Anti-neutrophil cytoplasmic antibodies in cholesterol embolism: A case report and literature review

Zhang, Jun; Zhang, Hengyuan; Chen, Shizhi; Huang, Jian

doi:10.3892/etm.2016.3349

Cited by 5 publications

(7 citation statements)

References 22 publications

(62 reference statements)

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“…Of those 12 patients, the mean age was 69 years; 10 were males with multiple medical comorbidities[11]. One-third of the patients had spontaneous atheroembolism like our patient[11]. The role of ANCA in atheroembolic diseases remains to be elucidated.…”

Section: Discussionmentioning

confidence: 94%

“…Although the impact of technology upon health care is immense, diagnosing AERD remains challenging[9,10]. The association between atheroembolism and ANCA positivity is exceedingly rare[11]. A recent literature review performed by Zhang et al[11] mentioned 12 cases (3 with c-ANCA, 6 with p-ANCA, 1 with both, 2 positive for ANCA by early indirect immunofluorescence) of cholesterol embolism with ANCA positivity.…”

Section: Discussionmentioning

confidence: 99%

“…The association between atheroembolism and ANCA positivity is exceedingly rare[11]. A recent literature review performed by Zhang et al[11] mentioned 12 cases (3 with c-ANCA, 6 with p-ANCA, 1 with both, 2 positive for ANCA by early indirect immunofluorescence) of cholesterol embolism with ANCA positivity. Of those 12 patients, the mean age was 69 years; 10 were males with multiple medical comorbidities[11].…”

Section: Discussionmentioning

confidence: 99%

“…A recent literature review performed by Zhang et al[11] mentioned 12 cases (3 with c-ANCA, 6 with p-ANCA, 1 with both, 2 positive for ANCA by early indirect immunofluorescence) of cholesterol embolism with ANCA positivity. Of those 12 patients, the mean age was 69 years; 10 were males with multiple medical comorbidities[11]. One-third of the patients had spontaneous atheroembolism like our patient[11].…”

Section: Discussionmentioning

confidence: 99%

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A rare presentation of spontaneous atheroembolic renal disease: A case report

Piranavan¹,

Rajan²,

Jindal³

et al. 2019

WJN

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BACKGROUNDAtheroembolic renal disease (AERD) is caused by occlusion of the small renal arteries from embolized cholesterol crystals arising from ulcerated atherosclerotic plaques. This usually manifests as isolated renal disease or involvement from systemic atheroembolic disease. Here we report a case of AERD that responded well to steroid therapy.CASE SUMMARYA 62-year-old woman with a history of hypertension and stage IIIa chronic kidney disease was referred for rapidly worsening renal function over a 4-mo period. She complained of swollen legs, dyspnea on exertion, and two episodes of epistaxis about a month prior to admission. She reported no history of invasive vascular procedures, use of radio contrast agents, or treatment with anticoagulants or thrombolytic agents. Urinalysis showed a few red blood cells and granular casts. Serology was positive for cytoplasmic antineutrophil cytoplasmic antibodies (c-ANCA). Non-contrast-enhanced computed tomography of the chest, abdomen, and pelvis showed diffuse atherosclerotic changes in the aortic arch. Thus, c-ANCA-associated vasculitis was suspected, and the patient was started on pulse intravenous methylprednisolone. Her renal biopsy showed evidence of AERD. She was discharged with oral prednisone, and her renal function continued to improve during the initial follow-up.CONCLUSIONIn cases of non-vasculitis-associated ANCA, a high degree of clinical suspicion is required to pursue the diagnosis of spontaneous AERD in patients with clinical or radiological evidence of atherosclerotic burden. Although no specific treatment is available, the potential role of statins and steroids requires exploration.

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Section: Discussionmentioning

confidence: 94%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

See 2 more Smart Citations

A rare presentation of spontaneous atheroembolic renal disease: A case report

Piranavan¹,

Rajan²,

Jindal³

et al. 2019

WJN

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“…We report the case of a 52-year-old Caucasian woman with a 4 months progressive onset of diffuse, very intense (10/10) The secondary causes are more pleomorphic (7) and may include parasitic, fungal or viral infections, autoimmune diseases, allergic disorders, Addison disease , drug-induced (9) hypereosinophilias, malignancies (both solid and hematological disorders) , cholesterol (1) embolism and multiple primary or secondary (8) immun deficiencies disorders.…”

Section: Case Reportmentioning

confidence: 99%

Primary FIP1L1-PDGFR Positive Hypereosinophilic Syndrome with Unusual Rheumatic Onset

