Unusual case of levamisole-induced dual-positive ANCA vasculitis and crescentic glomerulonephritis

Cocaine adulterated levamisole is an increasingly reported cause of skin necrosis, arthralgia and systemic vasculitis, but renal involvement is uncommon. We present a case of a 40-year-old Hispanic man with a history of cocaine abuse who presented with acute kidney injury to the rheumatology clinic where he was being treated for chronic inflammatory arthritis. He was found to have a serum creatinine of 2.5 mg/dL, microscopic haematuria and subnephrotic proteinuria, along with positive proteinase 3, myeloperoxidase, anticardiolipin antibodies and an elevated antinuclear antibody titre. The renal pathology revealed focal necrotising glomerulonephritis with crescentic features and mild immune type deposition. The patient was treated with cocaine abstinence, pulse dose steroids followed by maintenance prednisone, rituximab and cyclophosphamide. His renal function subsequently improved but did not normalise. We believe that his incomplete improvement was due to the degree of kidney injury on presentation as well as recidivism with cocaine use.

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Levamisole/Cocaine Induced Systemic Vasculitis and Immune Complex Glomerulonephritis

Case Reports in Nephrology ◽

10.1155/2015/372413 ◽

2015 ◽

Vol 2015 ◽

pp. 1-5 ◽

Cited By ~ 9

Author(s):

Lohit Garg ◽

Sagar Gupta ◽

Abhishek Swami ◽

Ping Zhang

Keyword(s):

Immune Complex ◽

Systemic Vasculitis ◽

Renal Involvement ◽

Morphological Properties ◽

High Dose ◽

Cocaine Use ◽

Immune Mediated ◽

History Of ◽

Psychotropic Effects ◽

Complex Deposition

Levamisole is an antihelminthic and immunomodulator medication that was banned by the USFDA in 1998. It has been increasingly used to adulterate cocaine due to its psychotropic effects and morphological properties. Adverse reactions including cutaneous vasculitis, thrombocytopenia, and agranulocytosis have been well described. Despite systemic vasculitis in this setting, renal involvement is uncommon. We report here a case of ANCA positive systemic vasculitis with biopsy proven immune complex mediated glomerulonephritis likely secondary to levamisole/cocaine. A 40-year-old Caucasian male with no past medical history presented with 3-week history of fatigue, skin rash, joint pains, painful oral lesions, oliguria, hematuria, worsening dyspnea on exertion, and progressive lower extremity edema. He had a history of regular tobacco and cocaine use. Lab testing revealed severe anemia, marked azotemia, deranged electrolytes, and 4.7 gm proteinuria. Rheumatologic testing revealed hypocomplementemia, borderline ANA, myeloperoxidase antibody, and positive atypical p-ANCA. Infectious and other autoimmune workup was negative. Kidney biopsy was consistent with immune mediated glomerulonephritis and showed mesangial proliferation and immune complex deposition consisting of IgG, IgM, and complement. High dose corticosteroids and discontinuing cocaine use resulted in marked improvement in rash, mucocutaneous lesions, and arthritis. There was no renal recovery and he remained hemodialysis dependent.

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Acute kidney injury as initial presentation of renal lymphoma: Diagnostic and therapeutic challenges in a resource-limited setting

SAGE Open Medical Case Reports ◽

10.1177/2050313x19845251 ◽

2019 ◽

Vol 7 ◽

pp. 2050313X1984525 ◽

Cited By ~ 1

Author(s):

Jeannine Anyingu Aminde ◽

Nkweta Eugene Adze ◽

Guisilla Ankwatia Dedino ◽

Leopold Ndemnge Aminde

Keyword(s):

Acute Kidney Injury ◽

State Of The Art ◽

Renal Involvement ◽

Kidney Injury ◽

Abdominal Distension ◽

Lymphoma Patient ◽

Limited Data ◽

Resource Limited ◽

History Of ◽

Limited Setting

Non-Hodgkin’s lymphoma is reportedly common in Africa; however, there is limited data on renal involvement. Acute kidney injury only at presentation is rare for lymphoproliferative malignancies. A 7-year old presented to our facility with a 2-week history of progressive abdominal distension and pain, examination revealed anasarca and hypertension. On further evaluation, there were bilateral nephromegaly, acute kidney injury (AKI) and cytomorphological findings suggestive of lymphoma. Patient management was mostly supportive, and evolution was unfavourable leading to his demise. We discuss diagnostic and therapeutic challenges due to unavailability of state-of-the-art facilities in resource-constrained settings.

