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Published on: February 2, 2014
Diagnosis of intravascular lymphoma by a novel biopsy site
Bryan S Lee1, Benjamin J Frankfort, Charles G Eberhart
1Wilmer Eye Institute, The Johns Hopkins Hospital, Baltimore, Maryland 21287, USA. bryanlee@jhmi.edu
Insights
This case report details the first diagnosis of intravascular lymphoma (IVL) using a lacrimal gland biopsy. This novel approach aids in diagnosing rare neurological conditions with eye involvement.
Area of Science:
- Ophthalmology
- Oncology
- Neurology
Background:
- Intravascular lymphoma (IVL) is a rare hematologic malignancy.
- Ophthalmic manifestations of IVL are uncommon and diagnosis can be challenging.
- Previous reports of IVL have documented involvement of the central nervous system, skin, bone marrow, liver, and spleen.
Observation:
- A 70-year-old man presented with progressive paraplegia and blurred vision.
- Ophthalmic examination revealed intraretinal hemorrhage and serous retinal detachment.
- Initial investigations including CSF studies, MRI, and bone marrow biopsies were inconclusive.
Findings:
- A bedside transconjunctival lacrimal gland biopsy was performed.
- Histopathology revealed atypical CD20-positive B cells within vessel lumina, consistent with IVL.
- This represents the first diagnosis of IVL via lacrimal gland biopsy.
Implications:
- Lacrimal gland biopsy can be a valuable diagnostic tool for cryptogenic neurological processes.
- The eye should be considered a potential end organ for IVL involvement.
- Early diagnosis of IVL, even with unusual presentations, is crucial for patient management.
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.
