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Article: Catastrophic antiphospholipid syndrome treated with rituximab: A case report.

Doğru, Atalay / Ugan, Yunus / Şahin, Mehmet / Karahan, Nermin / Tunç, Şevket Ercan

2017 Volume 4, Issue 2, Page(s) 145–147

Abstract: ... antiphospholipid antibodies. Here we report the case of a 19-year-old woman with CAPS. During the first week ... Catastrophic antiphospholipid syndrome (CAPS) is a rare and fatal condition that is characterized ... venous sinus thrombosis, and positive lupus anticoagulant. She was treated with corticosteroids ...

Abstract	Catastrophic antiphospholipid syndrome (CAPS) is a rare and fatal condition that is characterized by diffuse venous and/or arterial thromboembolism within a short period of time and histopathological confirmation of small-vessel occlusion in at least one organ or tissue in the presence of positive antiphospholipid antibodies. Here we report the case of a 19-year-old woman with CAPS. During the first week of her hospitalization, she was diagnosed with CAPS on the basis of skin necrosis, pulmonary artery thrombosis, cerebral venous sinus thrombosis, and positive lupus anticoagulant. She was treated with corticosteroids, intravenous immunoglobulins, plasmapheresis, and anticoagulants. Forty days after the onset of CAPS, cutaneous lesions were recurred during skin surgery. She required a high dose of corticosteroids, intravenous immunoglobulins, and rituximab. No further thrombotic events occurred. Rituximab may be an effective treatment option for patients with CAPS.
Language	English
Publishing date	2017-06-01
Publishing country	Turkey
Document type	Journal Article
ZDB-ID	2873727-1
ISSN	2148-4279 ; 2147-9720
ISSN (online)	2148-4279
ISSN	2147-9720
DOI	10.5152/eurjrheum.2017.160073
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Article ; Online: Successful Treatment of Catastrophic Antiphospholipid Syndrome Using Rituximab: Case Report and Review of the Literature.

Stanescu, Cristina / Andronesi, Andreea Gabriella / Jurcut, Ciprian / Gherghiceanu, Mihaela / Vornicu, Alexandra / Burcea, Florentina Andreea / Andronesi, Toader Danut / Lupusoru, Gabriela Elena / Iliuta, Luminita / Sorohan, Bogdan Marian / Obrisca, Bogdan / Ismail, Gener

Medicina (Kaunas, Lithuania)

2021 Volume 57, Issue 9

Abstract: ... involvement. A diagnosis of probable catastrophic antiphospholipid syndrome (CAPS) was established and ... TMA) secondary to antiphospholipid autoantibodies (aPL). Association between antiphospholipid syndrome ... treatment with low molecular weight heparin, intravenous methylprednisolone, plasmapheresis, and rituximab ...

Abstract	Background: Kidney involvement is a frequent complication of systemic lupus erythematosus (SLE) and kidney biopsy is essential in differentiating lupus nephritis (LN) from thrombotic microangiopathy (TMA) secondary to antiphospholipid autoantibodies (aPL). Association between antiphospholipid syndrome (APS) and acquired hemophilia due to inhibitors was very rarely described in SLE patients. Case presentation: We present the case of a 61-year-old male diagnosed with SLE who acquired deficiency of clotting factor VIII due to circulating inhibitors, admitted for acute kidney injury (AKI), microangiopathic hemolytic anemia, thrombocytopenia, and diplopia. Kidney biopsy showed TMA due to APS, but no signs of LN. Head computed tomography identified low dense areas in the white matter, suggesting small blood vessels' involvement. A diagnosis of probable catastrophic antiphospholipid syndrome (CAPS) was established and treatment with low molecular weight heparin, intravenous methylprednisolone, plasmapheresis, and rituximab was initiated, followed by resolution of AKI, diplopia, and TMA with complete depletion of CD19+B-lymphocytes (CD19+B-Ly) after one month. We further review the current knowledge regarding pathogenesis and management of CAPS in SLE patients. Conclusions: Targeted therapy was possible after kidney biopsy, improving renal and general prognosis. CD19+B-Ly repopulation preceded biological relapse, so monitoring of CD19+B-Ly may serve as a tool to predict relapses and guide rituximab therapy.
MeSH term(s)	Antibodies, Antiphospholipid ; Antiphospholipid Syndrome/complications ; Antiphospholipid Syndrome/diagnosis ; Antiphospholipid Syndrome/drug therapy ; Humans ; Lupus Erythematosus, Systemic/complications ; Lupus Erythematosus, Systemic/drug therapy ; Male ; Middle Aged ; Neoplasm Recurrence, Local ; Rituximab/therapeutic use
Chemical Substances	Antibodies, Antiphospholipid ; Rituximab (4F4X42SYQ6)
Language	English
Publishing date	2021-08-31
Publishing country	Switzerland
Document type	Case Reports ; Review
ZDB-ID	2188113-3
ISSN	1648-9144 ; 1010-660X
ISSN (online)	1648-9144
ISSN	1010-660X
DOI	10.3390/medicina57090912
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Article: Catastrophic antiphospholipid syndrome associated with systemic lupus erythematosus treated with rituximab: case report and a review of the literature.