et al. 2020

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Introduction. Hypereosinophilia (HE) is defined as an absolute eosinophil count >1.5 x 109/L in the peripheral blood on two examinations separated in time by at least one month and/or pathologic confirmation of tissue HE. A hypereosinophilic syndrome (HES) is defined by the association of HE with eosinophil-mediated organ damage and/or dysfunction if other potential causes of them have been excluded.Case report. We report a case of a 52-year-old woman admitted to Rheumatology Department for diffuse, highly intense (10/10) myalgia, associated with paravertebral, upper and lower limbs muscular edema and stiffness, having the onset of symptoms for about 4 months. During rheumatological evaluation, the patient developed posterior cervical diffuse pain with brachial irradiation associated with severe functional impotence reason for which it was indicated MRI of cervical spine. The blood count showed leucocytosis with hypereosinophilia. No evidence of Raynaud’s phenomenon, cutaneous and muscular sclerosis or visceral involvement. All muscular groups and deep fascia appeared affected on the whole-body MRI and the findings were suggestive for Shulman’s eosinophilic fasciitis or eosinophilic myopahty. All autoimmune, parasitic and allergic causes for myopathy were ruled out. Muscle tissue biopsy revealed high eosinophilic infiltrate predominantly in the deep muscular fascia, in the striated muscles and in the superficial dermis.The patient received methyl-prednisolone pulse-therapy with a low-maintenance dose of prednisone which showed clinical improvement and normalized peripheral blood eosinophilia. A hematologic evaluation revealed high percentage of bone marrow eosinophil count which harbored FIP1L1-PDGFR alpha mutation and data was suggestive for the diagnosis of myeloproliferative variant of HES syndrome along with Shulman’s eosinophilic fasciitis for which Imatinib treatment was indicated.Conclusion. The particularity of this case consists in the diagnosis of a myeloproliferative variant of HES with atypical presentation as a Shulman’s eosinophilic fasciitis, a rheumatological condition. Even though initial symptoms were suggestive for a myopathy, it was difficult to establish the positive diagnosis without any obvious paraclinical data. Further investigations such as MRI and muscle biopsy were conclusive for the diagnosis of Shulman’s eosinophilic fasciitis and clinical improvement was observed after corticosteroid treatment. Considering that after 4 months of evolution of the disease the patient did not present any atrophic lesions of the affected muscles and skin characteristic for Shulman’s eosinophilic fasciitis the patient was referred to Hematology Department with the diagnosis of HES. Advanced exams established the hematological condition as primary FIP1L1-PDGFR positive HES which could benefit from tyrosine-kinase inhibitors treatment (Imatinib) known to induce in this situation improved clinical and paraclinical status.

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2020 international consensus on ANCA testing beyond systemic vasculitis

Moiseev

Tervaert

Arimura

et al. 2020

Autoimmunity Reviews

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This document follows up on a 2017 revised international consensus on antineutrophil cytoplasm antibodies (ANCA) testing in granulomatosis with polyangiitis and microscopic polyangiitis and focuses on the clinical and diagnostic value of ANCA detection in patients with connective tissue diseases, idiopathic interstitial pneumonia, autoimmune liver diseases, inflammatory bowel diseases, anti-glomerular basement membrane (GBM) disease, infections, malignancy, and during drug treatment. Current evidence suggests that in certain settings beyond systemic vasculitis, ANCA may have clinical, pathogenic and/or diagnostic relevance.Antigen-specific ANCA targeting proteinase-3 and myeloperoxidase should be tested by solid phase immunoassays in any patient with clinical features suggesting ANCAassociated vasculitis and in all patients with anti-GBM disease, idiopathic interstitial pneumonia, and infective endocarditis associated with nephritis, whereas in patients with other aforementioned disorders routine ANCA testing is not recommended. Among patients with autoimmune liver diseases or inflammatory bowel diseases, ANCA testing may be justified in patients with suspected autoimmune hepatitis type 1 who do not have conventional autoantibodies or in case of diagnostic uncertainty to discriminate ulcerative colitis from Crohn's disease. In these cases, ANCA should be tested by indirect immunofluorescence as the target antigens are not yet well characterized. Many questions concerning the optimal use of ANCA testing in patients without ANCA-associated vasculitis remain to be answered.Testing for anti-neutrophil cytoplasm antibodies (ANCA) directed towards proteinase 3 (PR3) and myeloperoxidase (MPO) is commonly performed to support the diagnosis of ANCA-associated vasculitides (AAV) that encompasses granulomatosis with polyangiitis (GPA), microscopic polyangiitis (MPA), and eosinophilic granulomatosis with polyangiitis (EGPA). In 2017, a revised international consensus on testing of ANCA in GPA and MPA was proposed [1]. This consensus document focused on ANCA testing in AAV and briefly described other conditions that were reported to be associated with ANCA positivity, mostly in the context of the differential diagnosis of other inflammatory diseases [2]. The current document is a follow-up on the previous consensus statements [1,3,4] and highlights the clinical and diagnostic value of ANCA testing in patients with various autoimmune, infectious and neoplastic diseases. However, the number of high quality studies addressing this issue is limited, and many unresolved issues concerning the optimal use of ANCA testing in non-AAV remain to be elucidated. MethodsThis Consensus Statement was prepared by a group of experts based on the results of a comprehensive search in PubMed for disorders that can be associated with ANCApositivity, by using a variety of search terms, and all relevant available literature was critical reviewed. Additional publications were identified in the references of the available articles. The resulting manus...

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Anti-neutrophil cytoplasmic antibodies in cholesterol embolism: A case report and literature review

Cited by 5 publications

References 22 publications

A rare presentation of spontaneous atheroembolic renal disease: A case report

A rare presentation of spontaneous atheroembolic renal disease: A case report

Primary FIP1L1-PDGFR Positive Hypereosinophilic Syndrome with Unusual Rheumatic Onset

2020 international consensus on ANCA testing beyond systemic vasculitis

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