scholarly journals Renal-Limited Antiglomerular Basement Membrane Disease Related To Alemtuzumab: A Case Report

2020 ◽  
Vol 7 ◽  
pp. 205435812096268
Author(s):  
Amye M. Harrigan ◽  
Michael L. West ◽  
Laurette Geldenhuys ◽  
Penelope Poyah

Rationale: Alemtuzumab is a monoclonal antibody approved for the treatment of relapsing-remitting multiple sclerosis (RRMS). Many autoimmune-mediated adverse events have been associated with alemtuzumab, including renal-limited anti-glomerular basement membrane (GBM) disease. Presenting concern: A 52-year-old female with RRMS presented with acute kidney injury 39 months after receiving 1 cycle of alemtuzumab. She had a history of alemtuzumab-associated hypothyroidism and thrombocytopenia, urinary tract infections, and chronically abnormal urinalyses. Diagnosis: A diagnosis of renal-limited anti-GBM disease was made based on renal biopsy and positive anti-GBM serology. Alemtuzumab was thought to be the trigger of the anti-GBM disease as there were no other exposures or serologic findings suggesting other causes. Interventions: She was treated with corticosteroids, cyclophosphamide, and plasmapheresis. She required hemodialysis for acute renal failure. Outcomes: Despite treatment, the patient’s renal function did not recover. She remained dialysis-dependent and anti-GBM antibody titers remained elevated 6 months after presentation. Teaching points: Anti-GBM disease is a life-altering adverse event that can be associated with alemtuzumab. Our case highlights the limitations of monitoring urinalyses as a trigger for anti-GBM antibody testing in patients who have received alemtuzumab and have baseline abnormal urinalyses; such patients may require further protocolized anti-GBM antibody testing, although the optimal frequency of such antibody screening remains unclear.

2021 ◽  
Vol 22 (1) ◽  
Author(s):  
Alexander Winkler ◽  
Emanuel Zitt ◽  
Hannelore Sprenger-Mähr ◽  
Afschin Soleiman ◽  
Manfred Cejna ◽  
...  

Abstract Background Anti-glomerular basement membrane disease (GBM) disease is a rare autoimmune disease causing rapidly progressive glomerulonephritis and pulmonary haemorrhage. Recently, an association between COVID-19 and anti-glomerular basement membrane (anti-GBM) disease has been proposed. We report on a patient with recurrence of anti-GBM disease after SARS-CoV-2 infection. Case presentation The 31-year-old woman had a past medical history of anti-GBM disease, first diagnosed 11 years ago, and a first relapse 5 years ago. She was admitted with severe dyspnoea, haemoptysis, pulmonary infiltrates and acute on chronic kidney injury. A SARS-CoV-2 PCR was positive with a high cycle threshold. Anti-GBM autoantibodies were undetectable. A kidney biopsy revealed necrotising crescentic glomerulonephritis with linear deposits of IgG, IgM and C3 along the glomerular basement membrane, confirming a recurrence of anti-GBM disease. She was treated with steroids, plasma exchange and two doses of rituximab. Pulmonary disease resolved, but the patient remained dialysis-dependent. We propose that pulmonary involvement of COVID-19 caused exposure of alveolar basement membranes leading to the production of high avidity autoantibodies by long-lived plasma cells, resulting in severe pulmonary renal syndrome. Conclusion Our case supports the assumption of a possible association between COVID-19 and anti-GBM disease.


2020 ◽  
pp. 302-310
Author(s):  
Liza Kearl ◽  
Maureen McCollough

Renal emergencies in pediatric patients range from more common conditions such as urinary tract infections to rarer conditions such as hemolytic uremic syndrome. This chapter reviews emergency conditions that are less commonly seen, with potentially significant sequelae, and with possible nonspecific or more subtle presentations. Acute kidney injury can be due to a wide range of causes, including benign gastroenteritis and post-streptococcal or toxin-related causes. Less common but more serious conditions, such as nephrotic syndrome or Henoch–Schönlein purpura, present with nonspecific signs such as edema or rash. Acute glomerulonephritis needs to be considered in a child with a history of streptococcal pharyngitis or skin infection. Hemolytic uremic syndrome is typically Shiga toxin-related and should be considered in any ill-appearing child, especially those presenting with bloody stools. Disposition of a child with a renal emergency will depend on the severity of illness, laboratory results, and the ability to follow-up with their primary care provider or specialist.


2015 ◽  
Vol 41 (3) ◽  
pp. 241-247 ◽  
Author(s):  
Alvaro Orjuela ◽  
Adisak Suwanichkul ◽  
Debra Canter ◽  
Charles G. Minard ◽  
Sridevi Devaraj ◽  
...  

