A novel therapeutic strategy using extracorporeal membrane oxygenation in patients with anti-neutrophil cytoplasmic antibodies-associated vasculitis: a case report and literature review

Wang, Li; Jiang, Wang; Xu, Yi; Jiao, Jie; Xie, Lixin; Mo, Guoxin

doi:10.21037/atm-21-3133

Cited by 3 publications

(5 citation statements)

References 18 publications

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“…The indications for ECOM included oxygenation index less than 80%, ventilation failure in the prone position, life-threatening pneumothorax or haemoptysis 3. Only very few cases had been reported with the application of ECMO in DAH related to AAV, since such situation is rare 3. In our case, ECMO was an essential part of treatment and evidently improved the prognosis.…”

Section: Discussionmentioning

confidence: 61%

“…There are no absolute contraindications according to current ECMO guidelines, which advocate a case-by-case assessment 18. In the literature, no bleeding events occurred among the reported cases of AAV-related DAH requiring ECMO 3. As far as we know, no cases of DAH with subconjunctival haemorrhage have been reported.…”

Section: Discussionmentioning

confidence: 90%

“…When conventional mechanical ventilation fails, ECMO is used to provide oxygenation promptly. The indications for ECOM included oxygenation index less than 80%, ventilation failure in the prone position, life-threatening pneumothorax or haemoptysis 3. Only very few cases had been reported with the application of ECMO in DAH related to AAV, since such situation is rare 3.…”

Section: Discussionmentioning

confidence: 99%

“…Lung lesions are an important feature of AAV 2. ANCA-positive pulmonary capillaritis complicated by diffuse alveolar haemorrhage (DAH) is a potentially fatal situation for which extracorporeal membrane oxygenation (ECMO) can facilitate improved outcomes and potential cure 3 4. Here, we show a case of a young man, admitted with catastrophic DAH and subconjunctival haemorrhage caused by underlying AAV who ultimately required ECMO.…”

Section: Introductionmentioning

confidence: 99%

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Rare cause of diffuse alveolar haemorrhage and subconjunctival haemorrhage rescued by extracorporeal membrane oxygenation and rituximab

Feng

Zhang

et al. 2022

BMJ Case Rep

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A previously healthy man in his 20s presented with acute respiratory distress syndrome and subconjunctival haemorrhage. Imaging was indicative of pervasive pulmonary haemorrhage. There was no evidence of renal involvement. The patient rapidly deteriorated with aggravating respiratory failure regardless of invasive mechanical ventilation and required extracorporeal membrane oxygenation (ECMO). This maintained the patient adequate time to allow aggressive therapy. Skin biopsy indicated leucocytoclastic vasculitis. Given that the patient was C-antinuclear cytoplasmic autoantibody (ANCA) positive, pulse dose steroids and rituximab were initiated for the suspicion of ANCA-associated vasculitis (AAV) which resulted in improvement of airspace disease and subconjunctival haemorrhage. Only a few cases reported successful use of ECMO in severe diffuse alveolar haemorrhage (DAH) due to AAV, but no case was in DAH combined with subconjunctival haemorrhage. The need for systemic anticoagulation with pre-existing haemorrhage is still a challenging dilemma.

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Section: Discussionmentioning

confidence: 61%

Section: Discussionmentioning

confidence: 90%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Rare cause of diffuse alveolar haemorrhage and subconjunctival haemorrhage rescued by extracorporeal membrane oxygenation and rituximab

Feng

Zhang

et al. 2022

BMJ Case Rep

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“…ECMO was used as rescue therapy in some patients. ECMO is an alternative for patients who failed conventional mechanical ventilation to treat DAH in patients with ANCA-associated vasculitis [ 20 ].…”

Section: Reviewmentioning

confidence: 99%

Granulomatosis With Polyangiitis Presenting With Diffuse Alveolar Hemorrhage: A Systematic Review

