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Lupus 2022 Jun 23:9612033221108853. Epub 2022 Jun 23.

Department of Pediatric Rheumatology, Faculty of Medicine, 37515Hacettepe University, Ankara, Turkey.

Pediatric primary antiphospholipid syndrome (APS) is a very rare disease with significant distinctions from the APS in adults. Herein, we present our experience in the diagnosis and treatment of six pediatric primary APS patients, who met the updated Sapporo criteria for the APS diagnosis. One of them was also diagnosed as having probable catastrophic APS (CAPS) due to the involvement of three different organ systems simultaneously. Read More

Vaccines (Basel) 2022 Jun 16;10(6). Epub 2022 Jun 16.

Grupo Español de PTI, 28040 Madrid, Spain.

Worldwide vaccination against SARS-CoV-2 has allowed the detection of hematologic autoimmune complications. Adverse events (AEs) of this nature had been previously observed in association with other vaccines. The underlying mechanisms are not totally understood, although mimicry between viral and self-antigens plays a relevant role. Read More

Pathophysiology 2022 May 25;29(2):187-199. Epub 2022 May 25.

Department of Pathology, Faculty of Medicine, Saint Petersburg State University, Universitetskaya emb, 7-9, 199034 St. Petersburg, Russia.

Chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME) is considered to be associated with post-viral complications and mental stress, but the role of autoimmunity also remains promising. A comparison of autoimmune profiles in chronic fatigue of different origin may bring insights on the pathogenesis of this disease. Thirty-three patients with CFS/ME were divided into three subgroups. Read More

Respirol Case Rep 2022 Jul 13;10(7):e0990. Epub 2022 Jun 13.

Department of Rheumatology Eastern Health Box Hill Victoria Australia.

We describe the first case of antisynthetase syndrome (ASS) with antibodies to anti-glycyl tRNA synthetase (EJ-1) with antiphospholipid syndrome (APLS). A 66-year-old man presented with progressive dyspnoea, fever, dry cough and proximal muscle weakness over several months on a background of cryptogenic organizing pneumonia. Examination revealed bibasal fine chest crackles, proximal muscle weakness of the upper and lower limbs, digital skin thickening and facial telangiectasias. Read More

J Neurosurg Case Lessons 2022 May 23;3(21):CASE22127. Epub 2022 May 23.

Department of Neurosurgery, Brigham and Women's Hospital, Harvard Medical School, Boston, Massachusetts.

Background: Hypercoagulability with thrombosis and associated inflammation has been well-documented in COVID-19, and catastrophic cerebral venous sinus thromboses (CVSTs) have been described. Another COVID-19-related complication is bacterial superinfection, including sinusitis. Here, the authors reported three cases of COVID-19-associated sinusitis, meningitis, and CVST and summarized the literature about septic intracranial thrombotic events as a cause of headache and fever in COVID-19. Read More

Semin Thromb Hemost 2022 Jun 21. Epub 2022 Jun 21.

Department of Cardio-Thoracic-Vascular Sciences and Public Health, Thrombosis Research Laboratory, University of Padova, Padova, Italy.

Unprovoked (or provoked by mild risk factors) venous thromboembolism (VTE) in young patients, VTE in uncommon sites, or cases of unexplained VTE recurrence may be positive for antiphospholipid antibodies (aPL) and thus may be diagnosed with antiphospholipid syndrome (APS). The evaluation of aPL is standardized using immunological tests for anticardiolipin and anti-β2-glycoprotein I. The determination of functional antibodies (lupus anticoagulant) is less standardized, especially in patients on anticoagulant treatment. Read More

BMC Pregnancy Childbirth 2022 Jun 20;22(1):500. Epub 2022 Jun 20.

Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel.

Background: Preeclampsia is a multisystem disorder characterized by an abnormal vascular response to placentation associated with increased systemic vascular resistance. As liver involvement is one of the main clinical features of preeclampsia, we sought to determine if there is an association between chronic liver diseases and preeclampsia.

Methods: A retrospective matched case-control analysis was conducted in a tertiary medical center. Read More

J Thromb Haemost 2022 Jun 20. Epub 2022 Jun 20.

Department of Rheumatology, Mayo Clinic, Rochester, MN, USA.

Background: Anti-phosphatidylserine prothrombin antibodies (aPSPT) are reported to be highly associated with the lupus anticoagulant (LAC) in established APS and SLE cohorts. Further, aPSPT has been suggested to be a useful surrogate LAC marker. However, validation studies replicating this relationship in an all-comer study population in the diagnostic clinical setting are lacking. Read More

Ann Cardiol Angeiol (Paris) 2022 Jun 16. Epub 2022 Jun 16.

