ArticleGraefe's archive for clinical and experimental ophthalmology = Albrecht von Graefes Archiv fur klinische und experimentelle Ophthalmologie1995
Retinitis pigmentosa and the question of photoreceptor connecting cilium defects.
Article in Graefe's archive for clinical and experimental ophthalmology = Albrecht von Graefes Archiv fur klinische und experimentelle Ophthalmologie, 1995. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.
What it found
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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.
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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.
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Who cites it
3 citing papers in PubMed, 6 citations in OpenAlex.
- Ciliary transition zone (TZ) proteins RPGR and CEP290: role in photoreceptor cilia and degenerative diseases.Expert opinion on therapeutic targets · 2012Review
- RPGR-ORF15, which is mutated in retinitis pigmentosa, associates with SMC1, SMC3, and microtubule transport proteins.The Journal of biological chemistry · 2005Article
- Cilia-related diseases.The Journal of pathology · 2004Review
Corrections and comments
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Authors and funding
1 author at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
backgroundA generalized structural defect of the cilia in various tissues, including photoreceptor connecting cilium, has been postulated as occurring in some forms of retinitis pigmentosa (RP). However, the literature on ciliary abnormalities in RP contains contradictory findings.
methodsIn this study the fine structure of photoreceptors from 17 RP donors including X-linked RP, X-linked RP carrier state, autosomal dominant RP and autosomal recessive RP was examined by electron microscopy.
resultsPhotoreceptor preservation was commonly observed even in the most advanced cases of the disease, especially in the perimacular area, in the proximity of the optic nerve and in the periphery. Primary ciliary defects, expressed as additional or missing microtubules, were found in none of the samples. Comparison of photoreceptors in normal and RP retinae showed thinner cilia in RP cells but no defect in the microtubule arrangements within the connecting cilium.
conclusionAdditional or missing microtubules in ciliated cells are not uncommon and have been reported in the literature and recorded in some studies of RP tissue. Such defects, however, are believed to be acquired rather than inherited abnormalities of cilia and were not observed in the photoreceptor connecting cilia of RP patients examined in this study. Thinning of the cilium may also be a secondary effect related to cell shrinkage early during apoptosis, which is postulated to be a common pathway in photoreceptor degeneration.
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Registered trials
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