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Dynamically changing antineutrophil cytoplasmic antibodies in granulomatosis with polyangiitis:A case report

World Journal of Clinical Cases 2024年第16期12卷 2881-2886页

作者： Yan Zhang Qiao-Ding Dai Ji-An Wang Li-Ping Xu Qiang Chen Yang-Zi Jin Department of Rheumatism and Immunology The First Affiliated Hospital of Zhejiang Chinese Medical University/Zhejiang Provincial Hospital of Traditional Chinese MedicineHangzhou 310006Zhejiang ProvinceChina Department of Orthopaedics The Affiliated Xiaoshan Hospital of Hangzhou Normal UniversityHangzhou 311202Zhejiang ProvinceChina Department of Otolaryngology The First Affiliated Hospital of Zhejiang Chinese Medical University/Zhejiang Provincial Hospital of Traditional Chinese MedicineHangzhou 310006Zhejiang ProvinceChina

BACKGROUND granulomatosis with polyangiitis(GPA)is one of the most prevalent forms of the antineutrophil cytoplasmic antibody(ANCA)-associated *** is characterized histologically by necrotizing granulomatous inflammation in addition to *** diagnosis of GPA depends on clinical presentation,serological evidence of a positive ANCA,and/or histological evidence of necrotizing vasculitis or granulomatous destructive parenchymal *** ANCA(c-ANCA)is positive in 65%-75% of GPA patients,accompanied by proteinase 3(PR3),the main target antigen of c-ANCA,another 5% of GPA patients had negative *** SUMMARY The patient,a 52-year-old male,presented with unexplained nasal congestion,tinnitus,and hearing *** a duration of 4 months experiencing these symptoms,the patient subsequently developed fever and *** imaging examination revealed the presence of bilateral auricular mastoiditis and partial paranasal sinusitis,and the ANCA results were *** anti-infective therapy proved to be ineffective,but the patient's symptoms and fever were quickly relieved after 1 wk of treatment with methylprednisolone 40 mg once a ***,after continuous use of methylprednisolone tablets for 3 months,the patient experienced a recurrence of fever accompanied by right-sided migraine,positive c-ANCA and PR3,and increased total protein in cerebrospinal *** and cyclophosphamide 0.8 g monthly,the patient experienced alleviation of fever and ***,the ANCA levels became negative and there has been no *** For GPA patients with negative ANCA,there is a potential for early missed *** integration of histopathological results and multidisciplinary communication plays a crucial role in facilitating ANCA-negative GPA.

关键词： Anti-neutrophil cytoplasmic antibodies granulomatosis with polyangiitis Antineutrophil cytoplasmic antibodyassociated vasculitis Immunosuppressive therapy Case report

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A Bronchopulmonary Onset of Candidemia Revealing a granulomatosis with polyangiitis

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Case Reports in Clinical Medicine 2024年第5期13卷 147-154页

作者： Chaïmaâ Zeroual Mina Moudatir Khadija Echchilali Leila Barakat Hassan El Kabli Department of Internal Medicine Ibn Rochd University Hospital Center Casablanca Morocco

Candidemia is defined as being a yeast infection confirmed by the presence of at least one positive Candida blood culture. It is a life threatening infection causing high mortality. The clinical signs are generally compatible with the causative agent (whether there is a deep venous catheter or not). On the other hand and according to the 2012 Revised Chapel Hill Classification, granulomatosis with polyangiitis GPA is classified as a vasculitis associated with antineutrophil cytoplasmic antibodies ANCA. It is a systemic disease characterized by the anatomopathological aspect of granuloma. We report the case of a patient who presented an atypical and a very rare revealing mode of GPA which was a bronchopulmonary candidiasis complicated by candidemia. Despite its controversy, the combination in the acute phase of antifungal treatment based on intravenous voriconazole and glucocorticoid therapy has made it possible to control candidemia and calm vasculitis.

关键词： Candidemia C. glabrata Pulmonary Candidiasis Vasculitis Immunodepression granulomatosis with polyangiitis Cavitary Lesions Antifungal Therapy Immunosuppressants

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granulomatosis with polyangiitis presenting as high fever with diffuse alveolar hemorrhage and otitis media: A case report

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World Journal of Clinical Cases 2021年第10期9卷 2344-2351页

作者： Xiao-Jie Li Liu Yang Xiao-Feng Yan Chu-Ting Zhan Jiang-Hua Liu Department of General Practice The Second Affiliated Hospital of Guangxi Medical UniversityNanning 530007Guangxi Zhuang Autonomous RegionChina Department of General Practice General Practice School of Guangxi Medical UniversityNanning 530007Guangxi Zhuang Autonomous RegionChina

