Bilateral idiopathic peripapillary choroidal neovascularization in a child with bruton disease: a case report
To describe a rare, unexplained case of bilateral peripapillary choroidal neovascularization (PPCNV) in a child with X-linked agammaglobulinemia (XLA, Bruton disease). 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. 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.
Authors
- Chloé Henry
- Matthieu P. Robert (ORCID: https://orcid.org/0000-0003-3353-8117)
- J. Le Dinahet (ORCID: https://orcid.org/0000-0002-2783-3016)
- Sarah Kerkouri
- M. Bourhis
- Martin Castelle
- Béatrice Cochener-Lamard
Institutions
- Hôpital Necker-Enfants Malades (FR)
- Centre National de la Recherche Scientifique (FR)
- Inserm (FR)
- Université de Bretagne Occidentale (FR)
- Université Paris Cité (FR)
- Université Paris-Saclay (FR)
- Institut Necker Enfants Malades (FR)
- Assistance Publique – Hôpitaux de Paris (FR)
- Centre Hospitalier de Cornouaille (FR)
- Centre Hospitalier Régional Universitaire de Brest (FR)
- Laboratoire de Traitement de l'Information Médicale (FR)
Publication Details
- Journal
- BMC Ophthalmology
- Published
- 2026-03-03
- DOI
- https://doi.org/10.1186/s12886-026-04691-2
- Primary Topic
- Retinal Diseases and Treatments
- Type
- article
- Field-Weighted Citation Impact
- 0.00