Triple-Positive Antiphospholipid Syndrome with Splenic Infarction Triggered by Tocilizumab Withdrawal in A High-Titer ACPA-Positive Rheumatoid Arthritis Patient: An Unmasking Storm of Polyautoimmunity.
- 1Rheumatology Clinic, Department of Medical and Surgical Sciences, University of Foggia, Foggia, Italy.
Abstract
Introduction
We describe the occurrence of an uncommon thrombotic event, namely splenic infarction, as the onset of antiphospholipid syndrome (APS) after temporary suspension of anti-IL-6 treatment in a patient with rheumatoid arthritis (RA).Case description
A 53-year-old patient with anti-citrullinated protein antibody (ACPA) and rheumatoid factor-positive RA, previously showing transient positivity for immunoglobulin (Ig)G anticardiolipin antibodies and lupus anticoagulant (LAC), refractory to multiple biological disease-modifying anti-rheumatic drugs (DMARDs), anti-tumour necrosis factor (TNF)-α agents, and rituximab, had been treated with tocilizumab since 2013 with good clinical response. After temporary suspension of tocilizumab for minor orthopaedic surgery complicated by skin abscess, he developed acute abdominal pain and computed tomography evidence of splenic infarction. Laboratory evaluation revealed elevated inflammatory markers and triple positivity for LAC, aCL IgG, and aB2GPI IgG, leading to the diagnosis of APS. Treatment with warfarin, methotrexate, and baricitinib resulted in stable disease control without recurrent thrombotic events.Conclusion
Clinical stability in RA is a delicate balance between control of inflammation and avoidance of medication toxicity, especially in patients with high thrombotic risk and previous antiphospholipid positivity. Temporary withdrawal of biologic therapy may induce immunological rebound and thrombotic complications. This case highlights the importance of thrombotic risk stratification and multidisciplinary management when biologic therapies are suspended.Learning points
Tocilizumab may act as a suppressor of autoreactive B-cell clones producing antiphospholipid antibodies.A high anti-citrullinated protein antibody (ACPA) titer may be associated with an increased risk of developing autoantibody storm with possible prothrombotic complications in rheumatoid arthritis, such as antiphospholipid syndrome.In case of surgical interventions in patients with rheumatoid arthritis treated with biological disease-modifying anti-rheumatic drugs (bDMARDs) it is important to evaluate the stratification of thrombotic risk with multidisciplinary discussion before suspending therapy.
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BibTeX
@article{coviello2026triplepositive,
title = {{Triple-Positive Antiphospholipid Syndrome with Splenic Infarction Triggered by Tocilizumab Withdrawal in A High-Titer ACPA-Positive Rheumatoid Arthritis Patient: An Unmasking Storm of Polyautoimmunity.}},
author = {Francesco Elios Coviello and Cinzia Rotondo and Francesca D'Onofrio and Raffaele Barile and Francesco Paolo Cantatore and Addolorata Corrado},
journal = {European journal of case reports in internal medicine},
year = {2026},
month = oct,
doi = {10.12890/2026_007169},
url = {https://europepmc.org/article/MED/43011674},
note = {CC BY-NC-ND},
}
RIS
TY - JOUR
TI - Triple-Positive Antiphospholipid Syndrome with Splenic Infarction Triggered by Tocilizumab Withdrawal in A High-Titer ACPA-Positive Rheumatoid Arthritis Patient: An Unmasking Storm of Polyautoimmunity.
AU - Francesco Elios Coviello
AU - Cinzia Rotondo
AU - Francesca D'Onofrio
AU - Raffaele Barile
AU - Francesco Paolo Cantatore
AU - Addolorata Corrado
PY - 2026
DA - 2026/10/21
JO - European journal of case reports in internal medicine
DO - 10.12890/2026_007169
UR - https://europepmc.org/article/MED/43011674
AB - Introduction We describe the occurrence of an uncommon thrombotic event, namely splenic infarction, as the onset of antiphospholipid syndrome (APS) after temporary suspension of anti-IL-6 treatment in a patient with rheumatoid arthritis (RA).Case description A 53-year-old patient with anti-citrullinated protein antibody (ACPA) and rheumatoid factor-positive RA, previously showing transient positivity for immunoglobulin (Ig)G anticardiolipin antibodies and lupus anticoagulant (LAC), refractory to multiple biological disease-modifying anti-rheumatic drugs (DMARDs), anti-tumour necrosis factor (TNF)-α agents, and rituximab, had been treated with tocilizumab since 2013 with good clinical response. After temporary suspension of tocilizumab for minor orthopaedic surgery complicated by skin abscess, he developed acute abdominal pain and computed tomography evidence of splenic infarction. Laboratory evaluation revealed elevated inflammatory markers and triple positivity for LAC, aCL IgG, and aB2GPI IgG, leading to the diagnosis of APS. Treatment with warfarin, methotrexate, and baricitinib resulted in stable disease control without recurrent thrombotic events.Conclusion Clinical stability in RA is a delicate balance between control of inflammation and avoidance of medication toxicity, especially in patients with high thrombotic risk and previous antiphospholipid positivity. Temporary withdrawal of biologic therapy may induce immunological rebound and thrombotic complications. This case highlights the importance of thrombotic risk stratification and multidisciplinary management when biologic therapies are suspended.Learning points Tocilizumab may act as a suppressor of autoreactive B-cell clones producing antiphospholipid antibodies.A high anti-citrullinated protein antibody (ACPA) titer may be associated with an increased risk of developing autoantibody storm with possible prothrombotic complications in rheumatoid arthritis, such as antiphospholipid syndrome.In case of surgical interventions in patients with rheumatoid arthritis treated with biological disease-modifying anti-rheumatic drugs (bDMARDs) it is important to evaluate the stratification of thrombotic risk with multidisciplinary discussion before suspending therapy.
SN - 2284-2594
ER -