ANCA-associated vasculitis with cardiac valve vegetations in two teenage males: two case reports and a literature review

Theisen, Alexandra; Phillips, Carrie L.; Rodríguez, Martha

doi:10.1186/s12969-022-00750-2

Cited by 3 publications

(2 citation statements)

References 25 publications

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“…According to Goodfield, cardiac involvement is not as rare as initially presumed, present in ≤ 44% of cases of EGPA [ 10 ]. However, it has been documented that cardiac involvement in GPA is a rarer manifestation, “estimated to affect only 5% of pediatric GPA and MPA cases" [ 11 ]. Furthermore, a cohort involving 517 patients diagnosed with GPA demonstrated cardiac manifestations in as little as 3.3% of patients [ 12 ].…”

Section: Discussionmentioning

confidence: 99%

A Rare Presentation of Polyangiitis Overlapping Syndrome

Subramanian¹,

Hanchosky²,

Vuyyuru³

et al. 2023

Cureus

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This case follows a 38-year-old Caucasian male with no known medical history who presented to the emergency department for syncope. He also endorsed a two-month history of fevers, weight loss, oral ulcers, rashes, joint swelling and arthralgias. After extensive workup, he was given a working diagnosis of granulomatosis with polyangiitis (GPA). Conflicting diagnostic evidence made it increasingly difficult to distinguish between GPA and eosinophilic granulomatosis with polyangiitis. In conclusion, we believe the patient may be better diagnosed with polyangiitis overlapping syndrome.

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Section: Discussionmentioning

confidence: 99%

A Rare Presentation of Polyangiitis Overlapping Syndrome

Subramanian¹,

Hanchosky²,

Vuyyuru³

et al. 2023

Cureus

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“…This was further confirmed by follow-up PET/CT showing resolution of uptake after several months of treatment with rituximab. In a recent literature review of pediatric patients with cardiac ANCA-associated vasculitis, three of the five patients had valvular involvement ( 27 ).…”

Section: Discussionmentioning

confidence: 99%

Case Report: Immune checkpoint inhibitor–induced multiorgan vasculitis successfully treated with rituximab

et al. 2023

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Immune checkpoint inhibitors (ICIs) have revolutionized the treatment of cancer. ICIs have a unique side effect profile, generally caused by inflammatory tissue damage, with clinical features similar to autoimmune conditions. Acute kidney injury from ICIs has been well studied; incidence ranges from 1% to 5%, with higher incidence when combination ICI therapies are used. Although the overall reported incidence of ICI-associated glomerulonephritis is less than 1%, vasculitis is the most commonly reported ICI-related glomerulonephritis. Other biopsy findings include thrombotic microangiopathy, focal segmental glomerulosclerosis, minimal change disease, and IgA nephropathy with secondary amyloidosis. We report a case in which a woman previously treated with the PD-L1 inhibitor durvalumab for locally advanced non-small cell lung cancer with pre-existing antineutrophil cytoplasmic (anti-PR3) antibody who later developed multi-organ vasculitis after ICI exposure, which was successfully treated with rituximab, with continued cancer remission for 3 years.

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Skin, Heart, and CNS Involvement in Granulomatosis With Polyangiitis: A Case Report

Mohsin,

Rasool,

Saliba

2024

Cureus

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We discuss the case of a 60-year-old male who presented with ankle pain, a necrotic rash, and progressive weakness in both lower limbs and the right upper limb. An infectious workup of the skin lesions came back negative. Additionally, his kidney function tests indicated an acute kidney injury. This prompted investigations for vasculitis etiologies, which revealed a positive cytoplasmic antineutrophil cytoplasmic autoantibody (c-ANCA). His neurological deficits were also investigated, and imaging suggested embolic infarcts. Cardiac imaging showed valve vegetations and blood culture showed a lack of growth suggestive of a noninfective nature of these lesions. Based on all these findings, a kidney biopsy was obtained and demonstrated pauci-immune segmental vasculitis consistent with ANCA-associated glomerulonephritis. As such, the patient showed improvement with heavy pulse steroid and immunomodulator therapy. Although skin, heart, and CNS involvement have been previously reported with ANCA-associated vasculitis, it is rare, especially together, and can prove a diagnostic challenge. Therefore, it is important to consider vasculitis etiology in patients presenting similarly. In addition, this case highlights the overlapping clinical picture between infective endocarditis and vasculitis with valvular involvement, making differentiation between the two challenging.

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ANCA-associated vasculitis with cardiac valve vegetations in two teenage males: two case reports and a literature review

Cited by 3 publications

References 25 publications

A Rare Presentation of Polyangiitis Overlapping Syndrome

A Rare Presentation of Polyangiitis Overlapping Syndrome

Case Report: Immune checkpoint inhibitor–induced multiorgan vasculitis successfully treated with rituximab

Skin, Heart, and CNS Involvement in Granulomatosis With Polyangiitis: A Case Report

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