BACKGROUND granulomatosis with polyangiitis is a necrotizing inflammation of small andmedium-sized vessels accompanied by formation of granuloma, involvement ofprimary granulomatous upper and lower respiratory tracts, glomerulonephritis,and vasculitis of small *** SUMMARY Herein, we described a case of a 52-year-old man admitted with pulmonarynodules and high fever. Autoantibody workup revealed that the patient waspositive for c-anti-neutrophil cytoplasmic antibodies and proteinase-3 antineutrophilcytoplasmic antibodies. Pulmonary biopsies revealed a localgranulomatous structure. The patient received therapy with methylprednisoloneand intravenous immunoglobulin, and his clinical symptoms *** Intravenous immunoglobulin may act on granulomatosis with polyangiitis similarto immunosuppressants.

关键词： granulomatosis with polyangiitis Diffuse alveolar hemorrhage Otitis media Methylprednisolone Intravenous immunoglobulin Case report

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Cyclophosphamide-associated enteritis presenting with severe protein-losing enteropathy in granulomatosis with polyangiitis:A case report

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World Journal of Gastroenterology 2021年第20期27卷 2657-2663页

作者： Hiroko Sato Tsuyoshi Shirai Hiroshi Fujii Tomonori Ishii Hideo Harigae Department of Hematology and Rheumatology Tohoku University Graduate School of MedicineSendai 9808574Japan

BACKGROUND Although cyclophosphamide(CPA)is the key drug for the treatment of autoimmune diseases including vasculitides,it has some well-known adverse effects,such as myelosuppression,hemorrhagic cystitis,infertility,and ***,CPA-associated severe enteritis is a rare adverse effect,and only one case with a lethal clinical course has been ***,the appropriate management of patients with CPA-associated severe enteritis is *** SUMMARY We present the case of a 61-year-old woman diagnosed with granulomatosis with polyangiitis based on the presence of symptoms in ear,lung,and,kidney with positive myeloperoxidase-antineutrophil cytoplasmic *** received pulsed methylprednisolone followed by prednisolone 55 mg/d and intravenous CPA at a dose of 500 mg/*** days after the second course of intravenous CPA,she developed nausea,vomiting,and diarrhea,and was admitted to the *** testing revealed hypoalbuminemia,suggesting proteinlosing *** tomography revealed wall thickening of the stomach,small intestine,and colon with contrast enhancement on the lumen *** and immunosuppressive therapy were not effective,and the patient’s enteritis did not improve for>4 *** her condition became seriously exhausted,corticosteroids were tapered and supportive therapies including intravenous hyperalimentation,replenishment of albumin and gamma globulin,plasma exchange,and infection control were *** supportive therapies improved her condition,and her enteritis gradually *** was finally discharged 7 mo *** Immediate discontinuation of CPA and intensive supportive therapy are crucial for the survival of patients with CPA-associated severe enteritis.

关键词： Antineutrophil cytoplasmic antibody Cyclophosphamide Enteritis granulomatosis with polyangiitis Plasma exchange Vasculitis Case report

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Successful treatment of granulomatosis with polyangiitis using tocilizumab combined with glucocorticoids:A case report

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World Journal of Clinical Cases 2023年第5期11卷 1144-1151页

作者： Peng-Fang Tang Long-Chuan Xu Wen-Ting Hong Hong-Ying Shi Department of Rheumatology Second Affiliated Hospital of Fujian Medical UniversityQuanzhou 362000Fujian ProvinceChina Department of Pathology Second Affiliated Hospital of Fujian Medical UniversityQuanzhou 362000Fujian ProvinceChina Department of Immunology Second Affiliated Hospital of Fujian Medical UniversityQuanzhou 362000Fujian ProvinceChina

BACKGROUND Tocilizumab is a humanized monoclonal antibody against the interleukin-6(IL-6)receptor that is commonly used to treat large vessel vasculitis and antineutrophil cytoplasmic antibody-related small vessel ***,tocilizumab in combination with glucocorticoids for successfully treating granulomatosis with polyangiitis(GPA)has rarely been *** SUMMARY Here,we report a 40-year-old male patient who suffered GPA for 4 *** was treated with multiple rounds of drugs,including cyclophosphamide,Tripterygium wilfordii,mycophenolate mofetil,and belimumab,with no *** addition,he exhibited persistently high IL-6 *** tocilizumab treatment,his symptoms improved,and his inflammatory marker levels returned to *** Tocilizumab may be effective for treating GPA.

