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

Christopher K.H. Burris; Maria E. Rodriguez; Meisha L. Raven; Cat N. Burkat; Daniel M. Albert

doi:10.1016/j.ajoc.2016.03.006

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

Christopher K.H. Burris, Maria E. Rodriguez, Meisha L. Raven, Cat N. Burkat, Daniel M. Albert

Research output: Contribution to journal › Article › peer-review

15 Scopus citations

Abstract

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.

Original language	English (US)
Pages (from-to)	34-37
Number of pages	4
Journal	American Journal of Ophthalmology Case Reports
Volume	1
DOIs	https://doi.org/10.1016/j.ajoc.2016.03.006
State	Published - Apr 1 2016
Externally published	Yes

Keywords

Adult-onset asthma
IgG4-RD
Maxillary sinus
Orbital
Periocular xanthogranulomas
Xanthelasma

ASJC Scopus subject areas

Ophthalmology

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10.1016/j.ajoc.2016.03.006

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title = "Adult-onset asthma and periocular xanthogranulomas associated with systemic IgG4-related disease",

abstract = "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.",

keywords = "Adult-onset asthma, IgG4-RD, Maxillary sinus, Orbital, Periocular xanthogranulomas, Xanthelasma",

author = "Burris, {Christopher K.H.} and Rodriguez, {Maria E.} and Raven, {Meisha L.} and Burkat, {Cat N.} and Albert, {Daniel M.}",

note = "Publisher Copyright: {\textcopyright} 2016 The Authors",

year = "2016",

month = apr,

day = "1",

doi = "10.1016/j.ajoc.2016.03.006",

language = "English (US)",

volume = "1",

pages = "34--37",

journal = "American Journal of Ophthalmology Case Reports",

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T1 - Adult-onset asthma and periocular xanthogranulomas associated with systemic IgG4-related disease

AU - Burris, Christopher K.H.

AU - Rodriguez, Maria E.

AU - Raven, Meisha L.

AU - Burkat, Cat N.

AU - Albert, Daniel M.

PY - 2016/4/1

Y1 - 2016/4/1

N2 - 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.

AB - 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.

KW - Adult-onset asthma

KW - IgG4-RD

KW - Maxillary sinus

KW - Orbital

KW - Periocular xanthogranulomas

KW - Xanthelasma

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JO - American Journal of Ophthalmology Case Reports

JF - American Journal of Ophthalmology Case Reports

ER -

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

Abstract

Keywords

ASJC Scopus subject areas

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