Case Report

Therapeutic challenges after successful
thrombectomy in a patient with an
antiphospholipid syndrome associated
M1-occlusion: A case report

Interventional Neuroradiology
0(00) 1–5
! The Author(s) 2015
Reprints and permissions:
sagepub.co.uk/journalsPermissions.nav
DOI: 10.1177/1591019915590371
ine.sagepub.com

Katharina Stadler1, Johannes S Mutzenbach1, Gudrun Kalss1,
Johann Sellner1, Abdul R Al-Schameri2, Eugen Trinka1 and
Monika Killer-Oberpfalzer3

Abstract
Backround: Stroke is a frequent disorder in patients with an antiphospholipid syndrome (APS). Due to a high risk for further
thromboembolic events, appropriate anticoagulation therapy in patients with an APS-associated stroke seems mandatory
but drug eluting and duration is a matter of debate.
Case: A 48-year-old female patient presented with Broca’s aphasia and mild hemiparesis on the right side. Diagnostic workup revealed left middle cerebral artery (MCA) occlusion yet without diffusion-weighted lesions. Due to a thrombocytopenia
(67.00 g/l) systemic thrombolysis was not indicated and endovascular treatment was initiated 150 min after symptom onset.
After successful clot retrieval, recurrent re-occlusions lead to the necessity of stent implantation and anticoagulation,
respectively. On day 5 she developed a new severe right-sided hemiparesis. The magnetic resonance imaging (MRI)
showed a subtotal restenosis of the left MCA despite the regular anticoagulation regime leading to a new left MCA ischaemic
stroke. In the meantime, the unknown aetiology, the patients’ age and the thrombocytopenia let to further diagnostic
workup. Elevated blood parameters such as lupus anticoagulant (LA)-1, LA-ratio, positive anti-nuclear antibody (ANA), panti-neutrophil cytoplasmic antibodies (ANCA), c-ANCA confirmed the diagnosis of APS.
Conclusion: This case report showed the feasibility of mechanical clot retrieval and stent implantation in patients with APS.
Due to the elevated risk of in-stent thrombosis a prolonged therapy with glycoprotein (GP)IIb/IIIa receptor antagonists in the
initial postoperative period and further anticoagulation with coumarin derivate might be needed.

Keywords
Antiphospholipid syndrome, stroke, thrombectomy, stent

Background
The antiphospholipid syndrome (APS) is an acquired
autoimmune condition and presents as a prothrombotic
disorder in patients who have persistent antiphospholipid antibodies (aPLs). It is accompanied by recurrent
pregnancy complications and miscarriages, thrombocytopenia and thrombosis.1–3 Thrombosis in patients
with APS can occur in arterial, microvascular or
venous locations.4 Deep vein thrombosis and stroke
in patients with APS are major causes of morbidity
and mortality.3
Due to a high risk for further events, anticoagulation
in patients with an APS-associated stroke is of
high importance in pharmacological prophylaxis.2,4
Secondary prophylaxis with warfarin is recommended
in patients with APS and arterial thrombosis, an international normalized ratio (INR) level >3.0 is

sometimes recommended, the INR target 2.0–3.0 is
also supported.4–6
Mechanical thrombectomy in acute ischaemic stroke
with a large vessel occlusion e.g. the M1-segment of the
middle cerebral artery has improved over the last

1

ET Department of Neurology, University Hospital Salzburg, Paracelsus
Medical University, Salzburg, Austria
2
Department of Neurosurgery, University Hospital Salzburg, Paracelsus
Medical University, Salzburg, Austria
3
Department of Neurology/Researchinstitute of Neurointervention,
University Hospital Salzburg, Paracelsus Medical University, Salzburg,
Austria
Corresponding author:
Monika Killer-Oberpfalzer, Department of Neurology/Researchinstitute of
Neurointervention, University Hospital Salzburg, Paracelsus Medical
University, Salzburg, Austria.
Email: m.killer@salk.at

2
years.7,8 The currently common technique is the use of
a stent retriever in combination with aspiration. Most
of the newer studies report a reperfusion rate more than
80% with this technique.9,10
The need for a stent implantation after acute mechanical thrombectomy based on recurrent M1-occlusion
or restenosis is not common.8,11–14 In cardiology,
however, the use of stents in acute coronary artery
occlusion is frequently performed. Therefore patients
with APS and coronary acute syndrome have been
successfully treated with stents.15–18
The complications of cardiac stent thrombosis are
described in a few case reports.19,20 To our knowledge
this is the first report of mechanical thrombectomy with
stent implantation with further therapeutic challenges
in a patient with APS and acute stroke.

