Literature DB >> 21166862

Fifteen years of treatment with intravenous immunoglobulin in central nervous system Langerhans cell histiocytosis.

Désirée Gavhed1, Evaldas Laurencikas, Selma O Akefeldt, Jan-Inge Henter.   

Abstract

AIM: There is currently no well-accepted therapy for central nervous system Langerhans cell histiocytosis (CNS-LCH), a neuroinflammatory disease clinically characterized by often progressive, neurological symptoms including ataxia, dysarthria, dysphagia, hypertonicity, intellectual impairment and behavioural abnormalities. We applied immunomodulative/anti-inflammatory treatment on a patient with progressive CNS-LCH disease.
METHOD: Intravenous immunoglobulin (IVIG) was administered monthly for 15 years to a patient with severe, image-verified neurodegenerative CNS-LCH.
RESULTS: During the IVIG treatment, the neurological deterioration initially appeared to be haltered, but over time there was still some deterioration.
CONCLUSIONS: IVIG may be beneficial in partly haltering CNS-LCH neurodegeneration, but further studies are needed.
© 2011 The Author(s)/Acta Paediatrica © 2011 Foundation Acta Paediatrica.

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Year:  2011        PMID: 21166862     DOI: 10.1111/j.1651-2227.2010.02125.x

Source DB:  PubMed          Journal:  Acta Paediatr        ISSN: 0803-5253            Impact factor:   2.299


  5 in total

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

Authors:  Shinsaku Imashuku; Naoto Fujita; Yoko Shioda; Haruyoshi Noma; Shiro Seto; Toshinori Minato; Kazuo Sakashita; Nobuhiro Ito; Ryoji Kobayashi; Akira Morimoto
Journal:  Int J Hematol       Date:  2014-12-10       Impact factor: 2.490

Review 2.  Langerhans cell histiocytosis of the orbit: five clinicopathologic cases and review of the literature.

Authors:  Martina C Herwig; Ted Wojno; Qing Zhang; Hans E Grossniklaus
Journal:  Surv Ophthalmol       Date:  2012-12-13       Impact factor: 6.048

3.  Langerhans cell histiocytosis in an adult with involvement of the calvarium, cerebral cortex and brainstem: discussion of pathophysiology and rationale for the use of intravenous immune globulin.

Authors:  Christopher Dardis; Thandar Aung; William Shapiro; John Fortune; Stephen Coons
Journal:  Case Rep Neurol       Date:  2015-02-23

4.  Langerhans cell histiocytosis (LCH): guidelines for diagnosis, clinical work-up, and treatment for patients till the age of 18 years.

Authors:  Riccardo Haupt; Milen Minkov; Itziar Astigarraga; Eva Schäfer; Vasanta Nanduri; Rima Jubran; R Maarten Egeler; Gritta Janka; Dragan Micic; Carlos Rodriguez-Galindo; Stefaan Van Gool; Johannes Visser; Sheila Weitzman; Jean Donadieu
Journal:  Pediatr Blood Cancer       Date:  2012-10-25       Impact factor: 3.167

5.  Histiocytic lesions of the orbit: A study of 9 cases.

Authors:  A Kaan Gündüz; Emine Temel
Journal:  Saudi J Ophthalmol       Date:  2018-03-15
  5 in total

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