Adult-onset asthma and periocular xanthogranulomas associated with systemic IgG4-related disease

PURPOSE: The aim of this study was to report a case of Adult-Onset Asthma with Periocular Xanthogranulomas (AAPOX) associated with systemic IgG4-related disease (IgG4-RD). OBSERVATIONS: A 57-year-old man presented with bilateral periorbital swelling for 1 year. Histopathology of a left orbital biops...

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Autores principales: Burris, Christopher K.H., Rodriguez, Maria E., Raven, Meisha L., Burkat, Cat N., Albert, Daniel M.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Elsevier 2016
Materias:
Article
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4852489/
https://www.ncbi.nlm.nih.gov/pubmed/27152364
http://dx.doi.org/10.1016/j.ajoc.2016.03.006
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author Burris, Christopher K.H.
Rodriguez, Maria E.
Raven, Meisha L.
Burkat, Cat N.
Albert, Daniel M.
author_facet Burris, Christopher K.H.
Rodriguez, Maria E.
Raven, Meisha L.
Burkat, Cat N.
Albert, Daniel M.
author_sort Burris, Christopher K.H.
collection PubMed
description PURPOSE: The aim of this study was to report a case of Adult-Onset Asthma with Periocular Xanthogranulomas (AAPOX) associated with systemic IgG4-related disease (IgG4-RD). OBSERVATIONS: A 57-year-old man presented with bilateral periorbital swelling for 1 year. Histopathology of a left orbital biopsy showed fibro vascular connective tissue inundated with foamy, lipid-laden histiocytes and touton giant cells with lymphocytic inflammation. Additional stains revealed CD68 positivity, and S100 negativity. The IgG and IgG4 stained slides showed increased IgG4 positive plasma cells but did not meet the criteria for IgG4-related orbital disease. His IgG4 serology was elevated, and IgG4 staining of his tissue previously diagnosed as autoimmune sclerosing pancreatitis was found to meet the criteria for IgG4-RD. CONCLUSION AND IMPORTANCE: AAPOX can be associated with systemic IgG4-RD.
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spelling pubmed-48524892017-04-01 Adult-onset asthma and periocular xanthogranulomas associated with systemic IgG4-related disease Burris, Christopher K.H. Rodriguez, Maria E. Raven, Meisha L. Burkat, Cat N. Albert, Daniel M. Am J Ophthalmol Case Rep Article PURPOSE: The aim of this study was to report a case of Adult-Onset Asthma with Periocular Xanthogranulomas (AAPOX) associated with systemic IgG4-related disease (IgG4-RD). OBSERVATIONS: A 57-year-old man presented with bilateral periorbital swelling for 1 year. Histopathology of a left orbital biopsy showed fibro vascular connective tissue inundated with foamy, lipid-laden histiocytes and touton giant cells with lymphocytic inflammation. Additional stains revealed CD68 positivity, and S100 negativity. The IgG and IgG4 stained slides showed increased IgG4 positive plasma cells but did not meet the criteria for IgG4-related orbital disease. His IgG4 serology was elevated, and IgG4 staining of his tissue previously diagnosed as autoimmune sclerosing pancreatitis was found to meet the criteria for IgG4-RD. CONCLUSION AND IMPORTANCE: AAPOX can be associated with systemic IgG4-RD. Elsevier 2016-03-29 /pmc/articles/PMC4852489/ /pubmed/27152364 http://dx.doi.org/10.1016/j.ajoc.2016.03.006 Text en © 2016 The Authors http://creativecommons.org/licenses/by-nc-nd/4.0/ This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
spellingShingle Article
Burris, Christopher K.H.
Rodriguez, Maria E.
Raven, Meisha L.
Burkat, Cat N.
Albert, Daniel M.
Adult-onset asthma and periocular xanthogranulomas associated with systemic IgG4-related disease
title Adult-onset asthma and periocular xanthogranulomas associated with systemic IgG4-related disease
title_full Adult-onset asthma and periocular xanthogranulomas associated with systemic IgG4-related disease
title_fullStr Adult-onset asthma and periocular xanthogranulomas associated with systemic IgG4-related disease
title_full_unstemmed Adult-onset asthma and periocular xanthogranulomas associated with systemic IgG4-related disease
title_short Adult-onset asthma and periocular xanthogranulomas associated with systemic IgG4-related disease
title_sort adult-onset asthma and periocular xanthogranulomas associated with systemic igg4-related disease
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4852489/
https://www.ncbi.nlm.nih.gov/pubmed/27152364
http://dx.doi.org/10.1016/j.ajoc.2016.03.006
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