Sukara, G / Baresic, M / Sentic, M / Brcic, L / Anic, B

Acta reumatologica portuguesa

2015 Volume 40, Issue 2, Page(s) 169–175

Abstract: ... this is the first ever reported case of definitive CAPS associated with SLE treated with rituximab. There ... Catastrophic antiphospholipid syndrome (CAPS) is a rare, acute, life-threatening form ... of antiphospholipid syndrome. In the last several decades there has been a significant improvement in the treatment of patients ...

Abstract	Catastrophic antiphospholipid syndrome (CAPS) is a rare, acute, life-threatening form of antiphospholipid syndrome. In the last several decades there has been a significant improvement in the treatment of patients with CAPS, but the overall mortality is still very significant. The use of rituximab has been reported in the treatment of refractory cases of CAPS but the data are still scarce and inconclusive. We report a case of 47-year old male patient with long standing SLE and secondary APS who presented with acute thromboembolic incident (partial thrombosis of superior mesenteric artery). During the first week of his hospitalization he met the criteria for probable CAPS. He was treated with anticoagulants, glucocorticoids, intravenous immunoglobulins and systemic antibiotics. Finally he was treated with rituximab. There was no response to the implemented treatment and he eventually died. Autopsy showed evidence of small vessel thrombosis in the lung microvasculature. With this the criteria for definitive CAPS were fulfilled. To our knowledge, at present time, this is the first ever reported case of definitive CAPS associated with SLE treated with rituximab. There is a great need for further investigation to evaluate the effectiveness of rituximab in treatment of CAPS.
MeSH term(s)	Antiphospholipid Syndrome/complications ; Antiphospholipid Syndrome/drug therapy ; Humans ; Immunologic Factors/therapeutic use ; Lupus Erythematosus, Systemic/complications ; Male ; Middle Aged ; Rituximab/therapeutic use
Chemical Substances	Immunologic Factors ; Rituximab (4F4X42SYQ6)
Language	English
Publishing date	2015-04
Publishing country	Portugal
Document type	Case Reports ; Journal Article ; Review
ZDB-ID	442031-7
ISSN	0303-464X
ISSN	0303-464X
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Article: Catastrophic Antiphospholipid Syndrome: A Complex Diagnosis in the Setting of Lupus.

Liang, Jessica / Mahmood, Raai / Benchaala, Ilyes / York, Russel / Sarakbi, Housam

Cureus

2023 Volume 15, Issue 8, Page(s) e42922

Abstract: This case report aims to highlight the importance of keeping catastrophic antiphospholipid syndrome ... to understand the workup and treatment of the disease. Catastrophic antiphospholipid syndrome is a life ... bilateral leg swelling that eventually progressed to catastrophic antiphospholipid syndrome. She developed ...

