Diffuse chronic leptomeningitis with seropositive rheumatoid arthritis: report of a case successfully treated as rheumatoid leptomeningitis

Shimada, Kota; Matsui, Toshihiro; Kawakami, Misato; Hayakawa, Hiromi; Futami, Hidekazu; Michishita, Koji; Takaoka, Hirokazu; Ikenaka, Tatsuoh; Komiya, Akiko; Nakayama, Hisanori; Hagiwara, Futoshi; Suzuki, Shoji; Furukawa, Hiroshi; Ozawa, Yoshihito; Tohma, Shigeto

doi:10.1007/s10165-009-0186-9

Cited by 24 publications

(33 citation statements)

References 19 publications

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“…1 In contrast, relatively few cases of rheumatoid meningitis show diffuse symmetric meningeal enhancement. 2 Brain MRI findings, which demonstrate unilateral meningeal thickening with high signal intensities along the adjacent subarachnoid space on FLAIR images, and bright signals on DWI are helpful to support diagnosis 3,4 ; however, to our knowledge, the diagnostic importance of asymmetry on brain MRI in rheumatoid meningitis has not been systematically evaluated. To investigate how frequently asymmetry on brain MRI is observed in patients with rheumatoid meningitis, a systematic search of publications listed in PubMed from 2005 to 2016 was conducted using the keywords "rheumatoid" with "meningitis," "leptomeningitis," and "pachymeningitis."…”

Section: Oy-stersmentioning

confidence: 99%

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Pearls & Oy-sters: Asymmetric meningeal involvement is a common feature of rheumatoid meningitis

et al. 2017

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Pearls & Oy-sters: Asymmetric meningeal involvement is a common feature of rheumatoid meningitis PEARLS• Rheumatoid meningitis is a rare complication of rheumatoid arthritis that requires a meningeal biopsy for diagnosis.• Asymmetric meningeal involvement is a common, although not pathognomonic, brain MRI finding in rheumatoid meningitis. OY-STERS• Because various immunosuppressants are used for the treatment of rheumatoid arthritis, a thorough investigation to exclude opportunistic infections should be conducted before the diagnosis of rheumatoid meningitis.• Immunoglobulin G4-related disease, granulomatous diseases such as granulomatosis with polyangiitis, neurosarcoidosis, neurosyphilis, and meningeal metastasis are possible considerations in the differential diagnosis of rheumatoid meningitis.CASE REPORT A 65-year-old woman with a 3-year history of rheumatoid arthritis presented with headache, confusion, a decreased level of consciousness, and recurrent left hemiparesis. Her rheumatoid arthritis was well-controlled with oral prednisolone, methotrexate, and leflunomide. Serum levels of rheumatoid factor and anticyclic citrullinated peptide antibody were 69.3 and 48.8 U/mL, respectively. Brain MRI revealed mild hydrocephalus and diffuse asymmetric high signal intensities with patchy nodules, which were prominent along the leptomeninges and subarachnoid space of the right frontoparietal lobe on fluid-attenuated inversion recovery (FLAIR) images and diffusionweighted imaging (DWI). Gadolinium uptake was observed in the leptomeninges over the right hemisphere on postcontrast T1-weighted images (figure 1, A-C). The results of CSF analysis showed an opening pressure of 15 cm H 2 O, leukocyte count of 20/mL (54% neutrophils, 31% lymphocytes, 15% other cells), protein level of 113.4 mg/dL, and glucose level of 43 mg/dL; however, within 2 weeks, the CSF opening pressure became elevated, reaching 27 cm H 2 O. In addition, CSF rheumatoid factor was detected at a level of 17.6 IU/mL. An extensive workup for infectious diseases, autoimmune diseases including vasculitis, and leptomeningeal metastasis was performed. CSF cytology for malignant cells as well as Gram stain and culture were negative. Tests for Mycobacterium tuberculosis and fungus in the CSF were negative. CSF PCR results for Hemophilus influenzae, Streptococcus agalactiae, Neisseria meningitidis, Streptococcus pneumoniae, Listeria monocytogenes, herpes simplex virus, varicella-zoster virus, EpsteinBarr virus, cytomegalovirus, and enterovirus were negative. Serum levels of immunoglobulin G subclass IV (IgG4), antinuclear antibody, anti-neutrophil cytoplasmic antibodies, anti-dsDNA antibody, antiRo/La antibodies, and angiotensin converting enzyme were normal. The EEG results showed regional slowing in the right frontocentral region. A biopsy from the right frontal meninges revealed necrotic granulomatous inflammation with dense infiltration of CD68-positive histiocytes as well as focal vasculitis in the leptomeninges ( figure 1, D-F). Rheumatoid nodules were not...

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Section: Oy-stersmentioning

confidence: 99%

“…Unilateral meningeal involvement in rheumatoid meningitis is not considered uncommon. 1,2 Some previous cases had presented with stroke-like episodes of abrupt-onset hemiparesis. 1 In contrast, relatively few cases of rheumatoid meningitis show diffuse symmetric meningeal enhancement.…”

Section: Oy-stersmentioning

confidence: 99%

Pearls & Oy-sters: Asymmetric meningeal involvement is a common feature of rheumatoid meningitis

et al. 2017

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“…1,3,4 False negatives may be seen. 5,6 Presence of rheumatoid factor in the spinal fluid seems to be specifically associated with meningeal disease and could be used as a biomarker. 6 Imaging with cerebral MRI is helpful to support diagnosis.…”

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“…1 It is unclear whether earlier diagnosis with aggressive immunotherapy would improve survival and morbidity. 2,5,7 Corticosteroid alone or combined with cyclophosphamide, methotrexate, or azathioprine has been used with variable results. 1,5,6 The underlying pathophysiology may involve cortical spreading depression induced by inflammation of the adjacent meninges.…”

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confidence: 99%

“…2,5,7 Corticosteroid alone or combined with cyclophosphamide, methotrexate, or azathioprine has been used with variable results. 1,5,6 The underlying pathophysiology may involve cortical spreading depression induced by inflammation of the adjacent meninges. Vascular steal phenomenon appears less probable as no DWI abnormality was observed despite prolonged neurologic deficits lasting up to 8 hours.…”

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Rheumatoid meningitis presenting with stroke-like episodes

2014

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Rheumatoid Meningitis Presenting With Multiple Strokelike Episodes

2015

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cortex (14.2% astrocytes; 4.8% neurons; 325 cells), 6.4% in CA1 (18% astrocytes; 9.6% neurons; 941 cells), 5.7% in caudatum (20% astrocytes; 6.4% neurons; 470 cells), and 5.3% in cerebellum (3.2% astrocytes; 6.0% neurons; 225 cells). Discussion | Greco et al 6 described the presence of inclusions in 11 cases of men with FXTAS aged 67 to 87 years. In our patient, the number of inclusions were in the low range when compared with those cases. In summary, FXTAS is thought to be a disorder of aging in carriers of FMR1 premutation; however, this case documents that FXTAS can occur earlier in adult life, particularly if another disease process is occurring, such as substance abuse, that may exacerbate the pathological process of FXTAS.

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Diffuse chronic leptomeningitis with seropositive rheumatoid arthritis: report of a case successfully treated as rheumatoid leptomeningitis

Cited by 24 publications

References 19 publications

Pearls & Oy-sters: Asymmetric meningeal involvement is a common feature of rheumatoid meningitis

Pearls & Oy-sters: Asymmetric meningeal involvement is a common feature of rheumatoid meningitis

Rheumatoid meningitis presenting with stroke-like episodes

Rheumatoid Meningitis Presenting With Multiple Strokelike Episodes

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