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Clinical, neurovascular and neuropathological features in sneddon's syndrome

Arquivos de Neuro-Psiquiatria ◽

10.1590/s0004-282x2007000300005 ◽

2007 ◽

Vol 65 (2b) ◽

pp. 390-395 ◽

Cited By ~ 6

Author(s):

Jaqueline Luvisotto Marinho ◽

Élcio Juliato Piovesan ◽

Moacir Pereira Leite Neto ◽

Luiz Roberto Kotze ◽

Lúcia de Noronha ◽

...

Keyword(s):

Young Women ◽

Renal Involvement ◽

Cerebral Atrophy ◽

Anticardiolipin Antibodies ◽

Livedo Reticularis ◽

Sneddon's Syndrome ◽

Sneddon’S Syndrome ◽

Cerebrovascular Events ◽

History Of ◽

Ct And Mri

Sneddon's syndrome (SS) is characterized by ischemic cerebrovascular episodes and livedo reticularis. It is more common in young women and can also be associated with valvulopathy, a history of spontaneous abortion, renal involvement and vascular dementia. We describe three cases of young women with this disease. The patients had repeated ischemic cerebral episodes, livedo reticularis and thrombocytopenia. CT and MRI showed strokes and cerebral atrophy. Autopsy in one of the patients revealed cerebral infarctions. Anticardiolipin antibodies were detected in two patients. Antiphospholipid antibodies may be found in some patients with ischemic cerebrovascular events and livedo reticularis. SS may thus be associated with antiphospholipid syndrome. We described three new cases of SS and discuss the pathophysiology of this disease.

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Pauci-Immune Necrotizing and Crescentic Glomerulonephritis with Membranous Lupus Nephritis, Fifteen Years after Initial Diagnosis of Secondary Membranous Nephropathy

Case Reports in Nephrology ◽

10.1155/2015/120762 ◽

2015 ◽

Vol 2015 ◽

pp. 1-5 ◽

Cited By ~ 1

Author(s):

Ryan Burkhart ◽

Nina Shah ◽

Michael Abel ◽

James D. Oliver ◽

Matthew Lewin

Keyword(s):

Lupus Nephritis ◽

Renal Biopsy ◽

Immune Complex ◽

Lupus Erythematosus ◽

Membranous Nephropathy ◽

Crescentic Glomerulonephritis ◽

Renal Involvement ◽

Kidney Injury ◽

Rapid Decline ◽

Secondary Membranous Nephropathy

Renal involvement in systemic lupus erythematosus (SLE) is usually immune complex mediated and may have multiple different presentations. Pauci-immune necrotizing and crescentic glomerulonephritis (NCGN) refers to extensive glomerular inflammation with few or no immune deposits that may result in rapid decline in renal function. We report a case of a 79-year-old Hispanic male with a history of secondary membranous nephropathy (diagnosed by renal biopsy 15 years previously) who was admitted with acute kidney injury and active urinary sediment. P-ANCA titers and anti-myeloperoxidase antibodies were positive. The renal biopsy was diagnostic for NCGN superimposed on a secondary membranous nephropathy. A previous diagnosis of SLE based on American College of Rheumatology criteria was discovered via Veteran’s Administration records review after the completion of treatment for pauci-immune NCGN. ANCAs are detected in 20–31% of patients with SLE. There may be an association between SLE and ANCA seropositivity. In patients with lupus nephritis and biopsy findings of necrotizing and crescentic glomerulonephritis, without significant immune complex deposition, ANCA testing should be performed. In patients with secondary membranous nephropathy SLE should be excluded.

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Proliferative glomerulonephritis and mantle cell lymphoma: a rare association

BMJ Case Reports ◽

10.1136/bcr-2020-238153 ◽

2021 ◽

Vol 14 (2) ◽

pp. e238153

Author(s):

Bilel Mhedhbi ◽

Soumaya Chargui ◽

Amel Harzallah ◽

Rim Goucha

Keyword(s):

Acute Kidney Injury ◽

Mantle Cell Lymphoma ◽

Cell Lymphoma ◽

Oral Prednisone ◽

Renal Involvement ◽

Kidney Injury ◽

Antineutrophil Cytoplasmic Antibody ◽

Proteinase 3 ◽

Rare Association ◽

Mantle Cell

Renal involvement in mantle cell lymphoma (MCL) is rare. We present the case of a man followed for MCL presented with acute kidney injury and positive antineutrophil cytoplasmic antibody (ANCA) type anti proteinase 3 (PR3). He was treated as for a rapidly progressing glomerulonephritis with cyclophosphamide and methylprednisolone followed by oral prednisone. Renal biopsy revealed diffuse endocapillary proliferation and segmental extracapillary proliferation in four glomeruli. Immunohistochemistry confirmed the renal invasion of lymphomatous cells. He started improving his renal function shortly after starting treatment. The coexistence of renal MCL infiltration, extracapillary proliferation and ANCA positive is exceptional.