Abstract	This case report aims to highlight the importance of keeping catastrophic antiphospholipid syndrome (CAPS) high on the list of differentials in patients with lupus who present with digital ischemia and to understand the workup and treatment of the disease. Catastrophic antiphospholipid syndrome is a life-threatening variant of antiphospholipid syndrome (APS), and it is distinguished on the APS spectrum by its increased intensity and extent of thrombotic outcomes. Less than 1% of patients with APS develop CAPS and the demographic of patients affected are primarily females, 37 ± 14 years old, and have underlying primary APS or systemic lupus erythematosus (SLE). This is the case of a young female with lupus and end-stage renal disease secondary to lupus nephritis who presented to the emergency department for shortness of breath and bilateral leg swelling that eventually progressed to catastrophic antiphospholipid syndrome. She developed pulmonary embolisms, axillary hematoma, and bilateral lower extremity digital gangrene. The treatment course consisted of anticoagulation, steroids, intravenous immunoglobulin (IVIG), above-knee amputation, and eventually rituximab. Diagnosis and treatment of digital ischemia can be complex, especially, in the setting of lupus where the differential diagnosis is broad. A high index of suspicion for CAPS is essential for early diagnosis and treatment.
Language	English
Publishing date	2023-08-03
Publishing country	United States
Document type	Case Reports
ZDB-ID	2747273-5
ISSN	2168-8184
ISSN	2168-8184
DOI	10.7759/cureus.42922
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Article ; Online: A Case of Catastrophic Antiphospholipid Syndrome with Acute Multiorgan Thrombosis and Concerns for Re-Emergence.

Mittal, Sushmita / Chaudhary, Fizah S / Aung, Tun Tun / Babkir, Akram

The American journal of case reports

2024 Volume 25, Page(s) e942339

Abstract: BACKGROUND Catastrophic antiphospholipid syndrome (CAPS) is a rare, life-threatening form ... antiphospholipid syndrome, a true rheumatological emergency that requires aggressive treatment to prevent irreversible ... of antiphospholipid syndrome characterized by widespread thrombotic complications leading to multiorgan ischemia and failure ...

Abstract	BACKGROUND Catastrophic antiphospholipid syndrome (CAPS) is a rare, life-threatening form of antiphospholipid syndrome characterized by widespread thrombotic complications leading to multiorgan ischemia and failure. Although there are no standard treatment guidelines for CAPS, it often involves triple therapy with anticoagulation, corticosteroids, and plasma exchange. Recently, biologics such as rituximab and eculizumab have also shown promise as potential new therapies for CAPS, as observed in our case. CASE REPORT We describe a 59-year-old female patient who presented with altered mental status and diffuse weakness. Imaging studies revealed multiorgan thrombosis along with thrombocytopenia that markedly improved with plasma exchange therapy, steroids, and a heparin drip. While the exact etiology of CAPS remained unknown, it was likely precipitated by her warfarin discontinuation and confirmed Haemophilus influenzae infection. The patient's hospital course was complicated by hemorrhagic shock after a renal biopsy, followed by an acute drop in thrombocytopenia and new embolic infarcts in the brain that raised concern for CAPS re-emergence. To address the refractory nature of her condition, the patient underwent a trial of rituximab, which remarkably improved her clinical picture and platelet count by an 8-fold increase within 1 week. CONCLUSIONS This case highlights the importance of early recognition and diagnosis of catastrophic antiphospholipid syndrome, a true rheumatological emergency that requires aggressive treatment to prevent irreversible complications. Our patient's presentation and response to treatment also underscores the complexity of managing CAPS and the use of newer biological therapies in refractory cases.
MeSH term(s)	Female ; Humans ; Middle Aged ; Antiphospholipid Syndrome/complications ; Antiphospholipid Syndrome/diagnosis ; Rituximab/therapeutic use ; Thrombosis/etiology ; Heparin/therapeutic use ; Thrombocytopenia/complications
Chemical Substances	Rituximab (4F4X42SYQ6) ; Heparin (9005-49-6)
Language	English
Publishing date	2024-01-28
Publishing country	United States
Document type	Case Reports ; Journal Article
ZDB-ID	2517183-5
ISSN	1941-5923 ; 1941-5923
ISSN (online)	1941-5923
ISSN	1941-5923
DOI	10.12659/AJCR.942339
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Article ; Online: Catastrophic antiphospholipid syndrome in lupus-associated immune thrombocytopenia treated with eltrombopag A case series and literature review.

