Evidence map›Paper›PMID 40781274›Full record

ArticleBMC immunology2025

Immune landscape in children with X-linked retinoschisis.

Ying Hsu, Giulia Del Valle, Sarah Stanley, Brianna Lobeck, Sergei I Syrbu, Christine Sinkey, Christopher R Fortenbach, Alina Dumitrescu, Arlene V Drack

Abstract read
In one paragraph

Article in BMC immunology, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
–field-weighted citation impact
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

1 citing paper in PubMed.

  1. Article
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

9 authors.

Ying HsuDepartment of Ophthalmology and Visual Sciences, University of Iowa, 200 Hawkins Drive, Iowa City, IA, 52242, USA.
Giulia Del ValleDepartment of Ophthalmology and Visual Sciences, University of Iowa, 200 Hawkins Drive, Iowa City, IA, 52242, USA.
Sarah StanleyDepartment of Ophthalmology and Visual Sciences, University of Iowa, 200 Hawkins Drive, Iowa City, IA, 52242, USA.
Brianna LobeckDepartment of Ophthalmology and Visual Sciences, University of Iowa, 200 Hawkins Drive, Iowa City, IA, 52242, USA.
Sergei I SyrbuDepartment of Pathology, University of Iowa Hospitals and Clinics, 200 Hawkins Drive, Iowa City, IA, 52242, USA.
Christine SinkeyDepartment of Ophthalmology and Visual Sciences, University of Iowa, 200 Hawkins Drive, Iowa City, IA, 52242, USA.
Christopher R FortenbachDepartment of Ophthalmology, University of Washington, Seattle, WA, USA.
Alina DumitrescuDepartment of Ophthalmology and Visual Sciences, University of Iowa, 200 Hawkins Drive, Iowa City, IA, 52242, USA.
Arlene V DrackDepartment of Ophthalmology and Visual Sciences, University of Iowa, 200 Hawkins Drive, Iowa City, IA, 52242, USA. arlene-drack@uiowa.edu.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundX-linked retinoschisis is a retinovitreal disorder primarily affecting males, starting in childhood. Over time, patients experience deterioration of vision due to the lack of retinoschisin-1 function. In clinical trials performing intravitreal gene delivery in those affected by this disorder, ocular inflammation was observed, which may have masked efficacy. A subsequent study focusing on analyzing the populations of peripheral blood mononuclear cells and cytokines in adults with this disease reported aberrant dendritic cell numbers and cytokine levels in peripheral blood, indicating that adults with this disease may have an altered baseline immunity. Whether the aberrant peripheral immunity in affected adults was a consequence of advanced eye pathology remained unclear. This study focuses on analyzing the populations of blood lymphocyte subsets in children aged 0 to 7 years with X-linked retinoschisis and age-matched controls using flow cytometry.

resultsThe fractions of lymphocyte subsets that were CD16a+/CD56+, namely natural killer cells, were significantly lower in blood samples from children with X-linked retinoschisis. In children with X-linked retinoschisis, the fractions of CD3+/CD4 + T cells were higher, and the fractions of CD3 + CD8 + T cells were lower, despite having the same amounts of total CD3 + T cells within their lymphocyte populations. This resulted in a significantly greater CD4/CD8 ratio in children with X-linked retinoschisis compared to age-matched controls.

conclusionsAlterations were found in blood lymphocyte compositions of children with X-linked retinoschisis within both innate and adaptive immune axes. Some alterations including an elevation of CD4/CD8 ratio in X-linked retinoschisis mirror those previously found in adult patients with this disease. The fact that these abnormalities were present early in this disease indicates that retinoschisin-1 may play a role in regulating immunity in addition to retinal structure. The findings may have implications for future treatments such as ocular gene delivery.

Indexed as

Killer Cells, NaturalLymphocyte SubsetsRetinoschisisChildChild, PreschoolEye ProteinsFemaleHumansInfantInfant, NewbornMaleEye ProteinsCD4/CD8 ratioFlow cytometryRetinoschisinX-linked retinoschisis

Identifiers

PMID40781274
PMCPMC12333184

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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 OpenQuestion graph.