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Wagner syndrome is a rare autosomal dominant vitreoretinopathy caused by mutations in chondroitin sulphate proteoglycan 2 (CSPG2)/Versican (VCAN). Here, we present a retrospective case series of a family pedigree with genetically confirmed Wagner syndrome (heterozygous VCAN exon 8 deletion), as follows: a 34-year-old mother (P1), 12-year-old daughter (P2), and a 2-year-old son (P3). The phenotype included early-onset cataract (P1), optically empty vitreous with avascular membranes (P1, 2), nasal dragging of optic nerve heads associated with foveal hypoplasia (P1, 2), tractional retinoschisis on optical coherence tomography (P2), and peripheral circumferential vitreo-retinal interface abnormality resembling white-without-pressure (P3) progressing to pigmented chorio-retinal atrophy (P1, 2). P2 developed a macula-off retinal detachment, which was treated initially with encircling band + vitrectomy + gas, followed by vitrectomy + heavy silicone oil tamponade for re-detachment from new inferior breaks. Strong vitreo-retinal adhesion was noted intraoperatively, which prevented the separation of posterior hyaloid beyond the equator. Electroretinograms from P1&2 demonstrated attenuated b-waves, a-waves, and flicker responses in light- and dark-adapted conditions, suggestive of generalised retinal dysfunction. Our patients demonstrated the clinical spectrum of Wagner syndrome, highlighting nasal dragging with foveal disruption as a distinguishing feature from other inherited vitreoretinopathies. Surgical outcomes demonstrate significant challenges in managing vitreo-retinal traction and need for further research into strategies to prevent sight loss.
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http://dx.doi.org/10.3390/genes15091178 | DOI Listing |
Am J Ophthalmol Case Rep
September 2025
Department of Ophthalmology, Bascom Palmer Eye Institute, Miller School of Medicine, University of Miami, 900 NW 17th Avenue, Miami, FL, 33136, USA.
Purpose: To describe the clinical features of a patient with Wagner syndrome and a history of retinopathy of prematurity (ROP).
Observations: A 23-year-old Hispanic male was referred for retina evaluation. The patient had a history of regressed ROP in both eyes that was never treated.
Hum Genomics
April 2025
Eye Institute, Department of Ophthalmology, Eye & ENT Hospital, Fudan University, Shanghai, 200031, China.
Purpose: This study aimed to elucidate the reliability of clinical impressions based on ocular manifestations in patients suspected of heritable connective tissue disorders (HCTDs) compared to the final genetic diagnosis. Furthermore, it sought to determine the optimal diagnostic strategy for patients with HCTDs through pathogenicity analysis.
Methods: Clinical characteristics of 58 patients suspected of HCTDs were analyzed to establish provisional clinical diagnoses.
Ophthalmic Genet
August 2025
Scheie Eye Institute, Department of Ophthalmology, Perelman School of Medicine, University of Pennsylvania, Philadelphia, Pennsylvania, USA.
Purpose: To understand the retina micropathology in a family with a novel variant in .
Methods: Two sisters ages 16 (proband) and 18 years old and their 48-year-old father underwent comprehensive ophthalmic evaluations. Multimodal imaging was performed with spectral domain optical coherence tomography, ultrawide field short-wavelength fundus autofluorescence, and pseudocolor imaging.
Clin Exp Ophthalmol
April 2025
Cole Eye Institute, Cleveland Clinic, Cleveland, Ohio, USA.
Hereditary vitreoretinopathies (HVRs), also known as hereditary vitreoretinal degenerations comprise a heterogeneous group of inherited disorders of the retina and vitreous, collectively and variably characterised by vitreal abnormalities, such as fibrillary condensations, liquefaction or membranes, as well as peripheral retinal abnormalities, vascular changes in some, an increased risk of retinal detachment and early-onset cataract formation. The pathology often involves the vitreoretinal interface in some, while the major underlying abnormality is vascular in others. Recent advances in molecular diagnosis and identification of the responsible genes and have improved our understanding of the pathogenesis, risks and management of the HVRs.
View Article and Find Full Text PDFRetina
May 2025
Department of Ophthalmology, Rush University Medical Center, and Illinois Retina Associates, Chicago, Illinois.