Case report: Drug rash with eosinophilia and systemic symptoms syndrome in a patient with anti–interferon-γ autoantibody–associated immunodeficiency
A 56-year-old Chinese woman with previous disseminated mycobacterium avium complex infection and recurrent cervical abscesses from Burkholderia cepacia complex visited our hospital. She was diagnosed with adult-onset immunodeficiency (AOID) and tested positive for interferon-γ–neutralizing autoantib...
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| Published in | Frontiers in immunology Vol. 13; p. 969912 |
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| Main Authors | , , , , , , , , , |
| Format | Journal Article |
| Language | English |
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22.08.2022
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| Abstract | A 56-year-old Chinese woman with previous disseminated mycobacterium avium complex infection and recurrent cervical abscesses from
Burkholderia cepacia complex
visited our hospital. She was diagnosed with adult-onset immunodeficiency (AOID) and tested positive for interferon-γ–neutralizing autoantibody. Ceftazidime was administered as the initial antimicrobial treatment, which was later combined with sulfamethoxazole-trimethoprim (SMZ-TMP). She developed drug rash with eosinophilia and systemic symptoms (DRESS) syndrome after SMZ-TMP administration and improved after withdrawal of the culprit antibiotic and systemic glucocorticoids treatment. Her cervical infection was eventually cured after combined therapy of long-term antibiotics and anti–IFN-γ autoantibodies (AIGA) titer-lowering treatments including glucocorticoids, rituximab, and plasmapheresis. This is the first case of DRESS syndrome in the setting of AIGA-induced AOID and is worthy of notice. |
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| AbstractList | A 56-year-old Chinese woman with previous disseminated mycobacterium avium complex infection and recurrent cervical abscesses from Burkholderia cepacia complex visited our hospital. She was diagnosed with adult-onset immunodeficiency (AOID) and tested positive for interferon-γ-neutralizing autoantibody. Ceftazidime was administered as the initial antimicrobial treatment, which was later combined with sulfamethoxazole-trimethoprim (SMZ-TMP). She developed drug rash with eosinophilia and systemic symptoms (DRESS) syndrome after SMZ-TMP administration and improved after withdrawal of the culprit antibiotic and systemic glucocorticoids treatment. Her cervical infection was eventually cured after combined therapy of long-term antibiotics and anti-IFN-γ autoantibodies (AIGA) titer-lowering treatments including glucocorticoids, rituximab, and plasmapheresis. This is the first case of DRESS syndrome in the setting of AIGA-induced AOID and is worthy of notice.A 56-year-old Chinese woman with previous disseminated mycobacterium avium complex infection and recurrent cervical abscesses from Burkholderia cepacia complex visited our hospital. She was diagnosed with adult-onset immunodeficiency (AOID) and tested positive for interferon-γ-neutralizing autoantibody. Ceftazidime was administered as the initial antimicrobial treatment, which was later combined with sulfamethoxazole-trimethoprim (SMZ-TMP). She developed drug rash with eosinophilia and systemic symptoms (DRESS) syndrome after SMZ-TMP administration and improved after withdrawal of the culprit antibiotic and systemic glucocorticoids treatment. Her cervical infection was eventually cured after combined therapy of long-term antibiotics and anti-IFN-γ autoantibodies (AIGA) titer-lowering treatments including glucocorticoids, rituximab, and plasmapheresis. This is the first case of DRESS syndrome in the setting of AIGA-induced AOID and is worthy of notice. A 56-year-old Chinese woman with previous disseminated mycobacterium avium complex infection and recurrent cervical abscesses from Burkholderia cepacia complex visited our hospital. She was diagnosed with adult-onset immunodeficiency (AOID) and tested positive for interferon-γ–neutralizing autoantibody. Ceftazidime