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Isolated Ocular Relapse Following Prolonged Remission in Primary Central Nervous System Lymphoma: A Case Report
Hamoud Moza1, Engelbrecht Jansen2, Oakey Zackery3
1Department of Medicine, Eisenhower Health, Rancho Mirage, CA, USA.
Journal of Investigative Medicine High Impact Case Reports
|August 11, 2026
Summary
Primary central nervous system lymphoma (PCNSL) can relapse in the eye years after treatment, even without brain recurrence. Early ocular oncology evaluation is crucial for diagnosing and managing this rare, late-stage complication.
Area of Science:
- Neuro-oncology
- Ophthalmology
- Hematology
Background:
- Primary central nervous system lymphoma (PCNSL) is a rare malignancy primarily affecting the brain and leptomeninges.
- High-dose methotrexate (HD-MTX)-based therapies have improved PCNSL outcomes, but relapse, often within the CNS, remains a significant challenge.
Purpose of the Study:
- To report a rare case of isolated ocular relapse of PCNSL after a prolonged period of central nervous system (CNS) remission.
- To highlight the diagnostic challenges and importance of ophthalmologic surveillance in detecting late ocular recurrences of PCNSL.
Main Methods:
- Case report of a 78-year-old female diagnosed with PCNSL.
- Treatment with high-dose methotrexate (HD-MTX) and rituximab leading to complete remission.
- Subsequent development of unilateral visual decline, initially misdiagnosed, followed by ophthalmologic evaluation revealing ocular relapse.
Main Results:
- The patient achieved nearly five years of CNS disease remission.
- Isolated ocular relapse involving the vitreous and retina was confirmed despite negative serial neuroimaging.
- Retreatment with HD-MTX and rituximab led to significant clinical and fundoscopic improvement.
Conclusions:
- Delayed isolated ocular relapse is an uncommon but significant pattern in PCNSL survivors.
- Neuroimaging has limitations in detecting ocular disease, necessitating vigilant ophthalmologic surveillance, especially in patients with visual symptoms.
- Systemic retreatment with HD-MTX and rituximab can be effective for late ocular relapses in PCNSL.
