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Bilateral Complete Loss and Partial Regeneration of Photoreceptor Layers in a Pediatric Vogt-Koyanagi-Harada (VKH) Case
Vogt-Koyanagi-Harada (VKH) disease is a multisystem autoimmune disease affecting melanocyte-containing tissues in the eyes, meninges, ear, and skin. As far as we are aware, this is a unique case report documenting the regeneration of the photoreceptor layer after bilateral complete loss of the photo...
Autores principales: | , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
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Cureus
2022
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9823270/ https://www.ncbi.nlm.nih.gov/pubmed/36628028 http://dx.doi.org/10.7759/cureus.32273 |
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author | Aldossari, Shaikha Alhussaini, Nawaf A Barri, Abdulrahman Al Dhibi, Hassan |
author_facet | Aldossari, Shaikha Alhussaini, Nawaf A Barri, Abdulrahman Al Dhibi, Hassan |
author_sort | Aldossari, Shaikha |
collection | PubMed |
description | Vogt-Koyanagi-Harada (VKH) disease is a multisystem autoimmune disease affecting melanocyte-containing tissues in the eyes, meninges, ear, and skin. As far as we are aware, this is a unique case report documenting the regeneration of the photoreceptor layer after bilateral complete loss of the photoreceptor layer in a child with VKH. We report a case of a 12-year-old male with no significant past medical history who presented during the chronic stage of incomplete VKH. He was found to have a complete loss of photoreceptor layer in both eyes during a work-up to confirm the aforementioned disease. Upon receiving his first pulse dose of 500 mg IV methylprednisolone as a treatment course, he developed severe steroid-induced hyperglycemia (random blood glucose of 17.6 mmol/L). Additionally, a brain MRI revealed pituitary gland changes compatible with diabetes insipidus, which is a combination that was mentioned once in the literature before. A review of the systems did not suggest any other systemic diseases. The patient’s elevated blood sugar level was managed by a pediatrician, and it normalized. At his last visit, optical coherence tomography (OCT) showed hypertrophy/regeneration of the photoreceptor layer. This case report indicates that retinal photoreceptor layer regeneration can be sometimes observed with follow-up after the resolution of inflammation and uveitis. |
format | Online Article Text |
id | pubmed-9823270 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | Cureus |
record_format | MEDLINE/PubMed |
spelling | pubmed-98232702023-01-09 Bilateral Complete Loss and Partial Regeneration of Photoreceptor Layers in a Pediatric Vogt-Koyanagi-Harada (VKH) Case Aldossari, Shaikha Alhussaini, Nawaf A Barri, Abdulrahman Al Dhibi, Hassan Cureus Ophthalmology Vogt-Koyanagi-Harada (VKH) disease is a multisystem autoimmune disease affecting melanocyte-containing tissues in the eyes, meninges, ear, and skin. As far as we are aware, this is a unique case report documenting the regeneration of the photoreceptor layer after bilateral complete loss of the photoreceptor layer in a child with VKH. We report a case of a 12-year-old male with no significant past medical history who presented during the chronic stage of incomplete VKH. He was found to have a complete loss of photoreceptor layer in both eyes during a work-up to confirm the aforementioned disease. Upon receiving his first pulse dose of 500 mg IV methylprednisolone as a treatment course, he developed severe steroid-induced hyperglycemia (random blood glucose of 17.6 mmol/L). Additionally, a brain MRI revealed pituitary gland changes compatible with diabetes insipidus, which is a combination that was mentioned once in the literature before. A review of the systems did not suggest any other systemic diseases. The patient’s elevated blood sugar level was managed by a pediatrician, and it normalized. At his last visit, optical coherence tomography (OCT) showed hypertrophy/regeneration of the photoreceptor layer. This case report indicates that retinal photoreceptor layer regeneration can be sometimes observed with follow-up after the resolution of inflammation and uveitis. Cureus 2022-12-06 /pmc/articles/PMC9823270/ /pubmed/36628028 http://dx.doi.org/10.7759/cureus.32273 Text en Copyright © 2022, Aldossari et al. https://creativecommons.org/licenses/by/3.0/This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. |
spellingShingle | Ophthalmology Aldossari, Shaikha Alhussaini, Nawaf A Barri, Abdulrahman Al Dhibi, Hassan Bilateral Complete Loss and Partial Regeneration of Photoreceptor Layers in a Pediatric Vogt-Koyanagi-Harada (VKH) Case |
title | Bilateral Complete Loss and Partial Regeneration of Photoreceptor Layers in a Pediatric Vogt-Koyanagi-Harada (VKH) Case |
title_full | Bilateral Complete Loss and Partial Regeneration of Photoreceptor Layers in a Pediatric Vogt-Koyanagi-Harada (VKH) Case |
title_fullStr | Bilateral Complete Loss and Partial Regeneration of Photoreceptor Layers in a Pediatric Vogt-Koyanagi-Harada (VKH) Case |
title_full_unstemmed | Bilateral Complete Loss and Partial Regeneration of Photoreceptor Layers in a Pediatric Vogt-Koyanagi-Harada (VKH) Case |
title_short | Bilateral Complete Loss and Partial Regeneration of Photoreceptor Layers in a Pediatric Vogt-Koyanagi-Harada (VKH) Case |
title_sort | bilateral complete loss and partial regeneration of photoreceptor layers in a pediatric vogt-koyanagi-harada (vkh) case |
topic | Ophthalmology |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9823270/ https://www.ncbi.nlm.nih.gov/pubmed/36628028 http://dx.doi.org/10.7759/cureus.32273 |
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