Garra, Wakar / Carmi, Or / Kivity, Shaye / Levy, Yair

Medicine

2023 Volume 102, Issue 6, Page(s) e32949

Abstract: ... treated for refractory ITP with eltrombopag. Both developed catastrophic antiphospholipid syndrome ... a severe possible side-effect of eltrombopag as a trigger of catastrophic antiphospholipid syndrome, a rare ... treated with solumedrol, plasmapheresis, anticoagulation and rituximab.: Conclusions: We describe ...

Abstract	Background: Eltrombopag, a thrombopoietin receptor (TPO-R) agonist, is considered a second-line treatment for patients with refractory immune thrombocytopenia (ITP). Systemic lupus erythematosus (SLE) is frequently associated with ITP. In some cases, thrombocytopenia in SLE patients is attributed to concurrent antiphospholipid antibodies (APLA). Currently, data regarding treatment with TPO-R agonists for ITP in SLE or APLA patients are limited. The incidence of SLE flare or antiphospholipid syndrome while on TPO-R agonists has not been well-studied. Cases: We report 2 cases of female patients with SLE and concurrent triple positive APLA, without thrombotic events in their medical history, in our rheumatology clinic, who were treated for refractory ITP with eltrombopag. Both developed catastrophic antiphospholipid syndrome a few weeks after beginning treatment with eltrombopag. They were admitted to the intensive care unit and treated with solumedrol, plasmapheresis, anticoagulation and rituximab. Conclusions: We describe a severe possible side-effect of eltrombopag as a trigger of catastrophic antiphospholipid syndrome, a rare initial manifestation of antiphospholipid syndrome, in SLE patients with APLA. We suggest that APLA should be tested before initiating eltrombopag in patients with SLE-associated ITP. The safety of this treatment should be considered in these cases.
MeSH term(s)	Humans ; Female ; Purpura, Thrombocytopenic, Idiopathic/complications ; Purpura, Thrombocytopenic, Idiopathic/drug therapy ; Antiphospholipid Syndrome/complications ; Antiphospholipid Syndrome/drug therapy ; Antiphospholipid Syndrome/chemically induced ; Thrombocytopenia/etiology ; Thrombocytopenia/chemically induced ; Benzoates/therapeutic use ; Hydrazines/therapeutic use ; Lupus Erythematosus, Systemic/complications ; Lupus Erythematosus, Systemic/drug therapy ; Antibodies, Antiphospholipid
Chemical Substances	eltrombopag (S56D65XJ9G) ; Benzoates ; Hydrazines ; Antibodies, Antiphospholipid
Language	English
Publishing date	2023-01-05
Publishing country	United States
Document type	Review ; Journal Article
ZDB-ID	80184-7
ISSN	1536-5964 ; 0025-7974
ISSN (online)	1536-5964
ISSN	0025-7974
DOI	10.1097/MD.0000000000032949
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Article: A Rare Case of Refractory Catastrophic Antiphospholipid Syndrome Successfully Treated With Rituximab and Plasma Exchange.

Bangolo, Ayrton / Sagireddy, Sowmya / Mahamadeen, Sarah / Hasta, Felicia / Reddy, Sadhu A / Naz, Afshan / Ranganatha, Ravishankar / Ricketts, Cleveland / Muppalla, Padmavathi / Veliginti, Swathi / Arana, Georgemar / Sathyarajan, Dily T / Singh, Sachin / Shetty, Tanvi / Bhardwaj, Kshitij / Hashemy, Sayed / Duran, Roberto L / Kim, Sung H / Hipolito, Candice M /

Yoon, Kibo / Patel, Vrusha / Alshimari, Aseel / Inban, Pugazhendi / Yasmeen, Saaniya / Weissman, Simcha

Journal of community hospital internal medicine perspectives

2023 Volume 13, Issue 3, Page(s) 79–82

Abstract: ... with Plasmapheresis and Rituximab. With this case report, we hope to encourage the usage of Rituximab ... A small subset of patients with antiphospholipid syndrome (APS) may develop widespread thrombotic ... of refractory cases of CAPS. However, monoclonal antibodies such as rituximab and eculizumab have shown ...

