Evidence map›Paper›PMID 30513534›Full record

ArticleInvestigative ophthalmology & visual science2018

Longitudinal Assessment of Retinal Structure in Achromatopsia Patients With Long-Term Follow-up.

Nashila Hirji, Michalis Georgiou, Angelos Kalitzeos, James W Bainbridge, Neruban Kumaran, Jonathan Aboshiha, Joseph Carroll, Michel Michaelides

Open access · goldAbstract read
In one paragraph

Article in Investigative ophthalmology & visual science, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 33 papers.

0numbers the graph read from it
0cells of the map it votes in
33citing papers in PubMed
5.8field-weighted citation impact, top 3% of its field
1 · What the graph read from it

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.

2 · The registry

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.

3 · Its place in the literature

Who cites it

33 citing papers in PubMed, 51 citations in OpenAlex.

  1. Article
  2. Article
  3. Article
  4. Article
  5. Article
  6. Gene Therapy for Achromatopsia.International journal of molecular sciences · 2024
    Review
  7. Article
  8. Article
  9. Review
  10. A homozygous structural variant ofGenetics in medicine open · 2024
    Article
  11. Article
  12. Article
  13. Article
  14. Article
  15. Article
  16. Disease Progression inCurrent issues in molecular biology · 2021
    Article
  17. Review
  18. Article
  19. Article
  20. Observational
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

8 authors at 3 institutions in 2 countries.

Nashila HirjiUCL Institute of Ophthalmology, University College London, London, United Kingdom.
Michalis GeorgiouUCL Institute of Ophthalmology, University College London, London, United Kingdom.
Angelos KalitzeosUCL Institute of Ophthalmology, University College London, London, United Kingdom.
James W BainbridgeUCL Institute of Ophthalmology, University College London, London, United Kingdom.
Neruban KumaranUCL Institute of Ophthalmology, University College London, London, United Kingdom.
Jonathan AboshihaUCL Institute of Ophthalmology, University College London, London, United Kingdom.
Joseph CarrollDepartment of Ophthalmology and Visual Sciences, Medical College of Wisconsin, Milwaukee, Wisconsin, United States.
Michel MichaelidesUCL Institute of Ophthalmology, University College London, London, United Kingdom.
University College London · GBMoorfields Eye Hospital · GBMedical College of Wisconsin · US

Funding

MORPHOLOGY MODULEP30EY001931 · NEI · MEDICAL COLLEGE OF WISCONSIN · PI LINK, BRIAN A · 1985 to 2016
$9.8M
Assessing Photoreceptor Structure and Function in Normal and Diseased RetinaeR01EY017607 · NEI · MEDICAL COLLEGE OF WISCONSIN · PI Joseph Carroll · 2008 to 2026
$8.3M
Department of Health NIHR-RP-011-003Medical Research CouncilNEI NIH HHS P30 EY001931NEI NIH HHS R01 EY017607Wellcome TrustWellcome Trust 099173/Z/12/Z
6 · The paper itself

Abstract

Purpose: To longitudinally characterize structural retinal changes in achromatopsia (ACHM) over extended follow-up. Methods: Fifty molecularly confirmed ACHM subjects underwent serial spectral-domain optical coherence tomography (SD-OCT) and fundus autofluorescence (FAF) imaging. Foveal structure on SD-OCT was graded and compared for evidence of progression, and foveal total retinal thickness (FTRT) and outer nuclear layer (ONL) thickness were serially measured. FAF patterns were characterized and compared over time. Results: Mean SD-OCT follow-up was 61.6 months (age range at baseline, 6-52 years). Forty-five of the subjects had serial FAF (mean follow-up: 48.5 months). Only 6 (12%) of the subjects demonstrated qualitative change on serial foveal SD-OCT scans. Among the entire cohort, there was no statistically significant change over time in FTRT (P = 0.2459) or hyporeflective zone (HRZ) diameter (P = 0.3737). There was a small-but statistically significant-increase in ONL thickness (P = 0.0084). Three different FAF patterns were observed: centrally increased FAF (13/45), normal FAF (14/45), and well-demarcated reduced FAF (18/45), with the latter group displaying a small gradual increase in the area of reduced FAF of 0.055 mm2 over 43.4 months (P = 0.0011). Conclusions: This longitudinal study of retinal structure in ACHM represents the largest cohort and longest follow-up period to date. Our findings support the presiding notion that ACHM is essentially a stationary condition regarding retinal structure, and any change over time is likely to be small, slow, and variable across patients. This may potentially afford a wider window for therapeutic intervention.

Indexed as

AdolescentAdultChildColor Vision DefectsFemaleFluorescein AngiographyFollow-Up StudiesFovea CentralisGenetic Association StudiesHumansLongitudinal StudiesMaleMiddle AgedProspective StudiesRetinaTomography, Optical Coherence

Identifiers

PMID30513534
PMCPMC6280917
OpenAlexW2903153265

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC-ND
Read underepoch 390

Registered trials

None linked

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.