Background: Castleman Disease (CD) describes a rare group of heterogenous diseases characterized by lymph node enlargement. It has been associated with various neoplastic, autoimmune, and infectious processes. CD has two main histological forms, hyaline vascular and plasma cell type. Clinically, there are two forms of CDunicentric and multicentric. CD involvement of the central nervous system (CNS) is rare. We present a rare case of unicentric CD that presented as leptomeningeal enhancement and a review on the previous literature of similar cases.Case Report: A 41-year-old man with no past medical history presented with a first time seizure with persistent numbness of his left face, left arm, and left leg. MRI brain showed leptomeningeal involvement. Extensive workup done for malignant, autoimmune and infectious etiologies during admission was unremarkable. The patient was started on antiepileptics with clinical improvement and discharged. As an outpatient, he underwent meningeal biopsy showing CD of hyaline-vascular subtype. He was then seen in hematology clinic and the plan for the patient was to monitor disease progression with MRI. Radiation oncology and neurosurgery were then consulted, who stated that no intervention should be done at this time as the patient was asymptomatic. The patient remains clinically stable.
Conclusion:Leptomeningeal enhancement is a rare presentation of CD and further evidence-based data is warranted to better guide management. In cases like ours in which the patient had unresectable disease, we opted for conservative management with close clinical monitoring.