Case
A 48-year-old woman was referred to the stroke unit
due to a Broca’s aphasia and a mild paresis on the right
side National Institutes of Health Stroke Scale
(NIHSS) of 3.

Interventional Neuroradiology 0(00)
The patient’s history showed a chronic thrombocytopenia since 15 years with unknown origin and
arterial hypertension. The medication prior to presentation nebivolol 5 mg and lisinopril 10 mg was taken
orally once a day.
Acute diagnostic work-up revealed left middle cerebral artery (MCA) occlusion yet without a significant
disturbance of diffusion on magnetic resonance imaging
(MRI) including diffusion weighted imaging (DWI)
(Figure 1).
Due to a thrombocytopenia (67,00 g/l,) systemic
thrombolysis with alteplase (rtPA) was not indicated
and the patient was immediately referred to interventional therapy 2.5 h after symptom onset. After successful clot retrieval, recurrent re-occlusions due to a
remaining M1 stenosis lead to the necessity of implanting a stent (Figure 2).
In addition, a glycoprotein (GP) IIb/IIIa receptor
antagonist
(aggrastat,
tirofiban;
Correvio,
Geneva, Switzerland) was administered intra-arterially
as bolus (19.69 mg/kg), followed by a continuous intravenous administration (400 mg/h) for the duration of
24 h.

Figure 1. Magnetic resonance imaging (MRI) brain: (a) apparent diffusion coefficient (ADC) maps; (b) diffusion weighted imaging (DWI);
(c) fluid-attenuated inversion recovery (FLAIR): without ischemic brain lesion. Note that (a) and (b) show a discrete diffusion disorder
without an infarct demarcation but with hyperaemia in (c). (d) Perfusion weighted image (PWI): mismatch represent potential salvageable
tissue by reperfusion therapy; (e) MR angiography (MRA) intracranial shows M1-segment occlusion. Arrow indicates area of vessel
occlusion.

Stadler et al.

3

Figure 2. Digital subtraction angiography (DSA): (a) occlusion of left M1-segment; (b) recanalization with restenosis of left M1-segment
after mechanical thrombectomy; (c) left M1-segment re-occlusion; (d) recurrent stenosis of left M1-segment after concurrent mechanical
thrombectomy; (e) reperfusion after stent implantation without stenosis. Arrow indicates area of vessel occlusion.

Figure 3. Magnetic resonance imaging (MRI) brain on day 5: (a) diffusion weighted imaging (DWI) and (b) FLAIR and (c) T2. Panels
(a) and (b) show the diffusion disorder with an infarct demarcation in (c). Arrow indicates area of ischemia.

One day after the interventional procedure and overlapping with the end of the 24-hour intravenous administration of the GP IIb/IIIa receptor antagonist, dual
antiplatelet treatment with acetylsalicyl acid 100 mg/d
and clopidogrel 75 mg/d was started. The neurological
deficit of the patient was unchanged with a mild hemiparesis on the right side and a mild Broca’s aphasia
(NIHSS of 3).
Transcranial doppler (TCD) sonography showed a
recanalised M1 and M2 segment of the left MCA with a
moderate stenosis in the distal part of the stent (Vmax
300 cm/s).
On day 5, the patient suddenly presented a severe
right hemiparesis and a global aphasia. The MRI
revealed a new ischaemic stroke in the territory supplied by the left MCA (Figure 3), the MR-angiographic
evaluation showed a subtotal stenosis in the left MCA.
The TCD sonography showed an in-stent restenosis
with a loss of vessel capture in the M1 segment and
low poststenotic flow in the M1 segment. Due to the
high risk of reperfusion syndrome and haemorrhagic
transformation of the infarct area, an angioplasty of
the stent was not performed.