Service de Médecine Interne, Centre hospitalier Universitaire Joseph Raseta Befelatanana, Antananarivo, Madagascar.

Introduction: Venous thrombosis of unusual sites is much rarer than in the lower limbs and requires a rigorous etiological approach. The objective was to describe the clinical and progressive peculiarities of unusual localization venous thrombosis as well as their etiologies.

Patients And Methods: Multicenter descriptive retrospective study of hospitalized patient records in the two large Hospital Centers, Antananarivo, Madagascar between 2017 and 2020 in which the diagnosis of unusual venous thrombosis was confirmed by imaging. Read More

Int J Cardiol 2022 Jun 14. Epub 2022 Jun 14.

University Center of Excellence on Nephrologic, Rheumatologic and Rare Diseases (ERK-net, ERN-Reconnect and RITA-ERN Member) with Nephrology and Dialysis Unit and Center of Immuno-Rheumatology and Rare Diseases (CMID), San Giovanni Bosco Hub Hospital, and Department of Clinical and Biological Sciences of the University of Turin, Torino, Italy.; University Center of Excellence on Nephrologic, Rheumatologic and Rare Diseases (ERK-net, ERN-Reconnect and RITA-ERN Member) with Nephrology and Dialysis Unit and Center of Immuno-Rheumatology and Rare Diseases (CMID), San Giovanni Bosco Hub Hospital, and Department of Clinical and Biological Sciences of the University of Turin, Torino, Italy.. Electronic address:

Objectives: We aimed to apply and compare the QRISK3 and the adjusted Global AntiPhospholipid Syndrome (APS) Score (aGAPSS) in a cohort of systemic lupus erythematosus (SLE) patients, with and without a concomitant diagnosis of APS, in order to assess their augmented risk of developing cardiovascular diseases (CVDs).

Methods: Patients (25-85 yo) with a diagnosis of SLE and/or of Secondary APS (SAPS) were included. QRISK3 was calculated using the official online calculator; aGAPSS using the validated point-values based on aPL-profile and independent risk factors. Read More

J Thromb Haemost 2022 Jun 17. Epub 2022 Jun 17.

Coagulation Laboratory, Ghent University Hospital, Ghent, Belgium.

Background: Diagnosis of antiphospholipid syndrome (APS) requires persistent presence of lupus anticoagulant (LAC), anticardiolipin (aCL) IgG/IgM, or anti-β2 glycoprotein I (aβ2GPI) IgG/IgM antibodies. Other antiphospholipid antibodies (aPL) such as antiphosphatidylserine/prothrombin antibodies (aPS/PT) are promising in assessment of thrombotic APS (TAPS).

Aim: To evaluate the added value of aPS/PT IgG and IgM in TAPS. Read More

Cureus 2022 May 15;14(5):e25008. Epub 2022 May 15.

General Medicine, Jawaharlal Institute of Postgraduate Medical Education and Research, Pondicherry, IND.

Scrub typhus is an acute febrile disease caused by with a clinical course varying from mild to fatal. Vascular thrombosis is rare in scrub typhus. There is an increased risk of thrombotic events in Coronavirus disease 19 (COVID-19). Read More

Ultrastruct Pathol 2022 Jun 16:1-11. Epub 2022 Jun 16.

Department of Pathology and Laboratory Medicine, Division of Renal Pathology and Electron Microscopy, University of Rochester Medical Center, Rochester, NY, USA.

Rare cases of membranous glomerulopathy (MGN) with subepithelial deposits consisting of microspherular structures identified by electron microscopy have been described in the literature as either MGN with spherules or podocyte infolding glomerulopathy (PIG). The paucity of available studies shows a strong association with underlying autoimmune disease. To further understand the significance of subepithelial microspherular deposits, we retrospectively identified native kidney biopsies from 10 patients diagnosed as MGN with subepithelial microspherular structures identified by ultrastructural examination at the University of Rochester Medical Center (URMC) during an 11-year period. Read More

Vasc Health Risk Manag 2022 10;18:359-373. Epub 2022 May 10.

Therapeutic and Vascular Medicine Department, University Hospital of Saint Etienne, Saint Etienne, France.

