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Brain abnormalities in Sjogren syndrome with recurrent CNS manifestations: association with neuromyelitis optica

Authors
Min, J. H.Kim, H. J.Kim, B. J.Lee, K. W.Sunwoo, I. N.Kim, S. M.Kim, B. J.Kim, S. H.Park, M. S.Waters, P.Vincent, A.Sung, J. J.Lee, K. H.
Issue Date
9월-2009
Publisher
SAGE PUBLICATIONS LTD
Keywords
aquaporin 4; brain MRI; multiple sclerosis; neuromyelitis optica; Sjogren' s syndrome; vasogenic brain edema
Citation
MULTIPLE SCLEROSIS JOURNAL, v.15, no.9, pp.1069 - 1076
Indexed
SCIE
SCOPUS
Journal Title
MULTIPLE SCLEROSIS JOURNAL
Volume
15
Number
9
Start Page
1069
End Page
1076
URI
https://scholar.korea.ac.kr/handle/2021.sw.korea/119442
DOI
10.1177/1352458509106228
ISSN
1352-4585
Abstract
Background and objectives Optic neuritis or longitudinally extensive myelitis in Sjogren syndrome (SS) suggests a neuromyelitis optica spectrum disorder (NMOSD). However, brain abnormalities of SS remain to be elucidated for the association with neuromyelitis optica (NMO). Methods Twelve primary SS patients (all women, 42 +/- 13.2 years) who had recurrent central nervous system (CNS) manifestations with brain involvement were retrospectively identified. Brain MRI, and neurologic and serologic findings were analyzed with the measurement of anti-aquaporin-4 antibody (AQP4-Ab). Results All patients showed brain lesions characteristic of NMO as follows: 1) the involved sites adjacent to the third and fourth ventricles and in the posterior limb of the internal capsule, 2) unique configurations, such as the longitudinal course from the internal capsule to the midbrain, large cerebral or cerebellar lesions over 3 cm, and cavity-like formations. AQP4-Ab was positive in six of eight patients tested, and all the seropositive patients showed lesions with increased diffusion, suggestive of vasogenic edema. Four patients met the revised criteria of NMO, and nine had features of NMOSDs. Of the remaining three patients showing only brain involvement, one had AQP4-Ab. Conclusions This study demonstrates that SS patients with recurrent CNS involvement have brain abnormalities characteristic of NMO and AQP4-Ab in Korea. The presence of AQP4-Ab in one SS patient with only brain involvement may suggest that the coexistence of NMO should be explored in SS patients with recurrent CNS manifestations, even without optic neuritis or myelitis. Multiple Sclerosis 2009; 15: 1069-1076. http://msj.sagepub.com
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