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Family History of Autoimmune Disease in Patients with Aicardi-Goutières Syndrome

Purpose. The purpose of this study was to explore anecdotal evidence for an increase in the prevalence of autoimmune diseases in family members of patients with Aicardi-Goutières syndrome (AGS). Methods. Pedigrees of patients and controls were analyzed using chi-square and logistic regression to ass...

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Autores principales: Schmidt, Johanna L., Olivieri, Ivana, Vento, Jodie M., Fazzi, Elisa, Gordish-Dressman, Heather, Orcesi, Simona, Vanderver, Adeline
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Hindawi Publishing Corporation 2012
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3508752/
https://www.ncbi.nlm.nih.gov/pubmed/23251212
http://dx.doi.org/10.1155/2012/206730
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author Schmidt, Johanna L.
Olivieri, Ivana
Vento, Jodie M.
Fazzi, Elisa
Gordish-Dressman, Heather
Orcesi, Simona
Vanderver, Adeline
author_facet Schmidt, Johanna L.
Olivieri, Ivana
Vento, Jodie M.
Fazzi, Elisa
Gordish-Dressman, Heather
Orcesi, Simona
Vanderver, Adeline
author_sort Schmidt, Johanna L.
collection PubMed
description Purpose. The purpose of this study was to explore anecdotal evidence for an increase in the prevalence of autoimmune diseases in family members of patients with Aicardi-Goutières syndrome (AGS). Methods. Pedigrees of patients and controls were analyzed using chi-square and logistic regression to assess differences in reports of autoimmune disease among family members of cases and controls. Data was collected at Children's National Medical Center in Washington, DC, USA and at the International Aicardi-Goutières Syndrome Association Scientific Headquarters, C. Mondino National Institute of Neurology in Pavia, Italy. Results. The number of individuals with reported autoimmune disease is significantly related to having a family member with AGS (χ (2) = 6.25, P = 0.01); 10% (35/320) of relatives of patients with AGS had a reported autoimmune disease diagnosis compared to 5% (18/344) of relatives of controls. There was a greater percent of maternal relatives of patients with AGS reporting autoimmune disease (14.6%), compared to controls (6.8%), with the association being statistically significant. The association was not significant for paternal relatives. Conclusion. The prevalence of autoimmune disease in relatives of children with AGS is significantly increased compared to controls. More research is needed to better understand this association.
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spelling pubmed-35087522012-12-18 Family History of Autoimmune Disease in Patients with Aicardi-Goutières Syndrome Schmidt, Johanna L. Olivieri, Ivana Vento, Jodie M. Fazzi, Elisa Gordish-Dressman, Heather Orcesi, Simona Vanderver, Adeline Clin Dev Immunol Research Article Purpose. The purpose of this study was to explore anecdotal evidence for an increase in the prevalence of autoimmune diseases in family members of patients with Aicardi-Goutières syndrome (AGS). Methods. Pedigrees of patients and controls were analyzed using chi-square and logistic regression to assess differences in reports of autoimmune disease among family members of cases and controls. Data was collected at Children's National Medical Center in Washington, DC, USA and at the International Aicardi-Goutières Syndrome Association Scientific Headquarters, C. Mondino National Institute of Neurology in Pavia, Italy. Results. The number of individuals with reported autoimmune disease is significantly related to having a family member with AGS (χ (2) = 6.25, P = 0.01); 10% (35/320) of relatives of patients with AGS had a reported autoimmune disease diagnosis compared to 5% (18/344) of relatives of controls. There was a greater percent of maternal relatives of patients with AGS reporting autoimmune disease (14.6%), compared to controls (6.8%), with the association being statistically significant. The association was not significant for paternal relatives. Conclusion. The prevalence of autoimmune disease in relatives of children with AGS is significantly increased compared to controls. More research is needed to better understand this association. Hindawi Publishing Corporation 2012 2012-11-11 /pmc/articles/PMC3508752/ /pubmed/23251212 http://dx.doi.org/10.1155/2012/206730 Text en Copyright © 2012 Johanna L. Schmidt et al. https://creativecommons.org/licenses/by/3.0/ This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Research Article
Schmidt, Johanna L.
Olivieri, Ivana
Vento, Jodie M.
Fazzi, Elisa
Gordish-Dressman, Heather
Orcesi, Simona
Vanderver, Adeline
Family History of Autoimmune Disease in Patients with Aicardi-Goutières Syndrome
title Family History of Autoimmune Disease in Patients with Aicardi-Goutières Syndrome
title_full Family History of Autoimmune Disease in Patients with Aicardi-Goutières Syndrome
title_fullStr Family History of Autoimmune Disease in Patients with Aicardi-Goutières Syndrome
title_full_unstemmed Family History of Autoimmune Disease in Patients with Aicardi-Goutières Syndrome
title_short Family History of Autoimmune Disease in Patients with Aicardi-Goutières Syndrome
title_sort family history of autoimmune disease in patients with aicardi-goutières syndrome
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3508752/
https://www.ncbi.nlm.nih.gov/pubmed/23251212
http://dx.doi.org/10.1155/2012/206730
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