Alveolar Hemorrhage, a Rare and Life-Threatening Complication of Catastrophic Antiphospholipid Syndrome

Loza, Grace; Hallo, Carlos; Chiliquinga, Byron; Hallo, Alejandro

doi:10.1155/2019/3284258

Cited by 5 publications

(7 citation statements)

References 17 publications

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“… 9 Pulmonary manifestations include acute respiratory distress syndrome, pulmonary thromboembolism and pulmonary haemorrhage. 10 Nervous system involvement manifests as encephalopathy, stroke, seizures, headache and coma. Cardiac involvement includes heart failure, myocardial infarction and defects.…”

Section: Discussionmentioning

confidence: 99%

Cases of catastrophic anti-phospholipid syndrome in systemic lupus erythematosus: An experience

Hegde

Dorji

Asturkar

et al. 2022

SAGE Open Medical Case Reports

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Anti-phospholipid syndrome is an autoimmune disorder characterized by episodes of arterial and/or venous thrombosis and/or pregnancy morbidity in the presence of anti-phospholipid antibodies. Catastrophic anti-phospholipid syndrome is an accelerated form of the disease with rapid involvement of multiple organ systems often posing a diagnostic challenge. There is a paucity of literature on the myriad presentations of catastrophic anti-phospholipid syndrome owing to the orphan nature of the disease. We present three cases of catastrophic anti-phospholipid syndrome in patients with systemic lupus erythematosus that presented with episodes of thrombosis involving both arterial and venous systems and multisystem organ failure. Timely diagnoses were made based on a high index of suspicion and were managed with a combination of systemic glucocorticoids, cyclophosphamide, plasmapheresis, intravenous immunoglobulin and other supportive measures. However, despite providing the standard of care, we encountered a poor outcome in two of these patients, highlighting the high mortality associated with catastrophic anti-phospholipid syndrome.

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Section: Discussionmentioning

confidence: 99%

Cases of catastrophic anti-phospholipid syndrome in systemic lupus erythematosus: An experience

Hegde

Dorji

Asturkar

et al. 2022

SAGE Open Medical Case Reports

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“…This disease's thrombotic episodes may happen in any blood vessel, resulting in a broad range of symptoms. Peripheral thrombosis (deep vein thrombosis in 38.9% of cases) and neurology (20.2% of cases) are linked to the most frequent kinds of presentation [2]. It is characterized by multiorgan failure caused by microvascular thrombosis; the kidney is most often affected, followed by the lungs and the central nervous system (CNS).…”

Section: Introductionmentioning

confidence: 99%

Extensive Thrombosis in Catastrophic Antiphospholipid Syndrome in a Newly Diagnosed Systemic Lupus Erythematosus: A Case Report

Tanariyakul,

Nebrajas,

Saowapa

et al. 2024

Cureus

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In this case report, we present the development of catastrophic antiphospholipid syndrome (CAPS), a rare and potentially fatal consequence of systemic lupus erythematosus (SLE), in a 33-year-old Micronesian woman. CAPS is characterized by extensive arterial thrombosis and multiorgan failure. The patient first showed signs of neuropsychiatric symptoms, brain infarctions on imaging, and severe hypoxic respiratory failure brought into the hospital by diffuse alveolar hemorrhage (DAH) along with lupus nephritis (LN). Blood urea nitrogen (BUN) and creatinine (Cr) were progressively elevated to 102/4.1 mg/dL, respectively. A urinalysis revealed microscopic hematuria and proteinuria with a urine protein/creatinine ratio of 6052 mg/g. She was also found to have had microangiopathic hemolytic anemia (MAHA) and severe venous thrombosis, both of which were indicative of CAPS. An aggressive approach, including immunosuppressive medication, therapeutic plasma exchange, and anticoagulation, had positive results, including renal recovery and the cessation of thrombotic episodes. This instance highlights how crucial it is to identify CAPS patients early and take appropriate action to improve patient outcomes for this difficult and sometimes deadly disorder.

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“…Thrombosis in APS develop in any blood vessels and causes a wide spectrum of clinical picture [5]. The most common manifestation is peripheral thrombosis of deep veins in 39 % of cases and cerebral vessels.…”

mentioning

confidence: 99%

“…Such bleeding is usually associated with microangiopathic hаemolytic anaemia and thrombocytopenia [1,3]. In cases of APS many pulmonary complications develop, including embolism and pulmonary infarction, pulmonary hypertension, pulmonary microthrombosis, intra-alveolar pulmonary haemorrhage, fibrous alveolitis, acute respiratory distress syndrome, idiopathic pulmonary fibrosis, postpartum syndrome [1,5,6].…”

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confidence: 99%

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Human umbilical cord-derived mesenchymal stem cells and nitric oxide modulators attenuate the proinflammatory cytokine pattern in experimental antiphospholipid syndrome in mice

Mekhno,

Dovgalyuk,

Korda

et al. 2024

COT

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Antiphospholipid syndrome is an autoimmune disease characterized by vascular thrombosis and/or obstetric pathology and the presence of antibodies against membrane phospholipids or certain phospholipid-related proteins. Objective. The aim of the research is to study the effect of mesenchymal stem cells, nitric oxide modulators (L-arginine and aminoguanidine) on the level of cytokines in bronchoalveolar lavage in experimental antiphospholipid syndrome in mice. Material and methods. Antiphospholipid syndrome was modeled on female BALB/c mice by intramuscular injections of cardiolipin 1.2 mg/kg 4 times with a 14-day interval. Cryopreserved human umbilical cord-derived mesenchymal stem cells (5×106 cells/kg) were injected once intraperitoneally, L-arginine (25 mg/kg) and aminoguanidine (10 mg/kg) were administered intraperitoneally 1 time per day during 10 days after APS had developed. The cytokines concentration in bronchoalveolar lavage from the lungs was assessed by means of ELISA in 10 days after APS development. Results. In the bronchoalveolar lavage of the BALB/c mice with experimental APS, an increased level of pro-inflammatory cytokines IL-1β, IL-6, TNF-α and decreased level of anti-inflammatory IL-4 and IL-10 were found. It was established that in cases of APS and administration of stem cells the concentration of proinflammatory cytokines decreased: IL-1β by 32.4 %, IL-6 by 30.6 % and TNF-α by 36.1 %, respectively, compare to the APS animals. At the same time the level of IL-4 increased by 50.5 % and IL-10 – by 57.5 % in the group of animals administered with stem cells compare to those with APS. Conclusion. In cases of correction of modeled antiphospholipid syndrome in mice using mesenchymal stem cells and combined application of mesenchymal stem cells and nitric oxide modulators (L-arginine and aminoguanidine), a decrease in the level of pro-inflammatory cytokines (IL-1β, IL-6, TNF-α ) and an increase in the level of anti-inflammatory cytokines (IL-4 and IL-10) in bronchoalveolar lavage has been established.

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Alveolar Hemorrhage, a Rare and Life-Threatening Complication of Catastrophic Antiphospholipid Syndrome

Cited by 5 publications

References 17 publications

Cases of catastrophic anti-phospholipid syndrome in systemic lupus erythematosus: An experience

Cases of catastrophic anti-phospholipid syndrome in systemic lupus erythematosus: An experience

Extensive Thrombosis in Catastrophic Antiphospholipid Syndrome in a Newly Diagnosed Systemic Lupus Erythematosus: A Case Report

Human umbilical cord-derived mesenchymal stem cells and nitric oxide modulators attenuate the proinflammatory cytokine pattern in experimental antiphospholipid syndrome in mice

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