Background/Aims: There is a critical need for more noninvasive biomarkers to identify nephritis in patients with systemic lupus erythematosus (SLE). Recent studies in a model mouse and an adult SLE patient cohort suggest that anti-basement membrane antibody levels correlate well with lupus activity and kidney injury. The purpose of this study was to assess the anti-basement membrane reactivity in pediatric SLE (pSLE) patients with or without nephritis. Methods: Auto-antibodies to basement membrane antigens were assessed using an anti-matrigel ELISA. Endpoint titers were measured in pSLE patients and healthy children, as well as in autoimmune and non-immune mice, with good reproducing capabilities. Findings were also analyzed with respect to the presence or absence of nephritis, dsDNA antibodies, and other manifestations of pSLE. Results: MRL/lpr mice developed high-titer anti-matrigel antibodies, whereas C57BL/6 mice did not. In a cohort of 21 pSLE patients and 22 pediatric controls, high-titer anti-matrigel IgG, IgM and IgA antibody levels were specific for pSLE. High-titer anti-matrigel IgG3 levels could distinguish with good sensitivity the 13 pSLE patients with a history of nephritis from the 8 non-renal pSLE patients. High-titer anti-matrigel IgG, IgA, IgM or IgG3 did not correlate with positive anti-double stranded DNA, but defined an overlapping subset of patients. Conclusion: The addition of anti-basement membrane antibody testing to serologic testing in pSLE may help to monitor disease activity or to define important subsets of patients with risks for specific disease manifestations.


2012 ◽  
Vol 19 (3) ◽  
pp. 369-371 ◽  
Author(s):  
Deepti Anbarasan ◽  
Jonathan Howard

We discuss the case of a patient with a known history of relapsing–remitting multiple sclerosis (MS) who presented with the isolated complaint of altered visual perception in the absence of abnormalities on ophthalmological examination. To the best of the authors’ knowledge, this is the first documented case of both facial metamorphopsia and palinopsia occurring as the symptoms of demyelinating brain lesions consistent with an acute MS exacerbation. These symptoms appear to be related to active demyelination that either involved the optic radiations in the visual pathway or the visual association area in the temporo-occipital region of the left hemisphere.


2017 ◽  
Vol 3 (4) ◽  
pp. 205521731774534
Author(s):  
A Traboulsee ◽  
DKB Li ◽  
R Tam ◽  
G Zhao ◽  
A Riddehough ◽  
...  

Background Evolution of gadolinium-enhancing lesions into chronic black holes (CBH) may be reduced by interferon (IFN) therapy. Objective The objective of this paper is to assess the effect of IFN β-1a and placebo on CBH evolution and disability in patients with relapsing–remitting multiple sclerosis (RRMS), as well as CBH evolution in patients with secondary progressive multiple sclerosis (SPMS). Methods A post hoc, exploratory analysis of patients with RRMS and SPMS with monthly MRI scans (months –1 to 9) from two separate placebo-controlled clinical trials of IFN β-1a was conducted. Results In RRMS patients, the risk of ≥1 evolved CBH was lower for IFN β-1a versus placebo (odds ratio 0.42; p = 0.024); volume of newly evolved CBH was numerically reduced. A numerically higher proportion of patients with ≥1 evolving CBH vs no evolving CBH had confirmed three-month disability progression (four-year rate 55.8% vs 43.1%, respectively). Proportion of lesions evolving into CBH (patient level: 34.7% vs 12.6%, p < 0.0001; lesion level: 28.8% vs 11.0%, p < 0.0001) and evolved CBH volume (median 33.5 mm3 (Quartile 1, 0.0; Quartile 3, 173.4) vs 0.0 mm3 (0.0; 52.4); p = 0.0008) was higher for SPMS than RRMS patients treated with IFN β-1a. Conclusion In RRMS, IFN β-1a significantly decreased the proportion of new T1 Gd+ lesions evolving into CBH and the risk of developing a CBH. In patients with SPMS, more lesions develop to CBH, indicating reduced repair capacity, and the natural history of lesion development appears to be unaffected by IFN β-1a treatment.


Author(s):  
Ana Rita Matos ◽  
Elisabete Coelho ◽  
Sofia Caridade

A 77-year-old man with arterial hypertension and dyslipidaemia, treated with olmesartan/hydrochlorothiazide and simvastatin, was admitted with a 3-week history of anorexia, nausea, vomiting, profuse diarrhoea and weight loss. He was dehydrated and blood tests showed acute kidney injury. The aetiological study was inconclusive. The patient had a favourable clinical evolution during hospitalization and was discharged. However, after about 10 days at home, he was re-admitted to hospital with the same clinical presentation. It was noticed that olmesartan had not been prescribed during the previous admission but had been restarted on an outpatient basis. Biopsy examination showed duodenal mucosa with villous atrophy and polymorphic inflammatory infiltrate. Antibody testing for coeliac disease was negative. Based on these facts, it was hypothesized that the patient had olmesartan-induced enteropathy, which was subsequently confirmed.


2019 ◽  
Vol 12 (4) ◽  
pp. e227951 ◽  
Author(s):  
Yves Michiels ◽  
Olivier Bugnon ◽  
Jean-François Michiels ◽  
Sophie Mazellier

In addition to the TRANSFORMS, FREEDOMS, INFORMS studies, very few publications have identified new cases of skin cancer in patients treated with fingolimod. Here, we present the case of a 52-year-old Caucasian patient with relapsing remitting multiple sclerosis for 19 years, with a phototype II with blue eyes, light brown hair, no personal or family history of melanoma and a low number of naevi (<10). She did not experience intense sun exposure in childhood as well as severe sunburn and did not practise sessions in ultraviolet cabins. This case is distinguished from other published cases, usually superficial spreading malignant melanoma by its unclassifiable histological character. The occurrence of skin cancers in patients with multiple sclerosis remains exceptional, but new cases have recently emerged requiring the strengthening of dermatological follow-up of such patients.


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