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      Anti-neutrophil cytoplasmic antibody-negative granulomatosis with polyangiitis manifesting through pulmonary nodules and a genital lesion

      case-report

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          Abstract

          A 72-year-old female presented with bilateral pulmonary nodules before undergoing surgery for hysteroptosis. Transbronchial biopsy did not lead to a definitive diagnosis. The right mass in the upper lobe was resected through video-assisted thoracic surgery. Pathological findings showed granulomatosis with polyangiitis. However, the patient was negative for serum proteinase 3-anti-neutrophil cytoplasmic antibody. Although the nodule in the left lower lobe progressed, the serum inflammatory reaction yielded negative results. Resection of the nodule in the left lower lobe revealed identical pathological findings with those of the right pulmonary mass. Following total hysterectomy for hysteroptosis, the pathological findings indicated granulomatosis with polyangiitis.

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          The American College of Rheumatology 1990 criteria for the classification of wegener's granulomatosis

          Randi Leavitt, Anthony Fauci, Daniel A. Bloch … (1990)
          Article 0 comments Cited 96 times – based on 0 reviews     Review now
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            Granulomatosis with polyangiitis (Wegener): clinical aspects and treatment.

            Cloé Comarmond, Patrice Cacoub (2014)
            Granulomatosis with polyangiitis (GPA) is a systemic necrotizing vasculitis, which affects small- and medium-sized blood vessels and is often associated with cytoplasmic ANCA. GPA occurs in patients between 45 and 60 years old of both genders, and is rarely observed in blacks. The prevalence of GPA increases along a south-north gradient in Europe (20 to 150/million). The main clinical characteristics involve the upper and/or lower respiratory tract and kidneys. Ear, nose and throat manifestations with recurrent sinusitis and crusting rhinorrhea are usually severe. Lung nodules are frequently seen, sometimes excavated. Renal involvement is characterized by rapidly progressive necrotizing glomerulonephritis with extracapillary crescents. Limited forms of GPA predominantly affect the upper respiratory tract, whereas generalized forms of GPA include renal manifestations and/or alveolar hemorrhage and/or vital organ involvement with an altered general condition. The combination of immunosuppressant drugs and corticosteroids has converted this typically fatal illness into one in which 80% of patients achieve remission. However, despite considerable therapeutic progress over the last decades, relapses remain frequent (50% at 5 years), and maintenance treatment is now the main therapeutic challenge.
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              2022 American College of Rheumatology/European Alliance of Associations for Rheumatology classification criteria for granulomatosis with polyangiitis

              Joanna Robson, Peter C Grayson, Cristina Ponte … (2022)
              To develop and validate revised classification criteria for granulomatosis with polyangiitis (GPA). Patients with vasculitis or comparator diseases were recruited into an international cohort. The study proceeded in five phases: (1) identification of candidate criteria items using consensus methodology, (2) prospective collection of candidate items present at the time of diagnosis, (3) data-driven reduction of the number of candidate items, (4) expert panel review of cases to define the reference diagnosis and (5) derivation of a points-based risk score for disease classification in a development set using least absolute shrinkage and selection operator logistic regression, with subsequent validation of performance characteristics in an independent set of cases and comparators. The development set for GPA consisted of 578 cases of GPA and 652 comparators. The validation set consisted of an additional 146 cases of GPA and 161 comparators. From 91 candidate items, regression analysis identified 26 items for GPA, 10 of which were retained. The final criteria and their weights were as follows: bloody nasal discharge, nasal crusting or sino-nasal congestion (+3); cartilaginous involvement (+2); conductive or sensorineural hearing loss (+1); cytoplasmic antineutrophil cytoplasmic antibody (ANCA) or anti-proteinase 3 ANCA positivity (+5); pulmonary nodules, mass or cavitation on chest imaging (+2); granuloma or giant cells on biopsy (+2); inflammation or consolidation of the nasal/paranasal sinuses on imaging (+1); pauci-immune glomerulonephritis (+1); perinuclear ANCA or antimyeloperoxidase ANCA positivity (−1); and eosinophil count ≥1×10 9 /L (−4). After excluding mimics of vasculitis, a patient with a diagnosis of small- or medium-vessel vasculitis could be classified as having GPA if the cumulative score was ≥5 points. When these criteria were tested in the validation data set, the sensitivity was 93% (95% CI 87% to 96%) and the specificity was 94% (95% CI 89% to 97%). The 2022 American College of Rheumatology/European Alliance of Associations for Rheumatology classification criteria for GPA demonstrate strong performance characteristics and are validated for use in research.
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                Author and article information

                Contributors
                Kazutsugu Uematsu
                Journal
                Journal ID (nlm-ta): Respir Med Case Rep
                Journal ID (iso-abbrev): Respir Med Case Rep
                Title: Respiratory Medicine Case Reports
                Publisher: Elsevier
                ISSN (Electronic): 2213-0071
                Publication date PMC-release: 03 December 2023
                Publication date Collection: 2023
                Publication date (Electronic): 03 December 2023
                Volume: 46
                Electronic Location Identifier: 101951
                Affiliations
                [a ]Department of Pulmonary Medicine, Saitama Medical Center, Saitama Medical University, Saitama, Japan
                [b ]Department of Pathology, Saitama Medical Center, Saitama Medical University, Saitama, Japan
                [c ]Department of Medical Laboratory Science, Faculty of Health Sciences, Hokkaido University, Sapporo, Japan
                Author notes
                [∗ ]Corresponding author. Department of Pulmonary Medicine, Saitama Medical Center, Saitama Medical University, 1981 Kamoda, Kawagoe, Saitama 350-8550, Japan kuematsu@ 123456saitama-med.ac.jp
                [1]

                These authors contributed equally to this work.

                Article
                Publisher Item ID: S2213-0071(23)00146-6 Publisher ID: 101951
                DOI: 10.1016/j.rmcr.2023.101951
                PMC ID: 10714361
                PubMed ID: 38090378
                SO-VID: b06aef30-b02a-4f81-8499-64df33323090
                Copyright © © 2023 The Authors
                License:

                This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).

                History
                Date received : 23 August 2023
                Date revision received : 22 October 2023
                Date accepted : 18 November 2023
                Categories
                Subject: Case Report

                Keywords: granulomatosis with polyangiitis,anti-neutrophil cytoplasmic antibody-negative,female genital granulomatosis
                Data availability:
                Keywords: granulomatosis with polyangiitis, anti-neutrophil cytoplasmic antibody-negative, female genital granulomatosis

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