Abstract	A small subset of patients with antiphospholipid syndrome (APS) may develop widespread thrombotic disease with organ damage, referred to as catastrophic APS (CAPS) that is associated with a high mortality. Medical therapy typically involves a combination of anticoagulation, systemic glucocorticoids, plasmapheresis, and intravenous immune globulin (IVIG). There is currently no consensus for the management of refractory cases of CAPS. However, monoclonal antibodies such as rituximab and eculizumab have shown some benefits. Herein, we present a 29-year-old female with previous pulmonary embolism who presented with necrotic left toes and was eventually diagnosed with refractory CAPS, successfully treated with Plasmapheresis and Rituximab. With this case report, we hope to encourage the usage of Rituximab in the management of CAPS.
Language	English
Publishing date	2023-05-08
Publishing country	United States
Document type	Case Reports
ZDB-ID	2616884-4
ISSN	2000-9666
ISSN	2000-9666
DOI	10.55729/2000-9666.1175
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Article: Primary Refractory Catastrophic Antiphospholipid Syndrome Masquerading as Buerger's Disease.

Tannous, Toufic / Rosso, Claudia / Iannuccilli, Jenna / Tannous, Karim / Keating, Matthew

Cureus

2021 Volume 13, Issue 4, Page(s) e14350

Abstract: Catastrophic antiphospholipid syndrome (CAPS) is a lethal disease with up to 30% mortality rate ... cases, and eculizumab, a drug approved for use in atypical hemolytic uremic syndrome, has demonstrated ... disease control in a few case reports. We describe a unique case of primary refractory CAPS with an unusual ...

Abstract	Catastrophic antiphospholipid syndrome (CAPS) is a lethal disease with up to 30% mortality rate. It can occur as a primary disease or secondary to an underlying autoimmune disease. Current treatment focuses on disease control with anticoagulation and steroids. Plasma exchange and intravenous immunoglobulin (IVIG) have shown some benefit when added. Monoclonal drugs such as rituximab have shown some benefit in refractory cases, and eculizumab, a drug approved for use in atypical hemolytic uremic syndrome, has demonstrated disease control in a few case reports. We describe a unique case of primary refractory CAPS with an unusual presentation that was treated with five lines of therapy before disease control was established.
Language	English
Publishing date	2021-04-07
Publishing country	United States
Document type	Case Reports
ZDB-ID	2747273-5
ISSN	2168-8184
ISSN	2168-8184
DOI	10.7759/cureus.14350
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Article: A catastrophic seronegative anti-phospholipid syndrome: case and literature review.

Pinto, Vanda / Ministro, Augusto / Carreira, Nuno Reis / Cardoso, Ana / Gonçalves, Catarina Sousa / Henriques, Mickael / Rato, João / Silva, Emanuel / Pedro, Luís Mendes

Thrombosis journal

2021 Volume 19, Issue 1, Page(s) 103

Abstract: ... phospholipid antibodies, this case hardly fits into another diagnosis. Therefore, it was treated ... to catastrophic APS, a rare form characterized by acute multiorgan thrombosis and high mortality.: Case report ... Background: Antiphospholipid Syndrome (APS) is a multisystemic autoimmune disease characterized ...

