Diagnosis of Intravascular Lymphoma by a Novel Biopsy Site

Diagnosis of Intravascular Lymphoma by a Novel Biopsy Site
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DOI:
10.1016/j.ophtha.2010.08.001
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发表时间:
2011-03-01
期刊:
影响因子:
13.7
通讯作者:
Weinberg, Robert S.
Weinberg, Robert S.
中科院分区:
医学1区
文献类型:
--
作者:
Lee, Bryan S.;Frankfort, Benjamin J.;Weinberg, Robert S.

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目的:报告第一例已知的血管内淋巴瘤(IVL),一种罕见的疾病,通过泪腺活检诊断。设计:介入性病例报告。参与者:单个患者病例报告。干预:床边泪腺活检。主要结果测量:通过新的活检部位临床病理诊断IVL,一种只有少数眼科表现的肿瘤。结果:一位70岁的中国男性,患有6个月的进行性截瘫,主诉双眼视力模糊。他的脑脊液检查和磁共振成像显示异常胸髓信号和脑室周围脑白色物质变化,垂体和柄正常。散瞳眼底检查显示视网膜内和视网膜下多灶性出血伴浆液性视网膜脱离。检查包括血清学检查和2例正常骨髓活检,但未发现潜在病因。由于持续高度怀疑恶性过程,我们进行了床边经结膜活检的泪腺。这表明大的,非典型的CD 20阳性B细胞局限于血管腔,符合IVL,一种不寻常的形式的大B细胞lymphoma.Conclusions:这种情况下,IVL已被检测到通过泪腺活检,这可能是一个有用的方法来调查隐源性神经过程。此外,这是唯一的报告的情况下,眼科IVL诊断前尸检。IVL是一种罕见的疾病,但有多变的表现,涉及中枢神经系统,皮肤,骨髓,肝脏和脾脏。眼睛也应该被认为是受累的终末器官。
Purpose: To report the first known case of intravascular lymphoma (IVL), a rare disease, diagnosed via lacrimal gland biopsy.Design: Interventional case report.Participants: Single patient case report.Intervention: Bedside lacrimal gland biopsy.Main Outcome Measures: Clinicopathologic diagnosis of IVL, a neoplasm with only a few prior reports of ophthalmic manifestations, via a novel biopsy site.Results: A 70-year-old Chinese man with 6 months of progressive paraplegia complained of blurred vision in both eyes. He had unremarkable cerebrospinal fluid studies and magnetic resonance imaging that showed abnormal thoracic cord signal and periventricular brain white matter changes with a normal pituitary gland and stalk. Dilated fundus examination showed multifocal areas of intra-and subretinal hemorrhage with serous retinal detachment. The workup included serologies and 2 normal bone marrow biopsies but did not reveal the underlying etiology. Because of the continued high suspicion for a malignant process, we performed bedside transconjunctival biopsy of the lacrimal gland. This demonstrated large, atypical CD20-positive B cells confined to the vessel lumina, consistent with IVL, an unusual form of large B-cell lymphoma.Conclusions: This case represents the first time that IVL has been detected via lacrimal gland biopsy, which may be a useful way to investigate cryptogenic neurologic processes. Furthermore, it is one of the only reported cases of ophthalmic IVL diagnosed before autopsy. IVL is a rare disease but has protean manifestations involving the central nervous system, skin, bone marrow, liver, and spleen. The eye should also be considered an end organ for involvement.