Fundus albipunctatus-like retinopathy in hypovitaminosis A-related optic neuropathy with skull hyperostosis
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Updated: Sep 26, 2026

Application of Optical Coherence Tomography to a Mouse Model of Retinopathy
Published on: January 12, 2022
Adam Tomasz Chmiel1, Maria Franca2,3, Ana Luísa Carvalho3,4,5
1Department of Ophthalmology, National Medical Institute of the Ministry of the Interior and Administration, Warsaw, Poland.
Abstract:
A young man presented with progressive night blindness, optic neuropathy and bilateral retinal flecks, initially suggestive of an inherited retinal dystrophy. Genetic testing identified a heterozygous pathogenic variant in USH2A alongside multiple variants of uncertain significance, none of which fully explained the phenotype. Further investigation revealed severe vitamin A deficiency and cranial hyperostosis with narrowing of the optic canals. Retinal findings improved following vitamin A supplementation. This case illustrates the diagnostic complexity of retinal dystrophies, emphasises the need for a multidisciplinary approach by integrating findings from various specialties, including ophthalmology, neurology, medical genetics and neurosurgery, even in patients with genetic findings of potential relevance. Moreover, it highlights vitamin A deficiency as a reversible cause of retinal pathology.
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