Follow-up of pediatric patients treated by IVIG for Langerhans cell histiocytosis (LCH)-related neurodegenerative CNS disease View Full Text


Ontology type: schema:ScholarlyArticle     


Article Info

DATE

2015-02

AUTHORS

Shinsaku Imashuku, Naoto Fujita, Yoko Shioda, Haruyoshi Noma, Shiro Seto, Toshinori Minato, Kazuo Sakashita, Nobuhiro Ito, Ryoji Kobayashi, Akira Morimoto, Japan LCH Study Group (JLSG)

ABSTRACT

The follow-up of eight Japanese children with Langerhans cell histiocytosis (LCH)-related neurodegenerative central nervous system (ND-CNS) disease who were treated with intravenous immunoglobulin (IVIG) for >3 years is described. The patients developed ND-CNS disease at a median age of 5.2 (range 3.5-10.0) years and received IVIG treatment for a median duration of 6.5 + (range 3.7 to 10+) years. After a median follow-up period of 11.6 + (8.3+ to 13.9+) years after ND-CNS disease diagnosis, the median Expanded Disability Status Scale (EDSS) score of the eight patients was 4.0 (range 2.0-9.5). At the last follow-up as of March 2014, three patients have low EDSS scores (<3.0) and can walk without any assistance. Another three patients have EDSS scores of 3.5-4.5 and can walk by themselves, albeit occasionally with supports. However, the remaining two patients are wheelchair bound or bed ridden. The school performance of seven of the eight patients was below average. IVIG appeared to be most beneficial when it was administered soon after ND-CNS disease diagnosis when the EDSS scores were low (1.0-2.5). The patients who began receiving IVIG when their high EDSS scores were higher (4.5-7.0) appeared to obtain less benefit. To prevent progression of ND-CNS disease in patients with LCH, it is recommended to introduce IVIG early and to continue this therapy for >3 years. More... »

PAGES

191-197

References to SciGraph publications

Identifiers

URI

http://scigraph.springernature.com/pub.10.1007/s12185-014-1717-5

DOI

http://dx.doi.org/10.1007/s12185-014-1717-5

DIMENSIONS

https://app.dimensions.ai/details/publication/pub.1016626464

PUBMED

https://www.ncbi.nlm.nih.gov/pubmed/25491495


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