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An Unusual Case of Anti-GBM Antibody Elevation in HIV-Associated Nephropathy

Case Reports in Nephrology ◽

10.1155/2014/956475 ◽

2014 ◽

Vol 2014 ◽

pp. 1-6

Author(s):

Vinay Minocha ◽

Raafat Makary ◽

Andreea Poenariu

Keyword(s):

Hiv Infection ◽

Unusual Case ◽

Elevated Serum ◽

Kidney Injury ◽

African American Female ◽

Case Presentation ◽

A Value ◽

Biopsy Confirmation ◽

History Of ◽

Nephrotic Range Proteinuria

Introduction.The most commonly seen glomerular disease in HIV infected patients is HIV-associated nephropathy (HIVAN); however, a multitude of other nephropathies can occur in HIV infection with an almost equal cumulative frequency. We report an unusual case of a patient with clinical and histological evidence of HIVAN in which the diagnosis was initially confounded by the finding of an elevated serum anti-glomerular basement membrane (anti-GBM) antibody.Case Presentation.We present a case of a 27-year-old African American female with a history of schizophrenia, cocaine abuse, and HIV infection who upon admission to our hospital was found to have severe acute kidney injury requiring hemodialysis. Urine studies revealed nephrotic range proteinuria and a serological workup was positive for anti-GBM antibody elevation with a value of 91 units (normal: 0–20 units). A renal biopsy revealed HIVAN with no evidence of crescentic glomerulonephritis or anti-GBM disease.Conclusion.This case highlights the need for careful interpretation of anti-GBM antibody tests in HIV infected patients with kidney disease and, in particular, the need for biopsy confirmation of the diagnosis prior to starting therapy. More research is needed to study the prognostic correlation between the degree of anti-GBM antibody elevation in HIVAN and disease severity.

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Biopsy-Proven Kidney Involvement in Hypocomplementemic Urticarial Vasculitis

10.21203/rs.3.rs-471283/v1 ◽

2021 ◽

Author(s):

Alice Corthier ◽

Marie Jachiet ◽

Daniel Bertin ◽

Aude Servais ◽

Christelle Barbet ◽

...

Keyword(s):

Kidney Disease ◽

Kidney Biopsy ◽

Systemic Vasculitis ◽

Renal Involvement ◽

Kidney Injury ◽

Urticarial Vasculitis ◽

Kidney Involvement ◽

National Cohort ◽

Hypocomplementemic Urticarial Vasculitis

Abstract Background. Hypocomplementemic urticarial vasculitis (HUV) is a rare systemic vasculitis. We aimed to describe the kidney involvement of HUV in a multicenter national cohort with an extended follow-up. Methods. All patients with HUV (international Schwartz criteria) with a biopsy-proven kidney involvement, identified through a survey of the French Vasculitis Study Group, were included. A systematic literature review on kidney involvement of HUV was performed. Results. Twelve patients were included, among whom 8 had positive anti-C1q antibodies. All presented with proteinuria, from mild to nephrotic, and 8 displayed acute kidney injury (AKI), requiring temporary haemodialysis in 2. Kidney biopsy showed membrano-proliferative glomerulonephritis (GN) in 8 patients, pauci-immune crescentic GN or necrotizing vasculitis in 3 patients (with a mild to severe interstitial inflammation), and an isolated interstitial nephritis in 1 patient. C1q deposits were observed in the glomeruli (n=6), tubules (n=4) or renal arterioles (n=3) of 8 patients. All patients received corticosteroids, and 9 were also treated with immunosuppressants or apheresis. After a mean follow-up of 8.9 years, 6 patients had a preserved renal function, but 2 patients had developed stage 3-4 chronic kidney disease (CKD) and 4 patients had reached end-stage kidney disease (ESKD), among whom 1 had received a kidney transplant. Conclusion. Renal involvement of HUV can be responsible for severe AKI, CKD and ESKD. It is not always associated with circulating anti-C1q antibodies. Kidney biopsy shows mostly membrano-proliferative GN or crescentic GN, with frequent C1q deposits in the glomeruli, tubules or arterioles.

