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Bilateral Spontaneous Hyphema in Juvenile Xanthogranuloma: A Case Report

Juvenile xanthogranuloma (JXG) is an idiopathic granulomatous inflammatory condition that usually affects children. Intraocular involvement, especially bilateral, is rare in JXG. Most patients with ocular lesions are typically infants and usually present with hyphema, iridocyclitis, and secondary gl...

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Autores principales: Al-Owaid, Abdullah A., Al-Dossari, Shaikha, Maktabi, Azza, Edward, Deepak, Sesma, Gorka
Formato: Online Artículo Texto
Lenguaje:English
Publicado: S. Karger AG 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7841722/
https://www.ncbi.nlm.nih.gov/pubmed/33568985
http://dx.doi.org/10.1159/000510905
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author Al-Owaid, Abdullah A.
Al-Dossari, Shaikha
Maktabi, Azza
Edward, Deepak
Sesma, Gorka
author_facet Al-Owaid, Abdullah A.
Al-Dossari, Shaikha
Maktabi, Azza
Edward, Deepak
Sesma, Gorka
author_sort Al-Owaid, Abdullah A.
collection PubMed
description Juvenile xanthogranuloma (JXG) is an idiopathic granulomatous inflammatory condition that usually affects children. Intraocular involvement, especially bilateral, is rare in JXG. Most patients with ocular lesions are typically infants and usually present with hyphema, iridocyclitis, and secondary glaucoma. We report a case of a 3-month-old baby girl who presented to our emergency department with bilateral hyphema that started 3 weeks ago. She was medically free with no history of any recent trauma or preceding febrile illness. General physical examination showed a quiet baby with multiple hyperpigmented macules over the inner thigh and right upper arm, with one pinkish nodule over the occiput. She also had high intraocular pressures. A detailed ophthalmic assessment was done under general anesthesia. The nodular lesion was excised and sent for histopathological evaluation, which confirmed the diagnosis of JXG. Treatment of JXG cases present a challenge to ophthalmologist due to rebleeding and refractory glaucoma. Our case was admitted multiple times for rebleeding and refractory glaucoma and was treated with full antiglaucoma drops, steroid drops and peribulbar injection of steroid.
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spelling pubmed-78417222021-02-09 Bilateral Spontaneous Hyphema in Juvenile Xanthogranuloma: A Case Report Al-Owaid, Abdullah A. Al-Dossari, Shaikha Maktabi, Azza Edward, Deepak Sesma, Gorka Case Rep Ophthalmol Case Report Juvenile xanthogranuloma (JXG) is an idiopathic granulomatous inflammatory condition that usually affects children. Intraocular involvement, especially bilateral, is rare in JXG. Most patients with ocular lesions are typically infants and usually present with hyphema, iridocyclitis, and secondary glaucoma. We report a case of a 3-month-old baby girl who presented to our emergency department with bilateral hyphema that started 3 weeks ago. She was medically free with no history of any recent trauma or preceding febrile illness. General physical examination showed a quiet baby with multiple hyperpigmented macules over the inner thigh and right upper arm, with one pinkish nodule over the occiput. She also had high intraocular pressures. A detailed ophthalmic assessment was done under general anesthesia. The nodular lesion was excised and sent for histopathological evaluation, which confirmed the diagnosis of JXG. Treatment of JXG cases present a challenge to ophthalmologist due to rebleeding and refractory glaucoma. Our case was admitted multiple times for rebleeding and refractory glaucoma and was treated with full antiglaucoma drops, steroid drops and peribulbar injection of steroid. S. Karger AG 2020-12-21 /pmc/articles/PMC7841722/ /pubmed/33568985 http://dx.doi.org/10.1159/000510905 Text en Copyright © 2020 by S. Karger AG, Basel http://creativecommons.org/licenses/by-nc/4.0/ This article is licensed under the Creative Commons Attribution-NonCommercial-4.0 International License (CC BY-NC) (http://www.karger.com/Services/OpenAccessLicense). Usage and distribution for commercial purposes requires written permission.
spellingShingle Case Report
Al-Owaid, Abdullah A.
Al-Dossari, Shaikha
Maktabi, Azza
Edward, Deepak
Sesma, Gorka
Bilateral Spontaneous Hyphema in Juvenile Xanthogranuloma: A Case Report
title Bilateral Spontaneous Hyphema in Juvenile Xanthogranuloma: A Case Report
title_full Bilateral Spontaneous Hyphema in Juvenile Xanthogranuloma: A Case Report
title_fullStr Bilateral Spontaneous Hyphema in Juvenile Xanthogranuloma: A Case Report
title_full_unstemmed Bilateral Spontaneous Hyphema in Juvenile Xanthogranuloma: A Case Report
title_short Bilateral Spontaneous Hyphema in Juvenile Xanthogranuloma: A Case Report
title_sort bilateral spontaneous hyphema in juvenile xanthogranuloma: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7841722/
https://www.ncbi.nlm.nih.gov/pubmed/33568985
http://dx.doi.org/10.1159/000510905
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