关键词： Tocilizumab granulomatosis with polyangiitis Antineutrophil cytoplasmic antibody Case report

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Periungual Capillaroscopy Findings in a Series of Patients with granulomatosis with polyangiitis(Wegener):An Observational Study

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Open Journal of Rheumatology and Autoimmune Diseases 2013年第2期3卷 130-134页

作者： Uceda Montanés Julia Martínez Pérez Rosalía Velloso Feijoo María Luisa Marenco de la Fuente José Luis Rheumatology Unit Valme University HospitalSevilleSpain

Introduction: Periungual capillaroscopy studies skin microcirculation. Its main indication is in the differential diagnosis of Raynaud’s phenomenon as well as diagnosis and prognosis of systemic sclerosis (SS). Many rheumatic diseases present characteristic capillaroscopic alterations, but little is known at present about the alterations in skin microcirculation of systemic vasculitis. Objective: Our main objective was to study the alterations in skin microcirculation by capillaroscopy in patients with granulomatosis with polyangiitis (GPA). The secondary objective was to evaluate the association of capillaroscopic alterations with systemic alteration. Material and Methods: We studied patients in our unit with GPA, and we collected demographic and clinical variables. Periungual capillaroscopy was performed on all patients, and always by the same investigator. Results: We reviewed 10 patients with a mean age of 55.7. They were predominantly women. We found capillaroscopic alterations in 8 of the 10 patients. Morphological alteration was the most common finding (62.5%), followed by bleeding (50%). We found no mega-capillaries or neovessels. These findings were not related to any specific organ system involvement. Discussion: The tortuous pattern with bleeding was predominant in our series, with lower capillary density, findings which were unrelated to any specific organ system involvement. A single study describes capillaroscopic alterations in GPA, detecting a high percentage (92%) of avascular areas, in contrast with the predominance of a twisting pattern and bleeding found in our study. Further studies are warranted to confirm these results.

关键词： Periungual Capillaroscopy Vasculitis granulomatosis with polyangiitis

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Ocular Manifestations in granulomatosis with polyangiitis Patients from Saudi Arabia

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Open Journal of Rheumatology and Autoimmune Diseases 2021年第1期11卷 9-18页

作者： Abdurhman Saud Al Arfaj Najma Khalil Department of Medicine Rheumatology Division College of Medicine King Saud University Riyadh KSA College of Medicine Research Center College of Medicine King Saud University Riyadh KSA

Eye and/or orbit involvement occurs in granulomatosis with polyangiitis (GPA) patients frequently. The aim of our study was to describe the clinical manifestations, therapy and outcome of ocular involvement in our GPA patients. A retrospective study was conducted including patients with GPA who followed up in Rheumatology clinics during 1990-2016 at King Khalid University Hospital, Riyadh. Information relating to demographics, ocular manifestations, laboratory findings, therapy and outcome of GPA patients were noted. Ocular involvement was detected in 9 (39.1%) of the 23 GPA cases identified. The mean age of ocular GPA patients was 51.8 (range 27 - 62) years, the mean age at onset of disease was 39.6 (range 11 - 57) years and the mean duration of disease was 9.0 (range 2 - 19) years. Concomitant ear, nose, throat and sino-nasal manifestations occurred with ocular symptoms in 77.8% GPA patients. The most frequent manifestations were, eye pain (66.7%), scleritis/episcleritis (55.6%), eye redness and itching (55.6% each). Antineutrophil cytoplasmic antibodies (ANCA) were positive in 88.9% patients, 55.6% had c-ANCA and 33.3% had p-ANCA. Infections were observed in 22.2% of patients, which included pneumonia in one patient and esophageal candidiasis and bacterial meningitis in another. All patients received oral prednisolone, 44.4% received intravenous cyclophosphamide, 22.2% refractory cases received rituximab doses and the disease outcome was good. Comparison of ocular GPA with non-ocular GPA patients showed that 77.8% of ocular GPA patients had concomitant sino-nasal symptoms compared to 42.9% in non-ocular GPA patients and 22.2% of the ocular GPA patients had renal involvement compared to 64.3% in non-ocular GPA patients (p = 0.049). We found that the frequency of ocular manifestations in our GPA patients was similar to reports elsewhere, and the most frequent symptom was eye pain and scleritis/epi

关键词： granulomatosis with polyangiitis Ocular Manifestations Orbital Involvement Lacrimal Gland Involvement

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Eosinophilic granulomatosis with polyangiitis and Bowel Perforation: Case Report

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Journal of Pharmacy and Pharmacology 2022年第11期10卷 283-287页

