Steroid-responsive Devic’s variant in Sjögren’s syndrome

Mochizuki, Akihide; Hayashi, Akito; Hisahara, Shin; Shoji, Shin’ichi

doi:10.1212/wnl.54.6.1391

Cited by 43 publications

(21 citation statements)

References 6 publications

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“…Patient 2 presented with chorea, a previously reported atypical SS-related CNS manifestation [11]. Patient 3 had optic neuritis and recurrent acute transverse myelitis, a clinical presentation similar to neuromyelitis optica already described in SS-related CNS disease [12]. In all 3 patients, extensive testing did not reveal any alternate diagnosis for the CNS manifestations.…”

Section: Discussionmentioning

confidence: 75%

Cerebrospinal Fluid Anti-SSA Autoantibodies in Primary Sjögren’s Syndrome with Central Nervous System Involvement

Mégevand

Chizzolini²,

Chofflon³

et al. 2007

Eur Neurol

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Background: Central nervous system involvement in primary Sjögren’s syndrome is a matter of controversy, and its diagnosis remains difficult. Methods: We report 3 patients with primary Sjögren’s syndrome and central nervous system involvement in whom we assessed intrathecal immunoglobulin G synthesis and the presence of cerebrospinal fluid anti-SSA and anti-SSB autoantibodies. Results: We found intrathecal immunoglobulin G synthesis and presence of cerebrospinal fluid anti-SSA autoantibodies in all patients, with demonstration for the first time of specific anti-SSA autoantibody intrathecal synthesis in 2 patients. Conclusion: We suggest that cerebrospinal fluid anti-SSA autoantibodies could serve as a biomarker for Sjögren’s-syndrome-related central nervous system involvement.

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Section: Discussionmentioning

confidence: 75%

Cerebrospinal Fluid Anti-SSA Autoantibodies in Primary Sjögren’s Syndrome with Central Nervous System Involvement

Mégevand

Chizzolini²,

Chofflon³

et al. 2007

Eur Neurol

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“…Dans la plupart des cas le SGS se manifestait uniquement par une atteinte glandulaire. Les autres manifestations systémiques associées étaient un purpura hyperglobulinémique dans un cas [7] et une myélopathie régressive sous corticothé-rapie dans un deuxième cas [13].…”

Section: Discussionunclassified

Une névrite optique bilatérale : manifestation inaugurale d'un syndrome de Gougerot-Sjögren primitif

Essaadouni¹,

Louhab²,

Belaabidia

et al. 2006

Revue du Rhumatisme

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“…Our search disclosed 17 previously published articles which are individual case reports or small series of patients with the association of SS and NMOSD 9,10,11,12,13,14,15,16,17,18,19,20,21,22,23,24 . Adding our two patients, a total of 49 patients could be found harboring the association of both diseases according to current accepted diagnostic criteria (Table 3) 3, 4,8 .…”

Section: Casementioning

confidence: 99%

Sjögren syndrome and neuromyelitis optica spectrum disorder co-exist in a common autoimmune milieu

Carvalho

Tironi

Freitas

et al. 2014

Arq. Neuro-Psiquiatr.

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The relationship between Sjögren's syndrome (SS) and neuromyelitis optica spectrum disorder (NMOSD) is not completely understood. We report two patients with both conditions and review 47 other previously reported cases meeting currently accepted diagnostic criteria, from 17 articles extracted from PubMed. Out of 44 patients whose gender was informed, 42 were females. Mean age at onset of neurological manifestation was 36.2 years (10-74). Serum anti-AQP4-IgG was positive in 32 patients, borderline in 1, and negative in 4. Our Case 1 was seronegative for AQP4-IgG and had no non-organ-specific autoantibodies other than anti-SSB antibodies. Our Case 2 had serum anti-AQP4, anti-SSA/SSB, anti-thyreoglobulin and anti-acethylcholine-receptor antibodies, as well as clinical hypothyreoidism, but no evidence of myasthenia gravis. Our Cases and others, as previously reported in literature, with similar heterogeneous autoimmune response to aquaporin-4, suggest that SS and NMO co-exist in a common autoimmune milieu which is not dependent on aquaporin-4 autoimmunity.Keywords: neuromyelitis optica spectrum disorders, neuromyelitis optica, Sjögren's syndrome, autoimmunity, AQP4-IgG. RESUMOA relação entre síndrome de Sjögren (SS) e espectro da neuromielite óptica (ENMO) ainda não é bem compreendida. Relatamos dois pacientes com ambas as condições e revisamos 47 casos que preenchem critérios diagnósticos das duas doenças, descritos em 17 artigos extraídos da PubMed. Dos 44 pacientes cujo gênero foi informado 42 eram mulheres. A idade média ao início das manifestações neurológicas foi 36,2 anos (10-74). O anticorpo anti-AQP4 foi positivo em 32 dos 37 pacientes, em 1 foi "borderline". Nosso Caso 1 era soronegativo para AQP4-IgG, não tinha autoanticorpos não-órgão específicos, exceto anti-SSB. O Caso 2 era soropositivo para anticorpos anti-AQP4, anti-SSA/SSB, anti-tireoglobulina, e anti-receptor da acetilcolina; apresentava hipotireoidismo, mas não havia evidêncas de miastenia gravis. Nossos casos e outros similares, previamente relatados na literatura, com resposta autoimune heterogênea à aquaporina-4 sugerem que a SS e o ENMO coexistem em meio de autoimunidade não dependente da aquaporina-4.Palavras-chave: espectro de neuromielite óptica, neuromielite óptica, síndrome de Sjögren, autoimunidade, AQP4-IgG.

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Steroid-responsive Devic’s variant in Sjögren’s syndrome

Cited by 43 publications

References 6 publications

Cerebrospinal Fluid Anti-SSA Autoantibodies in Primary Sjögren’s Syndrome with Central Nervous System Involvement

Cerebrospinal Fluid Anti-SSA Autoantibodies in Primary Sjögren’s Syndrome with Central Nervous System Involvement

Une névrite optique bilatérale : manifestation inaugurale d'un syndrome de Gougerot-Sjögren primitif

Sjögren syndrome and neuromyelitis optica spectrum disorder co-exist in a common autoimmune milieu

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