During the past decade, emergence of direct oral anticoagulants (DOACs) has drastically improved the prevention of thrombosis. However, several unmet needs prevail in the field of thrombosis prevention, even in the DOACs' era. The use of DOACs is still constrained and the drugs cannot be administered in every clinical scenario, such as an increased anticoagulant-associated bleeding risk, particularly in some specific populations (cancer - notably those with gastrointestinal or genitourinary cancer - and frail patients), the impossibility to be used in certain patients (eg, end-stage kidney failure during hemodialysis, pregnancy and breastfeeding), and their lack of efficacy in certain clinical scenarios (eg, mechanical heart valves, triple-positive antiphospholipid syndrome). Read More

Rheumatol Ther 2022 Jun 15. Epub 2022 Jun 15.

Angelo Bianchi Bonomi Hemophilia and Thrombosis Center, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milan, Italy.

Thrombocytopenia is a common feature of antiphospholipid syndrome (APS) and rarely requires treatment. Here we present the case of a 71-year-old man hospitalized for severe immune thrombocytopenia (ITP) secondary to APS and concomitant SARS-CoV-2 infection. The patient was successfully treated with systemic corticosteroids, intravenous immunoglobulins, and plasma exchange (PEX). Read More

Expert Rev Clin Immunol 2022 Jun 19:1-3. Epub 2022 Jun 19.

Women & Children's Health, King's College London, UK.

Lupus Sci Med 2022 06;9(1)

Rheumatology, Barbara Volcker Center for Women and Rheumatic Diseases, Hospital for Special Surgery, Weill Cornell Medicine, New York, New York, USA

Objectives: To describe the outcomes of pregnancies in antiphospholipid antibody (aPL)-positive patients since the inception of the AntiPhospholipid Syndrome Alliance for Clinical Trials and InternatiOnal Networking Registry.

Methods: We identified persistently aPL-positive patients recorded as 'pregnant' during prospective follow-up, and defined as a composite of: (1) Preterm live delivery (PTLD) at or before 37th week due to pre-eclampsia (PEC), eclampsia, small-for-gestational age (SGA) and/or placental insufficiency (PI); or (2) Otherwise unexplained fetal death after the 10th week of gestation. The primary objective was to describe the characteristics of patients with and without aPL-related composite outcomes based on their first observed pregnancies following registry recruitment. Read More

J Clin Rheumatol 2022 Jun 13. Epub 2022 Jun 13.

Pulmonology, University Clinical Center of Serbia.

Background: Antiphospholipid syndrome (APS) is characterized by antiphospholipid antibodies (aPLs) associated with thrombosis (arterial and/or venous) and/or obstetrical manifestations. However, various manifestations, which are considered to be noncriteria manifestations, are frequently found in APS.

Aim: The purpose of this study was to evaluate whether noncriteria manifestations may be found more frequently in subjects with thrombotic and/or obstetrical APS ("criteria" manifestations) in a population of patients with primary APS (PAPS). Read More

Arthritis Res Ther 2022 Jun 11;24(1):140. Epub 2022 Jun 11.

Department of Rheumatology and Clinical Immunology, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China.

Background: Antiphospholipid syndrome (APS) is an autoimmune disease characterized by persistent antiphospholipid antibodies (aPLs) positivity with a wide manifestation spectrum. A risk stratification is needed for management guidance and prognosis assessment. We aimed to identify phenotypes among aPL-positive patients and assess the prognosis of each phenotype. Read More

Autoimmun Rev 2022 Jun 9;21(8):103130. Epub 2022 Jun 9.

Department of Laboratory Medicine, Peking University Third Hospital, Beijing, 100191, China; Core Unit of National Clinical Research Center for Laboratory Medicine, Peking University Third Hospital, Beijing 100191, China. Electronic address:

APS (antiphospholipid syndrome) is a systematic autoimmune disease accompanied with venous or arterial thrombosis and poor pregnant manifestations, partly attributing to the successive elevated aPL (antiphospholipid antibodies) and provoked prothrombotic and proinflammatory molecules production. Nowadays, most researches focus on the laboratory detection and clinic features of APS, but its precise etiology remains to be deeply explored. As we all know, the dysfunction of ECs (endothelial cells), monocytes, platelets, trophoblasts and neutrophils are key contributors to APS progression. Read More

Rheumatology (Oxford) 2022 Jun 10. Epub 2022 Jun 10.

MRC Biostatistics Unit, Cambridge, UK.

Objective: To determine whether BILAG-2004 index is associated with the development of damage in a cohort of SLE patients. Mortality and development of damage were examined.

Methods: This was a multicentre longitudinal study. Read More

Rheumatology (Oxford) 2022 Jun 10. Epub 2022 Jun 10.

Department of internal medicine, Reims university hospital, Rue du Général Koenig, 51100 Reims, France.