Silva¹,

Adhikari²

2022

Cureus

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Granulomatosis with polyangiitis (GPA) is a systemic disease that has variable clinical expression. GPA is the most common antineutrophilic cytoplasmic antibody (ANCA)-associated vasculitis (AAV). Diffuse alveolar hemorrhage (DAH) is one of the least common pulmonary manifestations in patients with GPA. DAH is clinically marked by hemoptysis, anemia, and diffuse alveolar infiltrates on imaging as well as hypoxemic respiratory failure. The diagnosis and treatment are challenging. Recommendations for ANCA-associated vasculitis, in general, are already established; however, there is a knowledge gap that addresses the association of GPA and DAH. The aim of this systematic review is to focus on the main clinical aspects and treatment of patients with GPA who present with DAH.Thorough research of available literature was performed, including studies published in the last 10 years, following the Preferred Items for Systematic Review and Meta-Analysis (PRISMA) 2020 recommendations. The following databases were included: PubMed, Medline, Embase, ClinicalTrials.com, Google Scholar, and Prospero. The search terms included: [granulomatosis] AND [polyangiitis] AND [diffuse alveolar hemorrhage] OR [diffuse pulmonary hemorrhage] NOT [microscopic polyangiitis] NOT [eosinophilic granulomatosis]. NOS was used to assess the internal validity of the study in four domains, including selection, ascertainment, causality, and reporting. Our initial search identified 8989 studies. After excluding duplicated data and using our predetermined inclusion and exclusion criteria, we were able to retrieve 18 studies. Twelve out of 18 (67%) studies were case reports. Five were retrospective cohorts and one controlled trial. The average age of patients with GPA with DAH was 49. 55 ± 17.54 years (18 -76). Male individuals had a slight predominance (59%) in comparison to female individuals (41%). The hemoglobin level at the time of presentation was 8.86 mg/dL ± 1.43. The majority of patients (61.5%) reported hemoptysis. Renal involvement was present in 66.7%. Patients who required mechanical ventilation represented 61.5%. Plasmapheresis was used in 71.4%. Mortality was 20%, and gender was not associated with mortality (p = 0.822). Hemoptysis was not associated with the need for mechanical ventilation (p = 0.928). Renal impairment was not a predictor of mechanical ventilation (p = 0.207).In summary, patients with GPA and DAH were severely ill, frequently had renal impairment upon admission, and frequently required mechanical ventilation. Steroids, rituximab, and cyclophosphamide are the first-line treatment, and plasmapheresis is still in use. Eventually, extracorporeal membrane oxygenation (ECMO) can be the salvage therapy. Randomized controlled clinical trials (RCTs) are needed to address the best therapeutic options for this population.

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Critically Ill Patients with Newly Diagnosed Anti-Neutrophil Cytoplasmic Antibody-Associated Vasculitis: Case Series and Literature Review

Rukavina,

Zlopasa,

Vukovic Brinar

et al. 2024

JCM

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ANCA-associated vasculitides (AAVs) are rare diseases with a prevalence of less than 200 cases per million persons and an incidence of less than 25 cases per million person-years. Their presenting features can vary from prodromal and nonspecific symptoms to dramatic organ-specific symptoms such as respiratory failure due to diffuse alveolar hemorrhage (DAH) and acute kidney injury (AKI). The latter two are hallmark features of pulmonary-renal syndrome, a potentially fatal condition that necessitates early recognition and treatment in intensive care units (ICUs) and rapid induction of immunosuppressive therapy. Background and case summaries: We described three patients with newly diagnosed AAV during the treatment of critical illness. All patients had DAH and two had AKI. The initial disease severity was extremely high in patients with myeloperoxidase (MPO)-AAV, reaching Sequential Organ Failure Assessment (SOFA) scores of 15 and 14 with predicted mortality ≥ 95.2%. Both patients needed mechanical ventilation, one additional venovenous extracorporeal membrane oxygenation (VV-ECMO), and renal replacement therapy. The patient with proteinase 3 (PR3)-AAV had a less severe disease, SOFA 3, requiring only modest oxygen supplementation and exhibiting only hematuria with normal renal function parameters. Immunosuppressive therapy was initiated during the ICU stay. The patient with the most severe clinical presentation died during the ICU stay because of sepsis, and the other two patients were discharged home. Conclusions: Patients with AAV presenting with pulmonary-renal syndrome necessitate various degrees of organ support. Nevertheless, these patients can be successfully treated in the early, critical stages of the disease and achieve remission.

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A novel therapeutic strategy using extracorporeal membrane oxygenation in patients with anti-neutrophil cytoplasmic antibodies-associated vasculitis: a case report and literature review

Cited by 3 publications

References 18 publications

Rare cause of diffuse alveolar haemorrhage and subconjunctival haemorrhage rescued by extracorporeal membrane oxygenation and rituximab

Rare cause of diffuse alveolar haemorrhage and subconjunctival haemorrhage rescued by extracorporeal membrane oxygenation and rituximab

Granulomatosis With Polyangiitis Presenting With Diffuse Alveolar Hemorrhage: A Systematic Review

Critically Ill Patients with Newly Diagnosed Anti-Neutrophil Cytoplasmic Antibody-Associated Vasculitis: Case Series and Literature Review

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