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A relapse of POEMS syndrome presenting as acute kidney injury

BMJ Case Reports ◽

10.1136/bcr-2018-228894 ◽

2019 ◽

Vol 12 (8) ◽

pp. e228894

Author(s):

David Owen Rees ◽

David Hywel Thomas ◽

Nilima Parry-Jones ◽

Gareth Roberts

Keyword(s):

Acute Kidney Injury ◽

Peripheral Neuropathy ◽

Rare Disease ◽

Blood Test ◽

Renal Involvement ◽

Kidney Injury ◽

Poems Syndrome ◽

Skin Changes ◽

Monoclonal Protein ◽

History Of

Peripheral neuropathy, organomegaly, endocrinopathy, monoclonal protein and skin changes (POEMS) syndrome is a rare disease, and only in a minority of cases, causes an impairment of kidney function. Here, we describe a case of a 55-year-old man with a history of POEMS syndrome who presented with acute kidney injury following a routine blood test. On further investigation, a relapse in POEMS syndrome was diagnosed, uniquely isolated to renal involvement.

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Abstract 16758: Unexplained Recurrent St-elevation Myocardial Infarction Complicated By Diffuse Alveolar Hemorrhage In Undiagnosed Granulomatosis With Polyangiitis

Circulation ◽

10.1161/circ.144.suppl_2.16758 ◽

2021 ◽

Vol 144 (Suppl_2) ◽

Author(s):

Juthipong Benjanuwattra ◽

Poemlarp Mekraksakit ◽

Mahmoud Abdelnabi

Keyword(s):

Chest Pain ◽

Granulomatosis With Polyangiitis ◽

Systemic Vasculitis ◽

Renal Involvement ◽

Diffuse Alveolar Hemorrhage ◽

Alveolar Hemorrhage ◽

Renal Functions ◽

History Of ◽

St Elevation ◽

Work Up

Case Presentation: A 53-year-old man with a history of well-controlled hypertension and dyslipidemia presented with chest pain due to anterior STEMI for which a proximal LAD stent was placed. A week later, he presented with recurrent chest pain and ST elevation in the anterior chest leads. Coronary angiography showed in-stent thrombosis with heavy thrombus burden extending through the LAD. Manual thrombectomy with a second stent placement was done. He was discharged on triple therapy (aspirin, clopidogrel, and rivaroxaban), later, he presented to our medical facility with a 1-week history of hemoptysis. His clinical condition deteriorated requiring intubation. Laboratory work-up revealed elevated renal functions, proteinuria, and significant hematuria. CXR showed diffuse patchy opacities while CT chest was consistent with diffuse alveolar hemorrhage (Figure 1). Further work-up revealed elevated anti-proteinase 3 (c-ANCA) and low C4. Based on microscopic hematuria, renal impairment, and a positive c-ANCA, a diagnosis of granulomatosis with polyangiitis (GPA) was made. Induction with methylprednisolone, cyclophosphamide, and rituximab was initiated. His condition gradually improved, he was extubated successfully with marked improvement of renal functions. He was discharged on clopidogrel and scheduled for follow-up. Discussion: Few case reports have described STEMI as the initial presentation of GPA, likely attributed to vasculitis and accelerated atherosclerosis from systemic inflammation. Our patient had well-controlled cardiovascular risk factors and renal involvement with hematuria and elevated creatinine at the time of STEMI diagnosis, together with intermittent joint pains, suggesting ongoing systemic vasculitis. Clinicians should have a high index of suspicion for the possibility of non-atherosclerotic coronary stenosis as in cases of coronary vasculitis for early detection and management of a potentially reversible condition.

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ATYPICAL PRESENTATION IN A CASE OF GRANULOMATOSIS WITH POLYANGIITIS

Romanian Journal of Rheumatology ◽

10.37897/rjr.2016.1.5 ◽

2016 ◽

Vol 25 (1) ◽

pp. 37-42

Author(s):

Cristina Capusa ◽

◽

Ana-Maria Mehedinti ◽

Claudia Toma ◽

Violeta Bojinca ◽

...

Keyword(s):

Crescentic Glomerulonephritis ◽

Granulomatosis With Polyangiitis ◽

Renal Involvement ◽

Proteinase 3 ◽

Atypical Presentation ◽

Upper Respiratory Tract ◽

Correct Treatment ◽

Anca Associated Vasculitis ◽

Vasa Recta ◽

Upper Respiratory

Granulomatosis with polyangiitis (GPA) is part of the anti-neutrophil cytoplasmic antibodies (ANCA) associated vasculitis, mostly being ANCA-c (anti proteinase 3) positive. Primarily it involves the upper respiratory tract and kidneys having an increased mortality in the absence of early diagnosis and correct treatment. The most common renal involvement is pauci-immune crescentic glomerulonephritis. We present the case of a patient with GPA with a particular onset of interstitial nephritis, possible by vasa recta vasculitis, in the absence of glomerulonephritis.

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