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Surgical Treatment for Adult-Onset Limbal Xanthogranuloma: A Case Report with 4-Year Follow-Up

Xanthogranuloma is a benign histiocytic disorder that generally appears in infants and children and often called juvenile xanthogranuloma (JXG). Typical reddish-yellow cutaneous papules or nodules are the most common presentation of JXG. Extracutaneous JXG affects eyes, brain, lungs, liver, spleen,...

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Autores principales: Chuephanich, Pichaya, Kitsirilarp, Warissara, Laoharojvongsa, Nutthaporn, Tantayakom, Theeratep
Formato: Online Artículo Texto
Lenguaje:English
Publicado: S. Karger AG 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9710439/
https://www.ncbi.nlm.nih.gov/pubmed/36466058
http://dx.doi.org/10.1159/000526711
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author Chuephanich, Pichaya
Kitsirilarp, Warissara
Laoharojvongsa, Nutthaporn
Tantayakom, Theeratep
author_facet Chuephanich, Pichaya
Kitsirilarp, Warissara
Laoharojvongsa, Nutthaporn
Tantayakom, Theeratep
author_sort Chuephanich, Pichaya
collection PubMed
description Xanthogranuloma is a benign histiocytic disorder that generally appears in infants and children and often called juvenile xanthogranuloma (JXG). Typical reddish-yellow cutaneous papules or nodules are the most common presentation of JXG. Extracutaneous JXG affects eyes, brain, lungs, liver, spleen, and other sites. Isolated ocular manifestation without skin lesion is rare, especially in adult patients. Here, we report a case of a 27-year-old man who presented with gradually growing yellowish mass at the corneoscleral area of the left eye for 5 months. The patient had worn soft contact lenses for more than 10 years. With atypical age of onset and the absence of skin lesion, total mass excision with lamellar corneoscleral graft and amniotic membrane transplantation was done, and the diagnosis of adult-onset limbal xanthogranuloma was made by histopathological and immunohistochemical examinations. Postoperatively, the patient had good vision with corrected distant visual acuity of 20/30, and the graft was clear. There was no evidence of recurrence at 4-year follow-up. We found that excision with lamellar corneoscleral graft in limbal xanthogranuloma shows good result with no recurrence. The same result occurred to other previous cases reported, so complete excision with graft could be an effective treatment of choice in patient with limbal xanthogranuloma.
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spelling pubmed-97104392022-12-01 Surgical Treatment for Adult-Onset Limbal Xanthogranuloma: A Case Report with 4-Year Follow-Up Chuephanich, Pichaya Kitsirilarp, Warissara Laoharojvongsa, Nutthaporn Tantayakom, Theeratep Case Rep Ophthalmol Case Report Xanthogranuloma is a benign histiocytic disorder that generally appears in infants and children and often called juvenile xanthogranuloma (JXG). Typical reddish-yellow cutaneous papules or nodules are the most common presentation of JXG. Extracutaneous JXG affects eyes, brain, lungs, liver, spleen, and other sites. Isolated ocular manifestation without skin lesion is rare, especially in adult patients. Here, we report a case of a 27-year-old man who presented with gradually growing yellowish mass at the corneoscleral area of the left eye for 5 months. The patient had worn soft contact lenses for more than 10 years. With atypical age of onset and the absence of skin lesion, total mass excision with lamellar corneoscleral graft and amniotic membrane transplantation was done, and the diagnosis of adult-onset limbal xanthogranuloma was made by histopathological and immunohistochemical examinations. Postoperatively, the patient had good vision with corrected distant visual acuity of 20/30, and the graft was clear. There was no evidence of recurrence at 4-year follow-up. We found that excision with lamellar corneoscleral graft in limbal xanthogranuloma shows good result with no recurrence. The same result occurred to other previous cases reported, so complete excision with graft could be an effective treatment of choice in patient with limbal xanthogranuloma. S. Karger AG 2022-11-17 /pmc/articles/PMC9710439/ /pubmed/36466058 http://dx.doi.org/10.1159/000526711 Text en Copyright © 2022 by The Author(s). Published by S. Karger AG, Basel https://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
Chuephanich, Pichaya
Kitsirilarp, Warissara
Laoharojvongsa, Nutthaporn
Tantayakom, Theeratep
Surgical Treatment for Adult-Onset Limbal Xanthogranuloma: A Case Report with 4-Year Follow-Up
title Surgical Treatment for Adult-Onset Limbal Xanthogranuloma: A Case Report with 4-Year Follow-Up
title_full Surgical Treatment for Adult-Onset Limbal Xanthogranuloma: A Case Report with 4-Year Follow-Up
title_fullStr Surgical Treatment for Adult-Onset Limbal Xanthogranuloma: A Case Report with 4-Year Follow-Up
title_full_unstemmed Surgical Treatment for Adult-Onset Limbal Xanthogranuloma: A Case Report with 4-Year Follow-Up
title_short Surgical Treatment for Adult-Onset Limbal Xanthogranuloma: A Case Report with 4-Year Follow-Up
title_sort surgical treatment for adult-onset limbal xanthogranuloma: a case report with 4-year follow-up
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9710439/
https://www.ncbi.nlm.nih.gov/pubmed/36466058
http://dx.doi.org/10.1159/000526711
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