Diffuse alveolar haemorrhage in a systemic lupus erythematosus patient successfully treated with rituximab: a case report

Nellessen, Cordula; Pöge, Uwe; Brensing, Karl August; Sauerbruch, Tilman; Klehr, H. U.; Rabe, Christian

doi:10.1093/ndt/gfm701

Cited by 39 publications

(28 citation statements)

References 14 publications

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“…The pro-inflammatory role of B-cells in DAH, suggested in the current study is also consistent with recent reports on successful treatment of cases of DAH in SLE by B cell depletion therapy using Rituxan (Rituximab) (38-40). Since the effects of Rituximab are rapid, far before any possible effect on the levels of circulating autoantibodies, it has been suggested that the main mechanism of work is via control of antigen presenting function, B-cell cytokine stimulation, and T-cell activation (40).…”

Section: Discussionsupporting

confidence: 93%

“…While targeting B cells seems to be a potentially attractive therapy, they represent a minor population found within the hemorrhagic lungs of both SLE patients and B6 mice. Nevertheless, B-cell depleting therapy may be considered as a potential treatment option based on the pathogenic role of B-cells shown in the current study and a few reports of successful treatment of alveolar hemorrhage in SLE (2, 38-40). …”

Section: Discussionmentioning

confidence: 85%

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Pathogenic role of B cells in the development of diffuse alveolar hemorrhage induced by pristane

Barker

Lee

Kelly-Scumpia

et al. 2011

Laboratory Investigation

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Diffuse alveolar hemorrhage is an uncommon yet often fatal complication of systemic lupus erythematosus (SLE). Advances in the treatment of alveolar hemorrhage have been hampered due to the heterogeneity of clinical findings and the lack of suitable animal models. A single intraperitoneal injection of pristane induces a lupus-like syndrome characterized by lupus-related autoantibodies and glomerulonephritis in non-autoimmune prone strains of mice. In addition, C57BL/6 (B6) mice frequently develop alveolar hemorrhage within a few weeks of pristane injection. Immunopathogenesis of pristane-induced alveolar hemorrhage was investigated in the present study. Early (2-4 weeks after injection) mortality due to hemorrhage was unique to C57BL/6 and C57BL/10 strains of mice. Recruitment of the macrophages and neutrophils preceded the hemorrhage by several days and hemorrhage started 3-7 days after pristane injection in some mice, peaked at 2 weeks (84% in B6) and then resolved by 4 weeks in a majority of mice. Alveolar hemorrhage was independent of MyD88-, or TLR7 pathways, in contrast to autoantibody production and glomerulonephritis, and also was independent of FcγR or Fas. Rag1-/- mice had a reduced prevalence of alveolar hemorrhage compared to B6 (P = 0.01) congenics. However, T-cell receptor deficient mice developed alveolar hemorrhage at a rate comparable to wild type controls, while B6 Igμ-/- mice surprisingly had a strikingly reduced prevalence (7% vs 84% in B6, P < 0.0001). Reconstitution of B6 Igμ-/- mice with wild type B cells increased the prevalence to 50% (P = 0.028). Pristane-induced alveolar hemorrhage is a useful model to study the pathogenesis and develop new therapy for this underappreciated and often life-threatening complication of SLE.

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Section: Discussionsupporting

confidence: 93%

Section: Discussionmentioning

confidence: 85%

Pathogenic role of B cells in the development of diffuse alveolar hemorrhage induced by pristane

Barker

Lee

Kelly-Scumpia

et al. 2011

Laboratory Investigation

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“…Plasmapheresis may be useful for patients who are not responsive to the above regimens [8]. In recent years, SLE-associated DAH treated successfully with rituximab without CYC administration has been reported in some cases, but lacks larger-scale clinical studies [9][10][11].…”

Section: Introductionmentioning

confidence: 99%

Allogeneic transplantation of umbilical cord-derived mesenchymal stem cells for diffuse alveolar hemorrhage in systemic lupus erythematosus

Shi

Wang

et al. 2012

Clin Rheumatol

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Umbilical cord-derived mesenchymal stem cell transplantation (UC-MSCT) has been proved to be effective in the treatment of systemic lupus erythematosus (SLE), based on animal experiments and clinical trials. Diffuse alveolar hemorrhage (DAH) is a rare complication of SLE with a high mortality usually over 50%. This study aimed to assess the efficacy of UC-MSCT in the treatment of SLE-associated DAH. Four SLE patients complicated with DAH, who underwent UC-MSCT, were included. Clinical changes before and after transplantation were assessed by measurements of hemoglobin, platelet level, oxygen saturation, and serological factors. High-resolution CT (HRCT) scans of the chest were performed to evaluate pulmonary manifestation. All the four patients showed dramatic improvements of their clinical manifestations. Hemoglobin was elevated after UC-MSCT and was sustained at a normal level 6 months after UC-MSCT in the four patients. Platelet level was upregulated in two patients who had thrombocytopenia at baseline. Oxygen saturation appeared to be normal at 1 month after UC-MSCT, and this result was confirmed by the HRCT scan of the chest. Serum albumin elevated to 3.5 g/dl 6 months after transplantation. Our findings suggest that UC-MSCT results in amelioration of oxygen saturation as well as hematological and serologic changes, which revealed that UC-MSCT could be applied as a salvage strategy for DAH patients.

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“…5 This may be especially true in SLE-associated DAH, as microscopic images of lung biopsies from SLE patients with DAH show immune complex deposition along alveolar and blood vessel walls. 6 Rituximab is a chimeric anti-CD20 monoclonal antibody used in the treatment of refractory autoimmune diseases. As CD20 is expressed by all B-cells, rituximab reduces cytokine secretion and autoantibody production by specifically targeting these B-cells.…”

Section: Discussionmentioning

confidence: 99%

Rituximab: an emerging treatment for recurrent diffuse alveolar hemorrhage in systemic lupus erythematosus

Tse

Cctc

McMahon

et al. 2014

Lupus

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Diffuse alveolar hemorrhage (DAH) is a rare manifestation of systemic lupus erythematosus (SLE) and is associated with high mortality rates. Treatment typically consists of aggressive immunosuppression with pulse-dose steroids, cyclophosphamide, and plasma exchange therapy. Mortality rates remain high despite use of multiple medical therapies. We present a case of recurrent DAH in a 52-year-old female with SLE after a deceased donor renal transplant who was successfully treated with rituximab. Our report highlights the pathophysiologic importance of B-cell-mediated immunosuppression in SLE-associated DAH and suggests that rituximab may represent a viable alternative to cyclophosphamide in the treatment of this disease. We also review eight other reported cases of rituximab use in SLE-associated DAH.

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Diffuse alveolar haemorrhage in a systemic lupus erythematosus patient successfully treated with rituximab: a case report

Cited by 39 publications

References 14 publications

Pathogenic role of B cells in the development of diffuse alveolar hemorrhage induced by pristane

Pathogenic role of B cells in the development of diffuse alveolar hemorrhage induced by pristane

Allogeneic transplantation of umbilical cord-derived mesenchymal stem cells for diffuse alveolar hemorrhage in systemic lupus erythematosus

Rituximab: an emerging treatment for recurrent diffuse alveolar hemorrhage in systemic lupus erythematosus

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