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印度尼西亚儿童皮肤朗格汉斯细胞组织细胞增多症病例系列;临床病理谱。

Case series of cutaneous Langerhans cell histiocytosis in Indonesian children; The clinicopathological spectrum.

作者信息

Widodo Irianiwati, Sahara Nita, Dwianingsih Ery Kus, Ferronika Paranita

机构信息

Department of Anatomical Pathology, Faculty of Medicine, Public Health and Nursing, Universitas Gadjah Mada, Yogyakarta, Indonesia.

出版信息

Dermatol Reports. 2020 Oct 22;12(2):8777. doi: 10.4081/dr.2020.8777. eCollection 2020 Sep 23.

Abstract

Langerhans Cell Histiocytosis (LCH) is a rare disease characterized by the clonal proliferation of Langerhans cells, which are immunoreactive to S-100 and CD-1a/ CD207 (Langerin). Cutaneous involvement is the most common presentation of LCH in children. It is suggested that the patients with single-system LCH limited to the skin have a better prognosis than those with systemic involvement. Three histologic reactions of cutaneous LCH have been reported and are associated with the clinical types of LCH. These histological reactions include: proliferative, granulomatous, and xanthomatous. This study presents the clinicopathological features of ten cutaneous LCH cases collected from Dr. Sardjito General Hospital Yogyakarta Indonesia between 2014-2018. The ten cases showed various clinical features, in which some features mimic other diseases. The microscopic features of skin biopsies showed granulomatous reaction in 80% of cases and proliferative reaction in the other 20%. Five patients (50% of cases) who died had systemic manifestation of thrombocytopenia, anemia, icterus, hepatosplenomegaly, and revealed the granulomatous type from their skin biopsy specimens. The clinical recognition of LCH and subsequent histological reaction determination are important since some cases may develop multisystem disease and have a poor prognosis.

摘要

朗格汉斯细胞组织细胞增多症(LCH)是一种罕见疾病,其特征为朗格汉斯细胞的克隆性增殖,这些细胞对S-100和CD-1a/CD207(朗格素)具有免疫反应性。皮肤受累是儿童LCH最常见的表现形式。有观点认为,局限于皮肤的单系统LCH患者的预后优于有全身受累的患者。已报道皮肤LCH的三种组织学反应,且这些反应与LCH的临床类型相关。这些组织学反应包括:增殖性、肉芽肿性和黄瘤性。本研究呈现了2014年至2018年期间从印度尼西亚日惹萨迪托综合医院收集的10例皮肤LCH病例的临床病理特征。这10例病例表现出各种临床特征,其中一些特征类似其他疾病。皮肤活检的微观特征显示,80%的病例有肉芽肿反应,另外20%有增殖反应。死亡的5例患者(占病例的50%)有血小板减少、贫血、黄疸、肝脾肿大等全身表现,且其皮肤活检标本显示为肉芽肿类型。LCH 的临床识别及随后的组织学反应判定很重要,因为一些病例可能发展为多系统疾病且预后不良。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c0a1/7772770/d5c307810cbc/dr-12-2-8777-g001.jpg

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