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      Granulomatosis with polyangiitis: The trigger cannot be long hidden

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          Abstract

          Granulomatosis with polyangiitis, or GPA, is a form of vasculitis that affects multiple organs especially the respiratory tract and the kidneys. The diagnosis is suspected with the clinical presentation and elevated serum titer of antineutrophil cytoplasmic antibodies and confirmed with the biopsy of the affected organ. Viral infection has been described as one of the triggers of the immune system in developing GPA. In this report, we describe a rare case of GPA that developed following cytomegalovirus infection in a patient with unknown immunocompromised medical condition.

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          Most cited references19

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          Emergence of a CD4+CD28- granzyme B+, cytomegalovirus-specific T cell subset after recovery of primary cytomegalovirus infection.

          Ajda Rowshani, Ineke J M ten Berge, Ann Vossen … (2004)
          Cytotoxic CD4(+)CD28(-) T cells form a rare subset in human peripheral blood. The presence of CD4(+)CD28(-) cells has been associated with chronic viral infections, but how these particular cells are generated is unknown. In this study, we show that in primary CMV infections, CD4(+)CD28(-) T cells emerge just after cessation of the viral load, indicating that infection with CMV triggers the formation of CD4(+)CD28(-) T cells. In line with this, we found these cells only in CMV-infected persons. CD4(+)CD28(-) cells had an Ag-primed phenotype and expressed the cytolytic molecules granzyme B and perforin. Importantly, CD4(+)CD28(-) cells were to a large extent CMV-specific because proliferation was only induced by CMV-Ag, but not by recall Ags such as purified protein derivative or tetanus toxoid. CD4(+)CD28(-) cells only produced IFN-gamma after stimulation with CMV-Ag, whereas CD4(+)CD28(+) cells also produced IFN-gamma in response to varicella-zoster virus and purified protein derivative. Thus, CD4(+)CD28(-) T cells emerge as a consequence of CMV infection.
          Article 0 comments Cited 109 times – based on 0 reviews     Review now
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            Wegener's granulomatosis: prospective clinical and therapeutic experience with 85 patients for 21 years.

            B Haynes, Mary S Wolff, Barry Katz … (1982)
            Eighty-five patients with Wegener's granulomatosis were studied for 21 years at the National Institutes of Health. Patients were treated with a protocol consisting of cyclophosphamide, 2 mg/kg body weight d, together with prednisone, 1 mg/kg body weight d, followed by conversion of the prednisone to an alternate-day regimen. Complete remissions were achieved in 79 of 85 patients (93%). The mean duration of remission for living patients was 48.2 (+/- 3.6) months. Twenty-three patients are off all therapy for a mean duration of 35.3 (+/- 6.3) months without therapy. This study provides a prospective experience with Wegener's granulomatosis and shows that long-term remissions can be induced and maintained in an extremely high number of patients by the combination of daily cyclophosphamide and alternate-day prednisone therapy.
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              Small-vessel vasculitis.

              Amanda Falk, Charles Jennette (1997)
              Article 0 comments Cited 92 times – based on 0 reviews     Review now
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                Author and article information

                Journal
                Journal ID (nlm-ta): Eur J Rheumatol
                Journal ID (iso-abbrev): Eur J Rheumatol
                Title: European Journal of Rheumatology
                Publisher: Medical Research and Education Association
                ISSN (Print): 2147-9720
                ISSN (Electronic): 2148-4279
                Publication date Collection: January 2022
                Publication date (Electronic): 08 February 2022
                Volume: 9
                Issue: 1
                Pages: 54-57
                Affiliations
                [1 ]Department of Medicine, Tufts Medical Center, Tufts University School of Medicine , Boston, Massachusetts, USA
                [2 ]Department of Medicine and Surgery, Tufts University School of Medicine , Boston, Massachusetts, USA
                [3 ]Division of Pulmonary & Critical Care and Sleep Medicine, Tufts Medical Center , Boston, Massachusetts, USA
                Author notes
                Address for Correspondence: Maher Tabba, Medicine and Surgery, Interventional Pulmonology, Critical Care Medicine-Melrose Wakefield Health System, Division of Pulmonary & Critical Care and Sleep Medicine, Tufts Medical Center, Tufts University School of Medicine, Boston, Massachusetts, USA E-mail: mtabba@ 123456tuftsmedicalcenter.org

                ORCID iDs of the authors: S.G. 0000-0002-5076-8419; D.Z. 0000-0001-7411-0091; E.S. 0000-0001-6108-9748; D.B. 0000-0002-8084-7767; M.T. 0000-0001-6058-7948.

                Cite this article as: Gayen S, Zhang D, Sternlicht E, Bulanowski D, Tabba M. Granulomatosis with polyangiitis: the trigger cannot be long hidden. Eur J Rheumatol. 2021;9(1):54-57.

                Author information
                http://orcid.org/0000-0002-5076-8419
                http://orcid.org/0000-0001-7411-0091
                http://orcid.org/0000-0001-6108-9748
                http://orcid.org/0000-0002-8084-7767
                http://orcid.org/0000-0001-6058-7948
                Article
                Publisher ID: ejr-9-1-54
                DOI: 10.5152/eurjrheum.2021.21093
                PMC ID: 10089136
                PubMed ID: 35110137
                SO-VID: 0e3fec28-e49e-4558-99aa-b02ec4220a9f
                Copyright © © Copyright by 2022 Medical Research and Education Association
                License:

                Content of this journal is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.

                History
                Date received : 12 July 2020
                Date accepted : 19 January 2021
                Page count
                Pages: 0
                Categories
                Subject: Case-based Review

                Keywords: granulomatosis polyangiitis,wegener's disease,cytomegalovirus,pneumonia
                Data availability:
                Keywords: granulomatosis polyangiitis, wegener's disease, cytomegalovirus, pneumonia

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