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Purtscher-like retinopathy in a 19-year-old with maturity-onset diabetes of the young: a case report

BACKGROUND: We report the first case of Purtscher-like retinopathy in a patient with 17q12 deletion-associated maturity-onset diabetes of the young. CASE PRESENTATION: A 19-year-old diabetic Hispanic male with history of cataracts and toe amputations presented with sudden onset of painless bilateral...

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Autores principales: Oh, Angela J., Javaheri, Michael, Hosseini, Hamid, Prasad, Pradeep S.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10278244/
https://www.ncbi.nlm.nih.gov/pubmed/37331978
http://dx.doi.org/10.1186/s13256-023-03985-z
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author Oh, Angela J.
Javaheri, Michael
Hosseini, Hamid
Prasad, Pradeep S.
author_facet Oh, Angela J.
Javaheri, Michael
Hosseini, Hamid
Prasad, Pradeep S.
author_sort Oh, Angela J.
collection PubMed
description BACKGROUND: We report the first case of Purtscher-like retinopathy in a patient with 17q12 deletion-associated maturity-onset diabetes of the young. CASE PRESENTATION: A 19-year-old diabetic Hispanic male with history of cataracts and toe amputations presented with sudden onset of painless bilateral vision loss for 1 week with no associated trauma. Visual acuity was counting fingers at six feet in both eyes. Dilated retinal examination revealed bilateral peripapillary cotton wool spots and intraretinal hemorrhages, and significant subretinal and intraretinal fluid on optical coherence tomography. Fluorescein angiography revealed arteriolar staining and leakage around the disc with areas of capillary nonperfusion, supporting the diagnosis of Purtscher-like retinopathy. Systemic workup revealed multiple diabetic complications including chronic osteomyelitis of multiple toes, nonhealing diabetic foot ulcers, neurogenic bladder and bowel, and bilateral lower-extremity muscular neuropathies. Genetic evaluation revealed a 17q12 deletion, which is associated with maturity-onset diabetes of the young 5. On follow-up examination, he received a single intravitreal antivascular endothelial growth factor injection in the left eye (off label) for persistent macular edema. Although his retinal edema improved, his visual acuity remained poor. CONCLUSIONS: The presentation of our patient’s multiple diabetic complications along visual symptoms suggests Purtscher-like retinopathy can be a sequela of uncontrolled diabetes. Purtscher-like retinopathy is a rare but possible consideration in diabetic patients who present with acute-onset vision loss.
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spelling pubmed-102782442023-06-20 Purtscher-like retinopathy in a 19-year-old with maturity-onset diabetes of the young: a case report Oh, Angela J. Javaheri, Michael Hosseini, Hamid Prasad, Pradeep S. J Med Case Rep Case Report BACKGROUND: We report the first case of Purtscher-like retinopathy in a patient with 17q12 deletion-associated maturity-onset diabetes of the young. CASE PRESENTATION: A 19-year-old diabetic Hispanic male with history of cataracts and toe amputations presented with sudden onset of painless bilateral vision loss for 1 week with no associated trauma. Visual acuity was counting fingers at six feet in both eyes. Dilated retinal examination revealed bilateral peripapillary cotton wool spots and intraretinal hemorrhages, and significant subretinal and intraretinal fluid on optical coherence tomography. Fluorescein angiography revealed arteriolar staining and leakage around the disc with areas of capillary nonperfusion, supporting the diagnosis of Purtscher-like retinopathy. Systemic workup revealed multiple diabetic complications including chronic osteomyelitis of multiple toes, nonhealing diabetic foot ulcers, neurogenic bladder and bowel, and bilateral lower-extremity muscular neuropathies. Genetic evaluation revealed a 17q12 deletion, which is associated with maturity-onset diabetes of the young 5. On follow-up examination, he received a single intravitreal antivascular endothelial growth factor injection in the left eye (off label) for persistent macular edema. Although his retinal edema improved, his visual acuity remained poor. CONCLUSIONS: The presentation of our patient’s multiple diabetic complications along visual symptoms suggests Purtscher-like retinopathy can be a sequela of uncontrolled diabetes. Purtscher-like retinopathy is a rare but possible consideration in diabetic patients who present with acute-onset vision loss. BioMed Central 2023-06-19 /pmc/articles/PMC10278244/ /pubmed/37331978 http://dx.doi.org/10.1186/s13256-023-03985-z Text en © The Author(s) 2023 https://creativecommons.org/licenses/by/4.0/Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) . The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/ (https://creativecommons.org/publicdomain/zero/1.0/) ) applies to the data made available in this article, unless otherwise stated in a credit line to the data.
spellingShingle Case Report
Oh, Angela J.
Javaheri, Michael
Hosseini, Hamid
Prasad, Pradeep S.
Purtscher-like retinopathy in a 19-year-old with maturity-onset diabetes of the young: a case report
title Purtscher-like retinopathy in a 19-year-old with maturity-onset diabetes of the young: a case report
title_full Purtscher-like retinopathy in a 19-year-old with maturity-onset diabetes of the young: a case report
title_fullStr Purtscher-like retinopathy in a 19-year-old with maturity-onset diabetes of the young: a case report
title_full_unstemmed Purtscher-like retinopathy in a 19-year-old with maturity-onset diabetes of the young: a case report
title_short Purtscher-like retinopathy in a 19-year-old with maturity-onset diabetes of the young: a case report
title_sort purtscher-like retinopathy in a 19-year-old with maturity-onset diabetes of the young: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10278244/
https://www.ncbi.nlm.nih.gov/pubmed/37331978
http://dx.doi.org/10.1186/s13256-023-03985-z
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