Abstract	Background: Antiphospholipid Syndrome (APS) is a multisystemic autoimmune disease characterized by arterial and venous thrombosis and / or obstetric morbidity in the presence of at least one circulating anti-phospholipid antibody. The spectrum of vascular events varies from deep venous thrombosis to catastrophic APS, a rare form characterized by acute multiorgan thrombosis and high mortality. Case report: We present the case of a 32-week pregnant woman arriving in the hospital emergency room with bilateral acute lower limb ischemia. In the obstetric evaluation, fetal death was declared. Computerized Tomography angiography showed pulmonary embolism of both pulmonary arteries, areas of splenic and right renal infarction and multiple arterial and venous thrombosis. The patient underwent urgent caesarean section and axillary-bifemoral bypass. No events registered. In the postoperative period, in an intensive care unit, treatment with rituximab and plasmapheresis were added to anticoagulant therapy. The laboratorial investigation was negative for thrombophilia and autoimmune diseases. Conclusion: Catastrophic APS develops quickly, with multiorgan involvement and high mortality rate. The presented case poses a multidisciplinary challenge, with the surgical approach of extra-anatomical revascularization being less invasive and guaranteeing immediate perfusion of the lower limbs. Although the serological tests were negative for anti-phospholipid antibodies, this case hardly fits into another diagnosis. Therefore, it was treated as a catastrophic APS, having shown a favorable evolution.
Language	English
Publishing date	2021-12-20
Publishing country	England
Document type	Journal Article
ISSN	1477-9560
ISSN	1477-9560
DOI	10.1186/s12959-021-00356-w
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Article ; Online: Successful Treatment of Catastrophic Antiphospholipid Syndrome Using Rituximab

Cristina Stanescu / Andreea Gabriella Andronesi / Ciprian Jurcut / Mihaela Gherghiceanu / Alexandra Vornicu / Florentina Andreea Burcea / Toader Danut Andronesi / Gabriela Elena Lupusoru / Luminita Iliuta / Bogdan Marian Sorohan / Bogdan Obrisca / Gener Ismail

Medicina, Vol 57, Iss 912, p

Case Report and Review of the Literature

2021 Volume 912

Abstract: ... involvement. A diagnosis of probable catastrophic antiphospholipid syndrome (CAPS) was established and ... secondary to antiphospholipid autoantibodies (aPL). Association between antiphospholipid syndrome (APS) and ... treatment with low molecular weight heparin, intravenous methylprednisolone, plasmapheresis, and rituximab ...

Abstract	Background: Kidney involvement is a frequent complication of systemic lupus erythematosus (SLE) and kidney biopsy is essential in differentiating lupus nephritis (LN) from thrombotic microangiopathy (TMA) secondary to antiphospholipid autoantibodies (aPL). Association between antiphospholipid syndrome (APS) and acquired hemophilia due to inhibitors was very rarely described in SLE patients. Case presentation: We present the case of a 61-year-old male diagnosed with SLE who acquired deficiency of clotting factor VIII due to circulating inhibitors, admitted for acute kidney injury (AKI), microangiopathic hemolytic anemia, thrombocytopenia, and diplopia. Kidney biopsy showed TMA due to APS, but no signs of LN. Head computed tomography identified low dense areas in the white matter, suggesting small blood vessels’ involvement. A diagnosis of probable catastrophic antiphospholipid syndrome (CAPS) was established and treatment with low molecular weight heparin, intravenous methylprednisolone, plasmapheresis, and rituximab was initiated, followed by resolution of AKI, diplopia, and TMA with complete depletion of CD19+B-lymphocytes (CD19+B-Ly) after one month. We further review the current knowledge regarding pathogenesis and management of CAPS in SLE patients. Conclusions: Targeted therapy was possible after kidney biopsy, improving renal and general prognosis. CD19+B-Ly repopulation preceded biological relapse, so monitoring of CD19+B-Ly may serve as a tool to predict relapses and guide rituximab therapy.
Keywords	lupus ; catastrophic antiphospholipid syndrome ; rituximab ; thrombotic microangiopathy ; case report ; Medicine (General) ; R5-920
Subject code	616 ; 610
Language	English
Publishing date	2021-08-01T00:00:00Z
Publisher	MDPI AG
Document type	Article ; Online
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