Abstract
Background
Leprechaunism is a rare congenital syndrome and the most severe form of insulin resistance syndrome, with mutations in the insulin receptor gene. Recombinant human insulin-like growth factor-1 (rhIGF-1) is currently applicable to the treatment for insulin resistance syndrome by its insulin-like effect. Although IGF-1 is thought to promote tissue proliferation and neovascularization, it is uncertain how it acts on the development of diabetic retinopathy.
Methods
Interventional case report.
Results
A 12-year-old girl with leprechaunism has been treated with IGF-1 since she was 6 months old. She presented with neovascular glaucoma in the left eye, but with no serious changes in the right fundus except for tortuosity and dilatation of retinal veins. Thereafter, retinal neovascularization in the right eye developed in 6 months to form a loop-shaped vascular network in the vitreous cavity despite panretinal photocoagulation.
Conclusions
Characteristics of retinal neovascularization and clinical course suggest that IGF-1 treatment was closely associated with the development of diabetic retinopathy in this case.
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Kitamei, H., Yokoi, M., Kase, M. et al. Retinal neovascularization during treatment with IGF-1 for insulin resistance syndrome. Graefe's Arch Clin Exp Ophthalmol 243, 715–717 (2005). https://doi.org/10.1007/s00417-004-1093-6
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DOI: https://doi.org/10.1007/s00417-004-1093-6