作者： PauloÁtila da Silva Viana Leonardo de Oliveira Cavalcante Liz Miranda Mateus Lara Farias Lustosa da Costa Deborah Giovanna Santana Rabelo Medical Residency in Rheumatology General Hospital Dr.Cesar CalsCeará60.015-050Brazil Service Coordinator Medical Residency in Rheumatology General Hospital Dr.Cesar CalsCeará60.015-050Brazil Medical-Academic League of Rheumatology of Fortaleza Christu University CenterFortaleza 60.160-230Brazil Medical-Academic League of Rheumatology of Fortaleza Cearástate UniversityFortaleza 60.714-903Brazil

The small and medium sized vessels vasculitis included in the group of vasculitis associated with anti-neutrophil cytoplasmic antibodies,is a rare disease characterized by the presence of vasculitis in association with asthma and *** objective of this report is to describe a case with a typical initial presentation of eosinophilic granulomatosis with polyangiitis-EGPA,that evolved with atypical clinical manifestations and findings in complementary *** the clinical evolution and comprehending the therapeutic response of the patient in question,with scientific embasement on published medical literature.

关键词： granulomatosis with polyangiitis Vasculitis Asthma Eosinophilia Acute abdomen

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Olfactory dysfunction in antineutrophil cytoplasmic antibodyassociated vasculitides: A review of the literature

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World Journal of Methodology 2021年第2期11卷 15-22页

作者： Alfonso Luca Pendolino Anika Kaura Annakan V Navaratnam Monica Pendolino Gerolamo Bianchi Samit Unadkat Giancarlo Ottaviano Premjit S Randhawa Peter J Andrews Department of Ear Nose and ThroatRoyal National ENT and Eastman Dental HospitalsLondon WC1E 6DGUnited Kingdom Ear Institute University College LondonLondon WC1X 8EEUnited Kingdom Division of Rheumatology Department of Locomotor SystemASL 3Genoa 16121Italy Department of Neurosciences DNS Otolaryngology SectionUniversity of PaduaPadua 35128Italy

Olfactory dysfunction(OD)has been described in patients with antineutrophil cytoplasmic antibody-associated vasculitides(AAV),but the underlying mechanisms are not completely *** causes of altered smell function can generally be divided into conductive,sensorineural or *** date no specific treatment is available for AAV-related OD and the efficacy of currently available options has not been *** aim of this review is to provide an overview of the causes that may lead to OD in patients with *** available treatments for OD and possible options in patients with AAV presenting with smell impairment are also mentioned.

关键词： Smell Olfactory dysfunction Antineutrophil cytoplasmic antibody-associated vasculitis diseases granulomatosis with polyangiitis Eosinophilic granulomatosis with polyangiitis Microscopic polyangiitis

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Urological manifestations and treatment of the primary systemic vasculitides

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World Journal of Clinical Urology 2015年第1期4卷 5-20页

作者： Javeria Peracha Matthew David Morgan School of Immunity and Infection College of Medical and Dental Sciences University of Birmingham Centre for Translational Inflammation Research University of Birmingham Research LaboratoriesQueen Elizabeth Hospital Birmingham

The primary systemic vasculitides(PSV) are a group of rare inflammatory disorders affecting blood vessels of varying size and multiple organs. Urological manifestations of PSV are uncommon. Testicular vasculitis is the most commonly reported finding and is associated with Polyarteritis Nodosa(PAN), Henoch-Sch?nleinPurpura(HSP), anti-neutrophil cytoplasm antibody associated Vasculitides(AAV), Giant Cell Arteritis(GCA) and Kawasaki disease. Prostatic vasculitis has been reported in association with GCA and AAV. Ureteric involvement has been noted in PAN, HSP and AAV. Other urogenital manifestations of PSV include genital ulceration and bladder dysfunction in Beh?ets Disease and haematuria which is commonly seen in many of the PSV. Finally, therapies used to treat the PSV, especially cyclophosphamide, are associated with urological sideeffects including haemorrhagic cystitis and urothelial malignancy. The aim of this review is to examine how the urological system is involved in the PSV. Each PSV is examined in turn, with a brief clinical description of the disease followed by a description of the urological manifestations and management. Identification of urological manifestations of PSV is important as in many cases symptoms may improve with immunosuppressive therapy, avoiding the need for invasive surgery. Additionally, patients who present with isolated urogenital PSV are at higher risk of developing subsequent systemic vasculitis and will need to be followed up closely.

关键词： Urology Vasculitis Takayasu arteritis Giant cell arteritis Polyarteritis Nodosa Kawasaki disease Henoch-Schonlein Purpura Anti-neutrophil cytoplasm antibody associated vasculitides granulomatosis with polyangiitis Microscopic polyangiitis Behcets disease Eosinophilic granulomatosis with polyangiitis Epididymo-orchitis Prostatitis Ureteric stenosis

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