Increased Prevalence of Autoimmune Disorders and Autoantibodies in Parents of Children with Opsoclonus-Myoclonus Syndrome (OMS)

Abstract

Opsoclonus-myoclonus syndrome (OMS) is a rare neurological disease in childhood which can be associated with neuroblastoma. Since autoantibodies have been detected in some patients with OMS, an autoimmune etiology is suspected. We compared the prevalence of autoimmune disorders and autoantibodies in parents of children with OMS and in a group of controls of same age and sex. Autoimmune diseases were found in 15.8% of the parents of OMS children, but only in 2.0% of the controls (p<0.001) There was also an increased prevalence of autoantibodies in the OMS parents (42.8% vs. 8.0%, p<0.001). Thyroid diseases were the most frequent autoimmune diseases found, followed by inflammatory rheumatic diseases. Interestingly, the OMS parents also had significantly more autoantibodies against CNS structures than the controls (p<0.01).These findings support the autoimmune hypothesis of childhood OMS and may also hint to a genetic susceptibility for OMS.

opsoclonus-myoclonus syndrome - autoantibodies - autoimmunity - parents - antineuronal

References

• 1 Bataller L, Rosenfeld MR, Graus F, Vilchez JJ, Cheung NK, Dalmau J. Autoantigen diversity in the opsoclonus-myoclonus syndrome.  Ann Neurol. 2003;  53 347-353
• 2 Blaes F, Holzer G, Merkelbach S, Strittmatter M, Schimrigk K, Schäfer KH. Detection of autoantibodies by indirect immunofluorescence in the diagnostics of neurological diseases.  J Lab Med. 1998;  22 33-39
• 3 Blaes F, Fühlhuber V, Korfei M, Tschernatsch M, Behnisch W, Rostasy K. et al . Surface-binding autoantibodies to cerebellar neurons in opsoclonus syndrome.  Ann Neurol. 2005;  58 313-317
• 4 Cataldo F, Marino V. Increased prevalence of autoimmune diseases in first-degree relatives of patients with celiac disease.  J Pediatr Gastroenterol Nutr. 2003;  36 470-473
• 5 Corporaal S, Bijl M, Kallenberg CG. Familial occurrence of auto-immune diseases and autoantibodies in a caucasian population of patients with systemic lupus erythematosus.  Clin Rheumatol.. 2002;  21 108-113
• 6 Hanukoglu A, Mizrachi A, Dalal I, Admoni O, Rakover Y, Bistritzeer Z. et al . Extrapancreatic autoimmune manifestations in type 1 diabetes patients and their first-degree parents: a multicenter study.  Diabetes Care.. 2003;  26 1235-1240
• 7 Hermann R, Turpeinen H, Laine AP, Veijola R, Knip M, Simell O. et al . HLA DR-DQ-encoded genetic determinants of childhood-onset type 1 diabetes in Finland: an analysis of 622 nuclear families.  Tissue Antigens:. 2003;  62 162-169
• 8 Korfei M, Fühlhuber V, Schmidt-Wöll T, Kaps M, Preissner KT, Blaes F. Functional characterisation of autoantibodies from patients with pediatric opsoclonus. myoclonus syndrome.  J Neuroimmunol.. 2005;  170 150-157
• 9 Pranzatelli MR. The immunopharmacology of the opsoclonus-myoclonus syndrome.  Clin Neuropharmacol:. 1996;  19 1-47
• 10 Petaros P, Martelossi S, Tommasini A, Torre G, Caradonna M, Ventura A. Prevalence of Autoimmune disorders in parents of patients with celiac disease.  Dig Dis Sci. 2002;  47 1427-1431

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