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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...

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Autores principales: Aldossari, Shaikha, Alhussaini, Nawaf A, Barri, Abdulrahman, Al Dhibi, Hassan
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Cureus 2022
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.
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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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