The dual antiplatelet treatment with acetylic salicylic
acid 100 mg/d and clopidogrel 75 mg/d has been
continued.
Due to the stroke, the young age (<50 years) and the
thrombocytopenia, further diagnostics (including
abdominal ultrasonography, chest X-ray, dopplerduplex sonography of the extra- and intracranial
arteries, transthoracic echocardiography, 24 h-electrocardiogram (ECG), electroencephalography) and
laboratory investigations (including clinical biochemistry, haematology, coagulation, thrombophilia, urine
analysis, hormones, immunology and antibodies) were
performed. Laboratory evaluations showed a typical
constellation with increased lupus anticoagulant
(LA)1 (77.00 s), increased LA-ratio (2.3), positive
anti-nuclear antibody (ANA), p-anti-neutrophil cytoplasmic antibodies (ANCA), c-ANCA, SS-A/FC, SSB/FC, Histon/FC and dsDNA/FC. The diagnosis of
APS could be confirmed.
Three months after discharge the patient recovered
to a NIHSS of 1. As a Broca’s aphasia remained as
neurological deficit, the patient was graded MRS (modified Rankin Scale) of 2.

4
Regarding stent implantation, a secondary prevention with a dual antiplatelet therapy could be deescalated to acetylsalicyl acid 100 mg/d. Due to the APS,
there was additional indication for oral anticoagulation
and we started a therapy with a long lasting coumarin
derivate (phenprocoumon) with a target INR 2.0–3.0
was started three month after the event.

Discussion
We conclude that a mechanical recanalisation with
stent-implantation can successfully be performed in
patients with APS-associated M1-occlusion. We propose that stent implantation may be required to maintain patency and that aggressive medical treatment is
required to prevent stent re-occlusion.
Coster et al. reported two cases with a full recovery
after a stroke based on an APS treated by intra-arterial
thrombolysis. There is lack of information about platelet count in these two patients where no stent implantation was performed.21 Several case studies report
patients with acute myocardial infarcts and APS as
underlying disease, who were treated with percutaneous
transluminal coronary angioplasty (PTCA). In these
patients, the immediate start of oral anticoagulation
was initiated due to the high risk of recurrent coronary
stent thrombosis.15,22 As an alternative to PTCA the
administration of intravenous GP IIb/IIIa receptor
antagonists was needed.19,23
Su et al. recommend the additional administration of
GP IIb/IIIa receptor antagonist and prolonged heparin
treatment in APS patients undergoing a percutaneous
coronary intervention (PCI). Warfarin (INR target
2.0–3.0) is recommended to be started as a secondary
prevention. Therefore, an over-lapping (bridging) use
of heparin and warfarin should be considered.19
Okuma et al. randomized 20 patients with ischaemic
stroke and APS either to acetylsalicyl acid alone
(n ¼ 11) or acetylsalicyl acid plus warfarin (target
INR 2.0–3.0, n ¼ 9) for secondary prophylaxis. The
cumulative incidence of stroke in patients with antiplatelet treatment alone was significantly higher than in
patients receiving the combination of antiplatelet and
anticoagulation therapy.24
Our case confirms the high risk for in-stent restenosis
without anticoagulation after acute treatment with GP
IIb/IIIa receptor antagonists despite following with
antiplatelet therapy. This will lead us to combine in
future an antiplatelet agent (acetylsalicyl acid or clopidogrel) with an anticoagulant (coumarin derivate) for
patients with APS after stent implantation, considering
the high risk for bleeding complications due to the
thrombocytopenia.

Conclusion
This case report showed the feasibility of mechanical
clot retrieval and stent implantation in patients with
APS. Due to the elevated risk of in-stent thrombosis,

Interventional Neuroradiology 0(00)
a prolonged therapy with GPIIb/IIIa receptor antagonists in the initial postoperative period and further
anticoagulation with coumarin derivate might be
needed.
Funding
The author(s) received no financial support for the research,
authorship, and/or publication of this article.

Declaration of conflicting interests
The author(s) declared no potential conflicts of interest with
respect to the research, authorship, and/or publication of this
article.