was administered as the initial antimicrobial treatment, which was later combined with sulfamethoxazole-trimethoprim (SMZ-TMP). She developed drug rash with eosinophilia and systemic symptoms (DRESS) syndrome after SMZ-TMP administration and improved after withdrawal of the culprit antibiotic and systemic glucocorticoids treatment. Her cervical infection was eventually cured after combined therapy of long-term antibiotics and anti–IFN-γ autoantibodies (AIGA) titer-lowering treatments including glucocorticoids, rituximab, and plasmapheresis. This is the first case of DRESS syndrome in the setting of AIGA-induced AOID and is worthy of notice. A 56-year-old Chinese woman with previous disseminated mycobacterium avium complex infection and recurrent cervical abscesses from Burkholderia cepacia complex visited our hospital. She was diagnosed with adult-onset immunodeficiency (AOID) and tested positive for interferon-γ–neutralizing autoantibody. Ceftazidime was administered as the initial antimicrobial treatment, which was later combined with sulfamethoxazole-trimethoprim (SMZ-TMP). She developed drug rash with eosinophilia and systemic symptoms (DRESS) syndrome after SMZ-TMP administration and improved after withdrawal of the culprit antibiotic and systemic glucocorticoids treatment. Her cervical infection was eventually cured after combined therapy of long-term antibiotics and anti–IFN-γ autoantibodies (AIGA) titer-lowering treatments including glucocorticoids, rituximab, and plasmapheresis. This is the first case of DRESS syndrome in the setting of AIGA-induced AOID and is worthy of notice. |
| Author | Zhang, Ting Wang, Jinglan Dong, Xiying Fan, Junping Zhang, Linqi Zhang, Ying Nie, Yuxue Ran, Jun Wang, Han Pan, Siqi |
| AuthorAffiliation | 2 Comprehensive AIDS Research Center, Center for Infectious Diseases Research, Beijing Advanced Innovation Center for Structural Biology, School of Medicine, Tsinghua University , Beijing , China 5 Department of International Medical Service, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences (CAMS) and Peking Union Medical College (PUMC) , Beijing , China 1 Department of Pulmonary and Critical Care Medicine, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences (CAMS) and Peking Union Medical College (PUMC) , Beijing , China 3 School of Clinical Medicine, Chinese Academy of Medical Sciences (CAMS) and Peking Union Medical College (PUMC) , Beijing , China 4 Heart Failure Center, Fuwai Hospital, National Center for Cardiovascular Diseases, Chinese Academy of Medical Sciences (CAMS) and Peking Union Medical College (PUMC) , Beijing , China |
| AuthorAffiliation_xml | – name: 3 School of Clinical Medicine, Chinese Academy of Medical Sciences (CAMS) and Peking Union Medical College (PUMC) , Beijing , China – name: 5 Department of International Medical Service, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences (CAMS) and Peking Union Medical College (PUMC) , Beijing , China – name: 1 Department of Pulmonary and Critical Care Medicine, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences (CAMS) and Peking Union Medical College (PUMC) , Beijing , China – name: 2 Comprehensive AIDS Research Center, Center for Infectious Diseases Research, Beijing Advanced Innovation Center for Structural Biology, School of Medicine, Tsinghua University , Beijing , China – name: 4 Heart Failure Center, Fuwai Hospital, National Center for Cardiovascular Diseases, Chinese Academy of Medical Sciences (CAMS) and Peking Union Medical College (PUMC) , Beijing , China |
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| Cites_doi | 10.1007/s00228-020-03005-9 10.1097/FPC.0000000000000153 10.1182/blood-2003-04-1065 10.1016/j.alit.2019.03.006 10.1016/j.jaci.2015.09.018 10.1016/j.jaci.2020.08.003 10.1016/j.jclinepi.2017.04.026 10.1111/bjd.12501 10.3390/ijms22042147 10.1007/s10875-015-0161-5 10.1016/j.amjmed.2011.01.017 |
| ContentType | Journal Article |
| Copyright | Copyright © 2022 Nie, Wang, Dong, Pan, Zhang, Ran, Zhang, Fan, Zhang and Wang. Copyright © 2022 Nie, Wang, Dong, Pan, Zhang, Ran, Zhang, Fan, Zhang and Wang 2022 Nie, Wang, Dong, Pan, Zhang, Ran, Zhang, Fan, Zhang and Wang |
