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On the knowledge of solitary juvenile xanthogranuloma of the eyelid: a case series and literature review

PURPOSE: Solitary eyelid juvenile xanthogranuloma (JXG) is extremely rare, and there is limited literature on its clinical features and treatment outcomes. Here, we present a case series and comprehensive review of the literature on patients with isolated eyelid JXG. METHODS: We systematically extra...

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Autores principales: Chen, Rongxin, Liu, Shu, Tang, Lijuan, Yu, Xinyue, Meng, Ziwei, Hu, Yu, Li, Jing, Liang, Xuanwei
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Springer Berlin Heidelberg 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9203400/
https://www.ncbi.nlm.nih.gov/pubmed/35084531
http://dx.doi.org/10.1007/s00417-022-05560-6
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author Chen, Rongxin
Liu, Shu
Tang, Lijuan
Yu, Xinyue
Meng, Ziwei
Hu, Yu
Li, Jing
Liang, Xuanwei
author_facet Chen, Rongxin
Liu, Shu
Tang, Lijuan
Yu, Xinyue
Meng, Ziwei
Hu, Yu
Li, Jing
Liang, Xuanwei
author_sort Chen, Rongxin
collection PubMed
description PURPOSE: Solitary eyelid juvenile xanthogranuloma (JXG) is extremely rare, and there is limited literature on its clinical features and treatment outcomes. Here, we present a case series and comprehensive review of the literature on patients with isolated eyelid JXG. METHODS: We systematically extracted data from our institution’s records of isolated eyelid JXG cases and conducted a search for additional cases from the literature utilising the PubMed, Wanfang, and Chinese National Knowledge Infrastructure (CNKI) databases. Patients with JXG were analysed with respect to age, sex, clinical presentation, therapy, and outcome. Group comparisons were performed. RESULTS: Thirty-two patients (including 13 at our institution and 19 from prior publications) were identified. The median age at first presentation was higher in current patients than in the patients from the published cases (median 9 years, range 1.2 to 47.0 years; median 2 years, range 0.5 months to 46.0 years, respectively, P = 0.014). Of the patients who had known characteristics, no significant differences were observed between the two groups in terms of sex, affected eye, eyelid site, type of cutaneous involvement, or duration of symptoms (each P > 0.05). Seventeen (54.8%) patients were male. The most common lesion location was the upper eyelid (n = 10, 62.5%). Twenty-four (75.0%) cutaneous lesions had full-thickness skin involvement; 8 (25.0%) subcutaneous masses had a chalazion-like appearance. Histologically, the JXG masses were characterised by Touton giant cells with inflammatory cells. Additionally, there was no significant difference in treatment modalities between the two groups (P = 0.072), and 24 (75.0%) patients underwent surgical excision. The overall recurrence-free survival was 3.6 to 52.8 (median 27.0) months in the current patients. For published cases with available follow-up information, there was no recurrence in 10 cases and improvement in 1 case, with a median follow-up of 9.5 months. CONCLUSION: Solitary eyelid JXG is a rare clinical entity and should be included in the differential diagnosis of eyelid mass lesions in patients of all age groups. Surgical excision is often selected for efficient treatment and to obtain an excisional biopsy. [Image: see text] SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1007/s00417-022-05560-6.
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spelling pubmed-92034002022-06-18 On the knowledge of solitary juvenile xanthogranuloma of the eyelid: a case series and literature review Chen, Rongxin Liu, Shu Tang, Lijuan Yu, Xinyue Meng, Ziwei Hu, Yu Li, Jing Liang, Xuanwei Graefes Arch Clin Exp Ophthalmol Oculoplastics and Orbit PURPOSE: Solitary eyelid juvenile xanthogranuloma (JXG) is extremely rare, and there is limited literature on its clinical features and treatment outcomes. Here, we present a case series and comprehensive review of the literature on patients with isolated eyelid JXG. METHODS: We systematically extracted data from our institution’s records of isolated eyelid JXG cases and conducted a search for additional cases from the literature utilising the PubMed, Wanfang, and Chinese National Knowledge Infrastructure (CNKI) databases. Patients with JXG were analysed with respect to age, sex, clinical presentation, therapy, and outcome. Group comparisons were performed. RESULTS: Thirty-two patients (including 13 at our institution and 19 from prior publications) were identified. The median age at first presentation was higher in current patients than in the patients from the published cases (median 9 years, range 1.2 to 47.0 years; median 2 years, range 0.5 months to 46.0 years, respectively, P = 0.014). Of the patients who had known characteristics, no significant differences were observed between the two groups in terms of sex, affected eye, eyelid site, type of cutaneous involvement, or duration of symptoms (each P > 0.05). Seventeen (54.8%) patients were male. The most common lesion location was the upper eyelid (n = 10, 62.5%). Twenty-four (75.0%) cutaneous lesions had full-thickness skin involvement; 8 (25.0%) subcutaneous masses had a chalazion-like appearance. Histologically, the JXG masses were characterised by Touton giant cells with inflammatory cells. Additionally, there was no significant difference in treatment modalities between the two groups (P = 0.072), and 24 (75.0%) patients underwent surgical excision. The overall recurrence-free survival was 3.6 to 52.8 (median 27.0) months in the current patients. For published cases with available follow-up information, there was no recurrence in 10 cases and improvement in 1 case, with a median follow-up of 9.5 months. CONCLUSION: Solitary eyelid JXG is a rare clinical entity and should be included in the differential diagnosis of eyelid mass lesions in patients of all age groups. Surgical excision is often selected for efficient treatment and to obtain an excisional biopsy. [Image: see text] SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1007/s00417-022-05560-6. Springer Berlin Heidelberg 2022-01-27 2022 /pmc/articles/PMC9203400/ /pubmed/35084531 http://dx.doi.org/10.1007/s00417-022-05560-6 Text en © The Author(s) 2022 https://creativecommons.org/licenses/by/4.0/Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) .
spellingShingle Oculoplastics and Orbit
Chen, Rongxin
Liu, Shu
Tang, Lijuan
Yu, Xinyue
Meng, Ziwei
Hu, Yu
Li, Jing
Liang, Xuanwei
On the knowledge of solitary juvenile xanthogranuloma of the eyelid: a case series and literature review
title On the knowledge of solitary juvenile xanthogranuloma of the eyelid: a case series and literature review
title_full On the knowledge of solitary juvenile xanthogranuloma of the eyelid: a case series and literature review
title_fullStr On the knowledge of solitary juvenile xanthogranuloma of the eyelid: a case series and literature review
title_full_unstemmed On the knowledge of solitary juvenile xanthogranuloma of the eyelid: a case series and literature review
title_short On the knowledge of solitary juvenile xanthogranuloma of the eyelid: a case series and literature review
title_sort on the knowledge of solitary juvenile xanthogranuloma of the eyelid: a case series and literature review
topic Oculoplastics and Orbit
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9203400/
https://www.ncbi.nlm.nih.gov/pubmed/35084531
http://dx.doi.org/10.1007/s00417-022-05560-6
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