Pediatric Orbital Juvenile Xanthogranuloma: A Case Series and Review of the Literature.

IF 1.2 4区 医学 Q3 OPHTHALMOLOGY
Ming-Han H Lee, Ebony J Smith, Thomas G Hardy, Nicole Graf, Krishna Tumuluri
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引用次数: 0

Abstract

Purpose: Juvenile xanthogranuloma (JXG) is a subtype of histiocytosis characterised histologically by foamy non-Langerhan cells with Touton giant cells. It typically manifests as a single self-limiting cutaneous nodule in the pediatric population. Orbital JXG is extremely rare, and its clinical course and management are not well understood or defined. Herein we present 3 cases of orbital JXG and provide a detailed literature review.

Methods: Review of 3 cases with orbital JXG and literature review of all published cases.

Results: Three presented cases demonstrate the heterogeneous clinical course of orbital JXG. Although centred around the use of steroids, there is neither robust evidence nor consensus on its management. The wider JXG literature is currently concentrated around the classification of JXG with respect to histiocytosis, especially the exclusion of extracutaneous JXG as separate diseases. This separation is based on clinical, histopathological, and molecular findings. It is unclear where orbital JXG best fits in this emerging classification of JXG.

Conclusion: Our review of the cases and literature on orbital JXG show that it may manifest with variable clinical course and its molecular pathogenic mechanism may be different to that of the cutaneous JXG.

小儿眼眶幼年黄疽瘤:病例系列和文献综述。
目的:幼年黄疽瘤(JXG)是组织细胞增生症的一种亚型,其组织学特征是泡沫状非朗格汉细胞和Touton巨细胞。在儿童中,它通常表现为单个自限性皮肤结节。眼眶 JXG 极其罕见,其临床病程和治疗方法尚未得到很好的理解和界定。在此,我们介绍 3 例眼眶 JXG 病例,并提供详细的文献综述:方法:回顾 3 例眼眶 JXG 病例,并对所有已发表病例进行文献综述:结果:三个病例显示了眼眶 JXG 不同的临床过程。虽然以类固醇的使用为中心,但在其管理方面既没有有力的证据,也没有达成共识。更广泛的 JXG 文献目前主要集中在 JXG 与组织细胞增生症的分类上,尤其是将皮外 JXG 作为独立疾病排除在外。这种分类基于临床、组织病理学和分子研究结果。目前还不清楚眼眶 JXG 在这一新出现的 JXG 分类中的最佳位置:我们对眼眶 JXG 病例和文献的回顾表明,眼眶 JXG 的临床表现可能各不相同,其分子致病机制也可能与皮肤 JXG 不同。
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来源期刊
CiteScore
2.50
自引率
10.00%
发文量
322
审稿时长
3-8 weeks
期刊介绍: Ophthalmic Plastic and Reconstructive Surgery features original articles and reviews on topics such as ptosis, eyelid reconstruction, orbital diagnosis and surgery, lacrimal problems, and eyelid malposition. Update reports on diagnostic techniques, surgical equipment and instrumentation, and medical therapies are included, as well as detailed analyses of recent research findings and their clinical applications.
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