References
1. Lim W. Antiphospholipid antibody syndrome.
Hematology Am Soc Hematol Educ Program 2009;
2009: 233–239.
2. Lim W. Antiphospholipid syndrome. Hematology Am
Soc Hematol Educ Program 2013; 2013: 675–680.
3. Cervera R, Piette J-C, Font J, et al. Antiphospholipid
syndrome: Clinical and immunologic manifestations and
patterns of disease expression in a cohort of 1,000
patients. Arthritis Rheum 2002; 46: 1019–1027.
4. Ruiz-Irastorza G, Hunt BJ and Khamashta MA. A systematic review of secondary thromboprophylaxis in
patients with antiphospholipid antibodies. Arthritis
Rheum 2007; 57: 1487–1495.
5. Finazzi G, Marchioli R, Brancaccio V, et al. A randomized clinical trial of high-intensity warfarin vs. conventional antithrombotic therapy for the prevention of
recurrent thrombosis in patients with the antiphospholipid syndrome (WAPS). J Thromb Haemost 2005; 3:
848–853.
6. Crowther MA, Ginsberg JS, Julian J, et al. A comparison
of two intensities of warfarin for the prevention of recurrent thrombosis in patients with the antiphospholipid
antibody syndrome. N Engl J Med 2003; 349: 1133–1138.
7. Mokin M, Khalessi AA, Mocco J, et al. Endovascular
treatment of acute ischemic stroke: The end or just the
beginning? Neurosurg Focus 2014; 36: E5.
8. Berkhemer OA, Fransen PSS, Beumer D, et al. A randomized trial of intraarterial treatment for acute ischemic
stroke. N Engl J Med 2015; 372: 11–20.
9. Broussalis E, Trinka E, Wallner A, et al. Thrombectomy
in patients with large cerebral artery occlusion: A singlecenter experience with a new stent retriever. Vasc
Endovascular Surg 2014; 48: 144–152.
10. Hausegger KA, Hauser M and Kau T. Mechanical
thrombectomy with stent retrievers in acute ischemic
stroke. Cardiovasc Intervent Radiol 2014; 37: 863–874.
11. Broderick JP, Palesch YY, Demchuk AM, et al.
Endovascular therapy after intravenous t-PA versus
t-PA alone for stroke. N Engl J Med 2013; 368: 893–903.
12. Ciccone A, Valvassori L, Nichelatti M, et al.
Endovascular treatment for acute ischemic stroke.
N Engl J Med 2013; 368: 904–913.
13. Kidwell CS, Jahan R, Gornbein J, et al. A trial of imaging selection and endovascular treatment for ischemic
stroke. N Engl J Med 2013; 368: 914–923.

Stadler et al.
14. Goyal M, Demchuk AM, Menon BK, et al. Randomized
assessment of rapid endovascular treatment of ischemic
stroke. N Engl J Med 2015; 372: 1019–1030.
15. Abid L, Frikha F, Bahloul Z, et al. Acute myocardial
infarction in young adults with antiphospholipid syndrome: Report of two cases and literature review.
Pan Afr Med J 2011; 8: 13.
16. Musuraca G, Imperadore F, Terraneo C, et al. Successful
treatment of post-exertion acute myocardial infarction by
primary angioplasty and stenting in a patient with antiphospholipid antibody syndrome. Blood Coagul
Fibrinolysis 2004; 15: 95–98.
17. Badak O, Güneri S, Kirimli O, et al. Primary stenting in a
patient with acute myocardial infarction and primary
antiphospholipid syndrome. J Invasive Cardiol 2002; 14:
194–197.
18. Timurkaynak T, Cemri M, Ozdemir M, et al. Primary
angioplasty and stenting in a patient with primary antiphospholipid
syndrome
and
thrombocytopenia.
J Invasive Cardiol 2001; 13: 395–400.
19. Su H-M, Lee K-T, Chu C-S, et al. Acute thrombosis after
elective direct intracoronary stenting in primary antiphospholipid syndrome: A case report. Kaohsiung J Med Sci
2003; 19: 177–182.

5
20. Mito T, Miura S-I, Takada K, et al. A case of coronary
artery disease with antiphospholipid syndrome that
showed repeated stent thrombosis. J Cardiol Cases
2011; 4: e80–e86.
21. Coster S, van Dijk LC, Treurniet FEE, et al. Successful
intra-arterial thrombolysis beyond the accepted 6-hour
time window in two young patients. J Neurol Sci 2010;
288: 182–185.
22. Biceroglu S, Ildizli Demirbas M, Karaca M, et al. Acute
thrombotic occlusion of right coronary and left circumflex coronary arteries in a patient with antiphospholipid
syndrome: Successful stent implantation. Case Rep Med
2010; 2010: 198594.
23. Smukowska-Gorynia A, Mularek-Kubzdela T and
Araszkiewicz A. Recurrent acute myocardial infarction
as an initial manifestation of antiphospholipid syndrome:
Treatment and management. Blood Coagul Fibrinolysis
2015; 26: 91–94.
24. Okuma H, Kitagawa Y, Yasuda T, et al.
Comparison between single antiplatelet therapy and
combination of antiplatelet and anticoagulation
therapy for secondary prevention in ischemic stroke
patients with antiphospholipid syndrome. Int J Med Sci
2009; 7: 15–18.