Pyrogenic cytokine interleukin-6 expression by a chordoid meningioma in an adult with a systemic inflammatory syndrome. Case report and review of the literature.

Department of Neurosurgery, Catholic University School of Medicine, Rome, Italy.
Journal of Neurosurgery (Impact Factor: 3.74). 10/2005; 103(3):555-8. DOI: 10.3171/jns.2005.103.3.0555
Source: PubMed


Chordoid meningioma is a rare meningothelial tumor characterized by chordoma-like histological features with lymphoplasmacellular infiltration. This tumor is often seen in children, but not in adults, with a systemic inflammatory syndrome (iron-resistant microcytic anemia and/or dysgammaglobulinemia) and very rarely with a persistent moderate hyperthermia. In the present report the authors describe a temporal chordoid meningioma in a 30-year-old woman who presented with fever, headache, and a serological inflammatory syndrome. The clinical symptomatology, chiefly the fever, disappeared immediately after removal of the tumor. To the authors' knowledge, only one similar patient with such clinical presentation and response to surgery has been mentioned in the literature. Interestingly, at immunohistochemical examination, the neoplasm showed focal positivity for the pyrogenic cytokine interleukin-6. The capacity of the tumor to produce this pyrogenic cytokine could explain both the patient's clinical presentation and her response to the surgical management.

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    ABSTRACT: Chordoid meningioma (CM) is a meningioma containing regions that are histologically similar to chordoma, with trabeculae of eosinophilic, vacuolated cells in a myxoid background and corresponds to WHO grade II [1]. Such a tumor is a rare variant first described by Kepes et al. in 1988 in young patients associated with Castleman syndrome [2]. Only a series [3] and a few case reports [4-18, 24-30] were published. To the best of our knowledge, about 80 cases have been previously published, but unpublished cases are often recorded. Ultrastructural features of these tumors are studied in only few reports and are not yet precisely ascertained. We present a new case of a relapsing chordoid meningioma with aggressive behavior and acute clinical presentation and a "chordoid progression'' of the tumor characterized by histological and ultrastructural modification.
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