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| Notes | ObjectType-Case Study-2 SourceType-Scholarly Journals-1 ObjectType-Feature-4 content type line 23 ObjectType-Report-1 ObjectType-Article-3 Edited by: Chi Chiu Mok, Tuen Mun Hospital, Hong Kong SAR, China This article was submitted to Autoimmune and Autoinflammatory Disorders, a section of the journal Frontiers in Immunology Reviewed by: Hugo Chapdelaine, Montreal Clinical Research Institute (IRCM), Canada; David Andrew Fulcher, Australian National University, Australia |
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| References | Höflich (B1) 2004; 103 Wang (B10) 2021; 147 Sharifzadeh (B7) 2021; 77 Riley (B5) 2017; 89 Cacoub (B6) 2011; 124 Hanitsch (B2) 2015; 35 Kongpan (B11) 2015; 25 Shiohara (B3) 2019; 68 Kardaun (B4) 2013; 169 Ku (B9) 2016; 137 Miyagawa (B8) 2021; 22 |
| References_xml | – volume: 77 year: 2021 ident: B7 article-title: Antibacterial antibiotic-induced drug reaction with eosinophilia and systemic symptoms (DRESS) syndrome: a literature review publication-title: Eur J Clin Pharmacol doi: 10.1007/s00228-020-03005-9 – volume: 25 year: 2015 ident: B11 article-title: Candidate HLA genes for prediction of co-trimoxazole-induced severe cutaneous reactions publication-title: Pharmacogenet Genomics doi: 10.1097/FPC.0000000000000153 – volume: 103 year: 2004 ident: B1 article-title: Naturally occurring anti-IFN-gamma autoantibody and severe infections with mycobacterium cheloneae and burkholderia cocovenenans publication-title: Blood doi: 10.1182/blood-2003-04-1065 – volume: 68 year: 2019 ident: B3 article-title: Drug-induced hypersensitivity syndrome (DiHS)/drug reaction with eosinophilia and systemic symptoms (DRESS): An update in 2019 publication-title: Allergol Int doi: 10.1016/j.alit.2019.03.006 – volume: 137 start-page: 945 year: 2016 ident: B9 article-title: Anti–IFN-γ autoantibodies are strongly associated with HLA-DR*15:02/16:02 and HLA-DQ*05:01/05:02 across southeast Asia publication-title: J Allergy Clin Immunol doi: 10.1016/j.jaci.2015.09.018 – volume: 147 year: 2021 ident: B10 article-title: Whole genome sequencing identifies genetic variants associated with co-trimoxazole hypersensitivity in asians publication-title: J Allergy Clin Immunol doi: 10.1016/j.jaci.2020.08.003 – volume: 89 year: 2017 ident: B5 article-title: CARE guidelines for case reports: explanation and elaboration document publication-title: J Clin Epidemiol doi: 10.1016/j.jclinepi.2017.04.026 – volume: 169 year: 2013 ident: B4 article-title: Drug reaction with eosinophilia and systemic symptoms (DRESS): an original multisystem adverse drug reaction. results from the prospective RegiSCAR study publication-title: Br J Dermatol doi: 10.1111/bjd.12501 – volume: 22 year: 2021 ident: B8 article-title: Current perspective regarding the immunopathogenesis of drug-induced hypersensitivity syndrome/drug reaction with eosinophilia and systemic symptoms (dihs/dress) publication-title: Int J Mol Sci doi: 10.3390/ijms22042147 – volume: 35 year: 2015 ident: B2 article-title: Late-onset disseminated mycobacterium avium intracellulare complex infection (mac), cerebral toxoplasmosis and salmonella sepsis in a german caucasian patient with unusual anti-interferon-gamma igg1 autoantibodies publication-title: J Clin Immunol doi: 10.1007/s10875-015-0161-5 – volume: 124 year: 2011 ident: B6 article-title: The DRESS syndrome: a literature review publication-title: Am J Med doi: 10.1016/j.amjmed.2011.01.017 |
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| SubjectTerms | adult-onset immunodeficiency due to anti-interferon-gamma antibodies anti-IFN-γ autoantibodies Burkholderia drug rash with eosinophilia and systemic symptoms Immunology infection sulfamethoxazole |
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| Title | Case report: Drug rash with eosinophilia and systemic symptoms syndrome in a patient with anti–interferon-γ autoantibody–associated immunodeficiency |
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