RESEARCH - Myelopathies secondary to Sjogren's syndrome: treatment with IV CYC associated with corticosteroids

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J Rheumatol. 2006 Apr;33(4):709-11.

Myelopathies secondary to Sjogren's syndrome: treatment with monthly

intravenous cyclophosphamide associated with corticosteroids.

de Seze J, Delalande S, Fauchais AL, Hachulla E, Stojkovic T, Ferriby D,

Hatron PY, Vermersch P.

Department of Neurology, Hopital R. Salengro, CHRU de Lille, Lille, France.

j-deseze@...

OBJECTIVE: Central nervous system manifestations in Sjogren's syndrome (SS)

include focal deficits, optic neuritis, and myelopathies. Acute and chronic

myelopathies are frequently severe and sometimes respond poorly to

corticosteroids. The efficacy of intravenous (IV) cyclophosphamide (CYC) has

been suggested in single case reports. METHODS: We describe the potential

usefulness of IV CYC in SS patients with severe myelopathies. Fourteen

patients [with acute (n = 6) and chronic (n = 8) myelopathies] were treated

with monthly CYC infusions (700 mg/m2) in addition to 500 mg of

corticosteroids for one year. We evaluated the disability before and after

CYC treatment using a walking distance calculation and the Expanded

Disability Status Scale (EDSS). RESULTS: CYC treatment was well tolerated in

all cases without serious adverse events. Nine patients (including the 6

with acute myelopathy) were improved after CYC treatment. Three patients

were stabilized and 2 patients with chronic myelopathies had moderate

progression of disability. The mean walking distance increased from 48.2 m

before to 180.4 m after CYC treatment (p < 0.02). Mean EDSS score decreased

from 6.6 to 5.7 (not significant). We found a correlation between the length

of time before CYC treatment and clinical improvement for both the walking

distance (p < 0.02) and the EDSS score (p < 0.05).

CONCLUSION: Although a randomized multicenter controlled study is warranted

to confirm our findings, IV CYC infusions seem to be useful for the

treatment of myelopathies secondary to SS, particularly in acute but also in

progressive cases. This treatment should be strongly considered as soon as

possible when disease progression is observed.

PMID: 16583474

http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?cmd=Retrieve & db=PubMed & list_uids=1\

6583474 & dopt=Abstract

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