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Erschienen in: Immunologic Research 1/2017

03.09.2016 | Mechanism in Autoimmunity

Idiopathic hypertrophic pachymeningitis: an autoimmune IgG4-related disease

verfasst von: Armando De Virgilio, Marco de Vincentiis, Maurizio Inghilleri, Giovanni Fabrini, Michela Conte, Andrea Gallo, Maria Ida Rizzo, Antonio Greco

Erschienen in: Immunologic Research | Ausgabe 1/2017

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Abstract

Hypertrophic pachymeningitis (HP) is a rare disorder that causes thickening of the dura mater. Inflammatory lesions may be located in the cerebral or spinal dura mater or, less frequently, in both locations simultaneously. Numerous clinico-pathological entities cause thickening of the pachymeninges. Indeed, HP is a potential manifestation of many different diseases, but the diagnosis often remains uncertain. Cases in which the pachymeningitis has no known aetiology are termed “idiopathic” HP (IHP). Recently, it has been suggested that IgG4-related disease represents a subset of cases previously diagnosed as idiopathic hypertrophic pachymeningitis. Little is known regarding the pathogenic events of IHP. In a general theory, the inflammatory infiltrate, mainly consisting of B and T lymphocytes, activates fibroblasts and induces collagen deposition, leading to tissue hypertrophy and increased dural thickness. Clinical manifestations of IHP depend upon the location of the inflammatory lesions and compression of the adjacent nervous structures. Three central pathological features are lymphoplasmacytic infiltration, obliterative phlebitis, and storiform fibrosis. MRI is the examination of choice for the preliminary diagnosis of IHP. Histopathological examination of a biopsy specimen of the dura mater would finally confirm the diagnosis. The differential diagnosis for HP is broad and includes infections, autoimmune disorders, and neoplasia. Currently, there is no consensus about treatment for patients with IHP. There is a preference for glucocorticoid treatment on diagnosis followed by the addition of other immunosuppressive agents in the event of a recurrence. Rituximab is used in patients who did not respond to glucocorticoids or to conventional steroid-sparing agents.
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Metadaten
Titel
Idiopathic hypertrophic pachymeningitis: an autoimmune IgG4-related disease
verfasst von
Armando De Virgilio
Marco de Vincentiis
Maurizio Inghilleri
Giovanni Fabrini
Michela Conte
Andrea Gallo
Maria Ida Rizzo
Antonio Greco
Publikationsdatum
03.09.2016
Verlag
Springer US
Erschienen in
Immunologic Research / Ausgabe 1/2017
Print ISSN: 0257-277X
Elektronische ISSN: 1559-0755
DOI
https://doi.org/10.1007/s12026-016-8863-1

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