Literature DB >> 25043664

Motor neuronopathy in Chediak-Higashi syndrome.

S Mathis1, P Cintas2, G de Saint-Basile3, L Magy4, B Funalot4, J-M Vallat5.   

Abstract

Chediak-Higashi syndrome is a rare autosomal recessive disease characterized by partial oculocutaneous albinism, recurrent pyogenic infections and the presence of giant granules in many cells such as leucocytes (hallmark of the disease). Neurological symptoms are rare. We describe two sisters who presented the same phenotype of slowly progressive motor neuronopathy (with Babinski sign in one patient); biopsy of the sural nerve showed an abnormal endoneurial accumulation of lipofuscin granules. We discuss these two observations and compare them with the few case reports of neuropathy in Chediak-Higashi syndrome.
Copyright © 2014 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  Chediak–Higashi syndrome; LYST; Lipofuscin; Motor neuron disorder; Motor neuropathy; Schwann cell

Mesh:

Year:  2014        PMID: 25043664     DOI: 10.1016/j.jns.2014.06.026

Source DB:  PubMed          Journal:  J Neurol Sci        ISSN: 0022-510X            Impact factor:   3.181


  3 in total

1.  Parkinsonism and Other Movement Disorders Associated with Chediak-Higashi Syndrome: Case Report and Systematic Literature Review.

Authors:  Bettina Balint; Kailash P Bhatia
Journal:  Mov Disord Clin Pract       Date:  2015-01-14

2.  The lysosomal trafficking regulator is necessary for normal wound healing.

Authors:  Jacob C Zbinden; Gabriel J M Mirhaidari; Kevin M Blum; Andrew J Musgrave; James W Reinhardt; Christopher K Breuer; Jenny C Barker
Journal:  Wound Repair Regen       Date:  2021-11-27       Impact factor: 3.617

Review 3.  Inherited Paediatric Motor Neuron Disorders: Beyond Spinal Muscular Atrophy.

Authors:  Hooi Ling Teoh; Kate Carey; Hugo Sampaio; David Mowat; Tony Roscioli; Michelle Farrar
Journal:  Neural Plast       Date:  2017-05-28       Impact factor: 3.599

  3 in total

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