ubiquitously in most vertebrate cells, serve a variety of functions ranging from cell and fluid movement, cell signaling, tissue homeostasis, to sensory perception. Meckelin is a component of ciliary and cell membranes and is encoded by Tmem67 (Mks3). In this study, the retinal morphology and ciliary function in a mouse model for Meckel Syndrome Type 3 (MKS3) throughout the course of photoreceptor development was examined.
methodsTo study the effects of a disruption in the Mks3 gene on the retina, the authors introduced a functional allele of Pde6b into B6C3Fe a/a-bpck/J mice and evaluated their retinas by ophthalmoscopic, histologic, and ultrastructural examination. In addition, immunofluorescence microscopy was used to assess protein trafficking through the connecting cilium and to examine the localization of ciliary and synaptic proteins in Tmem67(bpck) mice and controls.
resultsPhotoreceptors degenerate early and rapidly in bpck/bpck mutant mice. In addition, phototransduction proteins, such as rhodopsin, arrestin, and transducin, are mislocalized. Ultrastructural examination of photoreceptors reveal morphologically intact connecting cilia but dysmorphic and misoriented outer segment (OS) discs, at the earliest time point examined.
conclusionsThese findings underscore the important role for meckelin in intraciliary transport of phototransduction molecules and their effects on subsequent OS morphogenesis and maintenance.
Indexed as
Identifiers
What OpenQuestion holds
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.