Cargando…

A case of probable Vogt–Koyanagi–Harada disease in a 3-year-old girl

BACKGROUND: Vogt–Koyanagi–Harada (VKH) disease is a T-cell-mediated autoimmune disorder characterized by bilateral granulomatous panuveitis with various systemic manifestations. Although VKH disease rarely occurs in the pediatric population, the clinical course tends to be aggressive, and the visual...

Descripción completa

Detalles Bibliográficos
Autores principales: Katsuyama, Atsuko, Kusuhara, Sentaro, Awano, Hiroyuki, Nagase, Hiroaki, Matsumiya, Wataru, Nakamura, Makoto
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6693137/
https://www.ncbi.nlm.nih.gov/pubmed/31409322
http://dx.doi.org/10.1186/s12886-019-1192-0
_version_ 1783443650552791040
author Katsuyama, Atsuko
Kusuhara, Sentaro
Awano, Hiroyuki
Nagase, Hiroaki
Matsumiya, Wataru
Nakamura, Makoto
author_facet Katsuyama, Atsuko
Kusuhara, Sentaro
Awano, Hiroyuki
Nagase, Hiroaki
Matsumiya, Wataru
Nakamura, Makoto
author_sort Katsuyama, Atsuko
collection PubMed
description BACKGROUND: Vogt–Koyanagi–Harada (VKH) disease is a T-cell-mediated autoimmune disorder characterized by bilateral granulomatous panuveitis with various systemic manifestations. Although VKH disease rarely occurs in the pediatric population, the clinical course tends to be aggressive, and the visual prognosis is worse than that in adult patients due to severe ocular complications secondary to recurrent inflammation. CASE PRESENTATION: A 3-year-old girl with probable VKH was referred to Kobe University Hospital. She had severe bilateral panuveitis with posterior synechiae of the iris, marked optic disk swelling, and serous retinal detachment in both eyes, and her best corrected visual acuities (BCVAs) were 20/200 OD and 20/125 OS. A third course of therapy was administered because serous retinal detachment remained after two courses of therapy. She was treated with three courses of high-dose intravenous corticosteroid therapy, followed by slow tapering of oral corticosteroids. Her BCVAs recovered to 20/16 OU, and relapse of ocular inflammation and side effect of treatment were not observed during the 1.5-year follow-up period. CONCLUSIONS: We experienced a pediatric patient with probable VKH disease who was treated with three courses of high-dose intravenous corticosteroid therapy. With the favorable clinical course in our patient, initial treatment with repeated high-dose intravenous corticosteroid therapy might be beneficial in pediatric VKH disease.
format Online
Article
Text
id pubmed-6693137
institution National Center for Biotechnology Information
language English
publishDate 2019
publisher BioMed Central
record_format MEDLINE/PubMed
spelling pubmed-66931372019-08-16 A case of probable Vogt–Koyanagi–Harada disease in a 3-year-old girl Katsuyama, Atsuko Kusuhara, Sentaro Awano, Hiroyuki Nagase, Hiroaki Matsumiya, Wataru Nakamura, Makoto BMC Ophthalmol Case Report BACKGROUND: Vogt–Koyanagi–Harada (VKH) disease is a T-cell-mediated autoimmune disorder characterized by bilateral granulomatous panuveitis with various systemic manifestations. Although VKH disease rarely occurs in the pediatric population, the clinical course tends to be aggressive, and the visual prognosis is worse than that in adult patients due to severe ocular complications secondary to recurrent inflammation. CASE PRESENTATION: A 3-year-old girl with probable VKH was referred to Kobe University Hospital. She had severe bilateral panuveitis with posterior synechiae of the iris, marked optic disk swelling, and serous retinal detachment in both eyes, and her best corrected visual acuities (BCVAs) were 20/200 OD and 20/125 OS. A third course of therapy was administered because serous retinal detachment remained after two courses of therapy. She was treated with three courses of high-dose intravenous corticosteroid therapy, followed by slow tapering of oral corticosteroids. Her BCVAs recovered to 20/16 OU, and relapse of ocular inflammation and side effect of treatment were not observed during the 1.5-year follow-up period. CONCLUSIONS: We experienced a pediatric patient with probable VKH disease who was treated with three courses of high-dose intravenous corticosteroid therapy. With the favorable clinical course in our patient, initial treatment with repeated high-dose intravenous corticosteroid therapy might be beneficial in pediatric VKH disease. BioMed Central 2019-08-13 /pmc/articles/PMC6693137/ /pubmed/31409322 http://dx.doi.org/10.1186/s12886-019-1192-0 Text en © The Author(s). 2019 Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
spellingShingle Case Report
Katsuyama, Atsuko
Kusuhara, Sentaro
Awano, Hiroyuki
Nagase, Hiroaki
Matsumiya, Wataru
Nakamura, Makoto
A case of probable Vogt–Koyanagi–Harada disease in a 3-year-old girl
title A case of probable Vogt–Koyanagi–Harada disease in a 3-year-old girl
title_full A case of probable Vogt–Koyanagi–Harada disease in a 3-year-old girl
title_fullStr A case of probable Vogt–Koyanagi–Harada disease in a 3-year-old girl
title_full_unstemmed A case of probable Vogt–Koyanagi–Harada disease in a 3-year-old girl
title_short A case of probable Vogt–Koyanagi–Harada disease in a 3-year-old girl
title_sort case of probable vogt–koyanagi–harada disease in a 3-year-old girl
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6693137/
https://www.ncbi.nlm.nih.gov/pubmed/31409322
http://dx.doi.org/10.1186/s12886-019-1192-0
work_keys_str_mv AT katsuyamaatsuko acaseofprobablevogtkoyanagiharadadiseaseina3yearoldgirl
AT kusuharasentaro acaseofprobablevogtkoyanagiharadadiseaseina3yearoldgirl
AT awanohiroyuki acaseofprobablevogtkoyanagiharadadiseaseina3yearoldgirl
AT nagasehiroaki acaseofprobablevogtkoyanagiharadadiseaseina3yearoldgirl
AT matsumiyawataru acaseofprobablevogtkoyanagiharadadiseaseina3yearoldgirl
AT nakamuramakoto acaseofprobablevogtkoyanagiharadadiseaseina3yearoldgirl
AT katsuyamaatsuko caseofprobablevogtkoyanagiharadadiseaseina3yearoldgirl
AT kusuharasentaro caseofprobablevogtkoyanagiharadadiseaseina3yearoldgirl
AT awanohiroyuki caseofprobablevogtkoyanagiharadadiseaseina3yearoldgirl
AT nagasehiroaki caseofprobablevogtkoyanagiharadadiseaseina3yearoldgirl
AT matsumiyawataru caseofprobablevogtkoyanagiharadadiseaseina3yearoldgirl
AT nakamuramakoto caseofprobablevogtkoyanagiharadadiseaseina3yearoldgirl