Progression of X-linked adrenoleukodystrophy under interferon-β therapy View Full Text


Ontology type: schema:ScholarlyArticle     


Article Info

DATE

1997-03

AUTHORS

G. C. Korenke, H.-J. Christen, B. Kruse, D. H. Hunneman, F. Hanefeld

ABSTRACT

The cerebral phenotype of X-linked adrenoleukodystrophy (ALD) is a rapidly progressive neurodegenerative disorder characterized by a cerebral inflammatory response and elevated very long-chain fatty acids (VLCFA). Interferon-beta (INFB) is known to suppress the synthesis of tumour necrosis factor alpha and interferon-gamma, which have been reported to be elevated in the margin of the areas of demyelination in ALD brains. We report on treatment with interferon-beta in 8 patients with cerebral ALD, who additionally received glycerol trioleate/glycerol trierucate. INFB-1 a (Rebif, Serono, Switzerland) was given subcutaneously once a week, 3 million units for the first 3 months and 6 million units for the next 9 months. All patients showed an unimpeded progression of neurological symptoms during INFB therapy. Therapy was stopped within 6 months in 4 patients because of the fast neurological deterioration with loss of walking. In all patients the MRI demonstrated a progression of demyelination with a qualitatively unchanged gadolinium enhancement. Further studies are needed to elucidate the pathomechanism of demyelination in ALD in order to find an effective therapy for cerebral ALD patients. More... »

PAGES

59-66

Identifiers

URI

http://scigraph.springernature.com/pub.10.1023/a:1005361607523

DOI

http://dx.doi.org/10.1023/a:1005361607523

DIMENSIONS

https://app.dimensions.ai/details/publication/pub.1050454301

PUBMED

https://www.ncbi.nlm.nih.gov/pubmed/9061569


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