Introduction: Data on severe heart valve disease (HVD), including Libman-sacks endocarditis, associated with systemic lupus erythematosus (SLE) and/or antiphospholipid syndrome (APS) requiring valvular surgery are scarce.

Method: Observational retrospective multicentric analysis of 23 adults with SLE and/or APS and HVD, between 1996 and 2019 and available histopathological report evaluating long-term follow-up.

Results: Twenty-three individuals (20 females, median age 37 [range 17-76]) were included. Read More

Lupus 2022 Jun 10:9612033221107583. Epub 2022 Jun 10.

Department of Autoimmune Diseases, Hospital Clínic, Barcelona, Spain.

Objectives: To explore the prevalence and clinical significance of low complement levels in patients with catastrophic antiphospholipid syndrome (CAPS).

Methods: We reviewed data from the "CAPS Registry" on C3 and/or C4 complement plasma protein levels during acute CAPS episodes. Patients were classified into those with low and normal complement levels. Read More

Int J Clin Pract 2022 31;2022:4308470. Epub 2022 Jan 31.

Department of Obstetrics and Gynecology, West China Second University Hospital of Sichuan University, Chengdu, China.

Objective: Antiphospholipid syndrome (APS) is a chronic autoimmune disease with a high prevalence in females. Published data have identified pregnant women with APS may suffer from recurrent miscarriage, fetal death. However, the association between antiphospholipid antibody (aPL) and fetal growth restriction (FGR) remains controversial. Read More

Cureus 2022 Jun 4;14(6):e25659. Epub 2022 Jun 4.

Internal Medicine, Monmouth Medical Center, Long Branch, USA.

Venous thromboembolism includes deep venous thrombosis (DVT) and pulmonary embolism and is the most common cardiovascular disease after coronary artery disease and stroke. Antiphospholipid syndrome (APS) is an autoimmune disorder that is characterized by venous or arterial thrombosis with laboratory evidence of antiphospholipid antibodies. Long-term anticoagulation therapy is required to prevent recurrent DVTs, embolisms, and thrombosis-related complications. Read More

Lupus 2022 Jun 8:9612033221107538. Epub 2022 Jun 8.

Department of Rheumatology, 11297Aarhus University Hospital, Aarhus, Denmark.

Objectives: Over the past decades new international guidelines recommend that pregnant Systemic lupus erythematosus (SLE) patients are monitored closely in a multi-professional team throughout pregnancy. The importance of low disease activity before pregnancy and continued treatment during pregnancy has been established. However, there is still a high risk of adverse pregnancy outcome (APO). Read More

Semin Thromb Hemost 2022 Jun 8. Epub 2022 Jun 8.

Department of Diagnostic Sciences, Coagulation Laboratory, Ghent University Hospital, Ghent University, Ghent, Belgium.

The diagnosis of antiphospholipid syndrome (APS) relies on the detection of circulating antiphospholipid antibodies (aPL). Currently, lupus anticoagulant (LA), anticardiolipin (aCL), and anti-β2-glycoprotein I antibodies (aβ2GPI) IgG or IgM are the laboratory criteria if persistently present over time. As aCL and aβ2GPI are two out of the three laboratory criteria, the detection of aPL by solid phase assays is an essential step in the diagnosis of APS. Read More

Acta Biomed 2022 Jun 7;93(S1):e2022107. Epub 2022 Jun 7.

Clinical Neurology, Udine University Hospital, Udine, Italy.

SARS-CoV-2 infection is associated with an increased risk of thrombotic events, especially during severe forms of disease. Here we describe the clinical history of a patient with a mild form of Covid-19 infection presenting with multiple cerebral ischemic lesions that evolved in an atypical way. Read More

J Clin Lab Anal 2022 Jun 6:e24537. Epub 2022 Jun 6.

Laboratory of Immunology, Farhat Hached Hospital, Sousse, Tunisia.

Aim: To determine the frequency of serological markers of RA in patients with anti-β2 glycoprotein I antibodies (aβ2GPI) of IgA isotype.

Material And Methods: A retrospective study was conducted on 67 patients with aβ2GPI-IgA. Ninety healthy blood donors (HBD) were used as a control group. Read More

Eur Heart J Case Rep 2022 May 11;6(5):ytac199. Epub 2022 May 11.

Internal Medicine Department, Cardiology Division, Texas Tech University Health Sciences Center, Lubbock, TX, USA.

Background: Antiphospholipid syndrome (APS) is an autoimmune response characterized clinically by arterial or venous thrombosis. One of the rare and series forms of APS is the catastrophic APS (CAPS). The incidence of CAPS has been reported in 0. Read More