Clinical characterization and successful treatment of 6 patients with Churg-Strauss syndrome-associated neuropathy.

Abstract:

OBJECTIVE:To confirm the reported findings and clarify unknown clinical features of Churg-Strauss syndrome (CSS)-associated neuropathy and design appropriate treatment. PATIENTS AND METHODS:We assessed the clinical features of 6 patients with CSS-associated neuropathy. RESULTS:Mononeuritis multiplex was present in 4 cases and polyneuropathy in the remaining cases. Both groups progressed to sensori-motor polyneuropathy in an acute or subacute course. All cases showed bronchial asthma and eosinophilia. Two cases with serum antineutrophil cytoplasmic antibodies to myeloperoxidase (MPO-ANCA) had an acute clinical course and severe symptoms. Nerve conduction studies (NCS) of these 2 cases revealed conduction blocks at the initial stage, although NCS finally indicated sensori-motor axonopathy at the involved extremities. For treatment, high-dose corticosteroid therapy for 4 cases, and cyclophosphamide combined with corticosteroids for 1 case, were effective. For the remaining case, intravenous immunoglobulin (IVIg) at the chronic phase resulted in a slow improvement of neuropathy in the symptomatic aspect. There was no relapse of neuropathy with low-dose corticosteroid treatment for 14-24 months after the initial treatment, except 1 case. There was also no relapse in the other case that was treated with moderate-dose steroids. CONCLUSION:Our study showed that CSS-associated neuropathy is a treatable disorder and that the first choice therapy is high-dose corticosteroid. In cases where corticosteroids are ineffective or for severe cases, immunosuppressive therapy (cyclophosphamide) with steroids should be considered, and IVIg might be a treatment option.

journal_name

Clin Neurol Neurosurg

authors

Nakamura M,Yabe I,Yaguchi H,Kishimoto R,Mito Y,Fujiki N,Houzen H,Tsuji-Akimoto S,Niino M,Sasaki H

doi

10.1016/j.clineuro.2009.07.004

subject

Has Abstract

pub_date

2009-10-01 00:00:00

pages

683-7

issue

8

eissn

0303-8467

issn

1872-6968

pii

S0303-8467(09)00172-3

journal_volume

111

pub_type

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