| Literature DB >> 25043664 |
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.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