Chorea-acanthocytosis with polyclonal antibodies to ganglioside GM1

Hirayama, Masaaki; Hamano, Tadanori; Shiratori, Masayuki; Mutoh, Tatsuro; Kumano, Takanori; Aita, Takashi; Kuriyama, Masaru

doi:10.1016/s0022-510x(97)00072-5

Cited by 8 publications

(3 citation statements)

References 9 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…Some reports have suggested a demyelinating process, but these changes may have been secondary to axonal degeneration [2,41,58]. One case reported had a large fibre axonal neuropathy and antibodies to ganglioside GM1, although it appears likely that this was an inflammatory response secondary to the axonal degeneration [25].…”

Section: S Haematologymentioning

confidence: 99%

Acanthocytosis and neurological disorders

Stevenson

Hardie

2001

Journal of Neurology

View full text Add to dashboard Cite

Acanthocytosis occurs because of ultrastructural abnormalities of the erythrocyte membranous skeleton resulting in reduced membrane fluidity. At least three hereditary neurological conditions are associated with it, although as yet the pathogenesis of the neurological features is unknown. In abetalipoproteinaemia, an autosomal recessive condition, vitamin E deficiency results in a progressive spinocerebellar syndrome associated with peripheral neuropathy and retinitis pigmentosa. Neuroacanthocytosis is also probably an autosomal recessive condition and is characterised by chorea, orofaciolingual dyskinesia, dysarthria, areflexia, seizures and dementia. McLeod syndrome is an X-linked recessive disorder usually presenting in males as a benign myopathy with areflexia, in association with a particular abnormality of expression of Kell blood group antigens. However, occasionally the neurological features are more severe and indistinguishable from those of neuroacanthocytosis. Recent advances in molecular genetics may assist better understanding of the disease mechanisms and the search for more effective treatments.

show abstract

Section: S Haematologymentioning

confidence: 99%

Acanthocytosis and neurological disorders

Stevenson

Hardie

2001

Journal of Neurology

View full text Add to dashboard Cite

show abstract

“…Although some of these disorders have been described in a previous review (Yu and Ariga, 1998), and will not be repeated here, the following list cites some more recent notable examples of anti‐GSL‐associated neuropathies. Anti‐GM1 IgM antibodies have been suggested to cause chorea‐acanthocytosis associated with axonal neuropathy (Hirayama et al, 1997) and may be involved in the pathogenesis of chronic acquired demyelinating polyneuropathy (Bech et al, 1998). Antiasialo‐GM1 (GA1) and anti‐GM1 antibodies may be involved in the pathogenesis of peripheral neuropathy in Chagas disease (Avila et al, 1998; Mitelman et al, 1999) and systemic lupus erythematosus with CNS involvement (Gatterbauer et al, 1998).…”

Section: Peripheral Neuropathiesmentioning

confidence: 99%

“…Anti‐GM1 IgM antibodies have been suggested to cause chorea‐acanthocytosis associated with axonal neuropathy (Hirayama et al, 1997) and may be involved in the pathogenesis of chronic acquired demyelinating polyneuropathy (Bech et al, 1998).…”

Section: Peripheral Neuropathiesmentioning

confidence: 99%

Recent studies on the roles of antiglycosphingolipids in the pathogenesis of neurological disorders

Ariga¹,

Miyatake²,

Yu³

2001

J of Neuroscience Research

View full text Add to dashboard Cite

Evidence is mounting to suggest a causal role of humoral immunity arising from antiglycosphingolipid (GSL) antibodies in a variety of neurological disorders. These disorders include the demyelinating and axonal forms of Guillain-Barre syndrome, multifocal motor neuropathy, chronic inflammatory demyelinating polyradiculoneuropathy, and IgM paraproteinemia. Many claims have been made regarding other neurological disorders, which should be carefully scrutinized for their validity, based on several criteria proposed in this review. These criteria include 1) characterization of the causative antigens and immunoglobulins, 2) correlation of the pathological lesions and clinical manifestation of the antigens, 3) establishment of animal models using pure GSLs as the antigens, 4) immunopathogenic mechanisms of the neurodenerative process, 5) mechanisms for the malfunctioning of blood-nerve barrier and the ensuing leakage of circulating antibodies into peripheral nerve parenchyma, and 6) the roles of anti-GSL antibodies that may cause humorally mediated nerve dysfunction and injury as well as interference with ion channel function at the node of Ranvier, where carbohydrate epitopes are located. Finally, the origin of the anti-GSL antibodies is discussed in light of the recent circumstantial evidence pointing to a molecular mimicry mechanism with infectious agents. With a better understanding of the immunopathogenic mechanisms, it will then be possible to devise rational and effective diagnostic and therapeutic strategies for the treatment of these neurological disorders.

show abstract

Neuromuscular Findings in Eight Italian Families with Neuroacanthocytosis

Dotti

Malandrini

Federico

Neuroacanthocytosis Syndromes

View full text Add to dashboard Cite

Chorea-acanthocytosis with polyclonal antibodies to ganglioside GM1

Cited by 8 publications

References 9 publications

Acanthocytosis and neurological disorders

Acanthocytosis and neurological disorders

Recent studies on the roles of antiglycosphingolipids in the pathogenesis of neurological disorders

Neuromuscular Findings in Eight Italian Families with Neuroacanthocytosis

Contact Info

Product

Resources

About