Evidence map›Paper›PMID 41776486›Full record

ArticleBMC ophthalmology2026

Bilateral idiopathic peripapillary choroidal neovascularization in a child with bruton disease: a case report.

Sarah Kerkouri, Jeanne Le Dinahet, Marianne Bourhis, Chloé Henry, Martin Castelle, Matthieu P Robert, Béatrice Cochener-Lamard

Abstract readCase Reports
In one paragraph

Article in BMC ophthalmology, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

0numbers the graph read from it
0cells of the map it votes in
0citing 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

0 citing papers in PubMed.

No citing paper in PubMed yet.

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

7 authors.

Sarah KerkouriUniversité de Bretagne Occidentale, Brest, Bretagne, France. sarah.kerkouri@chu-brest.fr.
Jeanne Le DinahetUniversité de Bretagne Occidentale, Brest, Bretagne, France.
Marianne BourhisService d'ophtalmologie, CHU Brest, Brest, F-29200, France.
Chloé HenryINSERM, UMR 1101, Brest, F-29 200, France.
Martin CastelleDepartment of Ophthalmology, Necker-Enfants Malades University Hospital, APHP, Paris, France.
Matthieu P RobertDepartment of Ophthalmology, Necker-Enfants Malades University Hospital, APHP, Paris, France.
Béatrice Cochener-LamardUniversité de Bretagne Occidentale, Brest, Bretagne, France.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

purposeTo describe a rare, unexplained case of bilateral peripapillary choroidal neovascularization (PPCNV) in a child with X-linked agammaglobulinemia (XLA, Bruton disease). OBSERVATION: A 13-year-old boy with XLA on regular intravenous immunoglobulin replacement presented with rapidly progressive bilateral vision loss. Best-corrected visual acuity (BCVA) was 20/100 OU. Fundus exam and multimodal imaging confirmed bilateral PPCNV with subretinal fluid. Extensive systemic, infectious, and immunologic investigations, including MRI, CT, cerebrospinal fluid, and broad serology panels, were unrevealing; interpretation of serologies was confounded by IVIG therapy. Intravitreal aflibercept led to initial anatomical and functional improvement, but macular atrophy subsequently developed, with limited visual recovery. No ocular inflammation or congenital optic disc anomaly was identified. CONCLUSION AND IMPORTANCE: Pediatric PPCNV is rare and usually secondary to inflammation or congenital anomalies. To our knowledge, this is the first report of PPCNV in XLA. Although the association appears coincidental, collaborative reporting of similar cases is needed to explore possible immunologic contributions and refine treatment strategies.

Indexed as

AgammaglobulinemiaChoroidal NeovascularizationGenetic Diseases, X-LinkedVision, LowAdolescentAngiogenesis InhibitorsAtrophyHumansIntravitreal InjectionsMacula LuteaMaleMultimodal ImagingReceptors, Vascular Endothelial Growth FactorRecombinant Fusion ProteinsVisual AcuityafliberceptAngiogenesis InhibitorsReceptors, Vascular Endothelial Growth FactorRecombinant Fusion ProteinsAfliberceptAgammaglobulinemiaBruton diseasePediatric choroidal neovascularizationPeripapillary CNV

Identifiers

PMID41776486
PMCPMC12961900

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.