A 69-year-old woman presented with severe subacute painful meningoradiculoneuritis. Neurophysiology showed a patchy, proximal axonal process with widespread denervation. Cerebrospinal fluid (CSF) was lymphocytic (normal T-cell predominant) with negative cytology. MRI revealed multiple sites of enhancement, but fluorodeoxyglucose positron emission tomography was negative. Bone marrow aspirate and trephine (BMAT) showed no evidence of a lymphoproliferative condition. Right brachial plexus biopsy demonstrated mixed T-cell/B-cell endoneurial inflammation not fulfilling criteria for vasculitis. She was stabilised with high-dose steroids and cyclophosphamide, followed by mycophenolate for inflammatory myeloradiculoneuritis. However, symptoms recurred when prednisolone was weaned. Although T-cell receptor gene analysis from the initial CSF demonstrated clonal rearrangements, it was only when the same clones were identified on two repeat BMATs and CSF that T-cell neurolymphomatosis, an exceedingly rare condition, was diagnosed. This case highlights the diagnostic challenge in peripheral neurolymphomatosis related to patchy disease, variable sensitivity and specificity of investigative tools, and the influence of therapies on traditional cytological definitions of lymphoma. The clinical picture, exhaustive exclusion of alternative causes and the persistence of an abnormal T-cell clone ultimately lead to a diagnostic consensus between specialist neurology and haematology clinicians.
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http://dx.doi.org/10.1136/bcr-2019-232538 | DOI Listing |
BJC Rep
April 2024
Beth Israel Deaconess Medical Center & Harvard Medical School, Boston, MA, USA.
Background: Neurolymphomatosis is a lymphoid malignancy of the peripheral nervous system and its natural history is poorly understood.
Methods: We performed PubMed search and extracted clinical data for Kaplan-Meier statistics to determine outcome parameters over time. Kruskal-Wallis test was performed to compare prognostic factors.
Am J Hematol
December 2024
Department of Neurology, Division of Neuro-Oncology, Massachusetts General Hospital Cancer Center, Harvard Medical School, Boston, Massachusetts, USA.
Lymphomatous infiltration of the peripheral nervous system (PNS), termed neurolymphomatosis, represents a distinct extranodal non-Hodgkin lymphoma variant with dismal outcome. CD19-directed chimeric antigen receptor (CD19-CAR) T-cell therapy has emerged as a safe and effective treatment for B-cell lymphomas. We aimed to assess toxicity and efficacy of CD19-CAR T-cells in neurolymphomatosis.
View Article and Find Full Text PDFNeurology
September 2024
From the Departments of Neurology (M.P.S., J.M.M.-T., M.L.M., C.J.K., P.J.D., P.J.B.D.), Laboratory Medicine and Pathology (R.L.K., W.R.M.), Hematology (T.M.H., P.B.J., I.N.M., A.K.), Radiology (K.A.), Neurosurgery (R.J.S.), and Quantitative Health Sciences (J.M.), Mayo Clinic, Rochester, MN; and Neurological Institute of Thailand (N.S.), Bangkok.
Background And Objectives: Neurolymphomatosis (NL) is characterized by lymphomatous infiltration of the peripheral nervous system presenting as the initial manifestation of a lymphoma (primary NL [PNL]) or in relapse of a known lymphoma (secondary NL [SNL]). This report details and compares the neurologic clinicopathologic characteristics of these 2 groups.
Methods: This retrospective study was performed on patients diagnosed with pathologically confirmed NL in nerve between January 1, 1992, and June 31, 2020.
J Med Case Rep
July 2024
General Medicine Center, Shimane University Hospital, Shimane, Japan.
Background: Primary central nervous system lymphoma is rare, and primary central nervous system T cell lymphoma is relatively uncommon, contributing to < 5% of all cases. Lymphomatosis cerebri, a rare subtype of primary central nervous system lymphoma, is characterized by extensive white-matter lesions on magnetic resonance imaging and nonspecific symptoms, such as cognitive decline and depression. Reports of lymphomatosis cerebri in adult T cell leukemia/lymphoma are limited.
View Article and Find Full Text PDFLeuk Lymphoma
December 2024
Department of Hematology and Oncology, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan.
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