The SARS-CoV-2 responsible for phlegmasia cerulea dolens: a case report

Introduction:

Coronavirus disease 2019 (COVID-19) is a worldwide pandemic systemic infection that is responsible for serious coagulopathies similar to disseminated intravascular coagulation.

Case Presentation:

The authors report the case of a COVID-19 patient who presented with phlegmasia cerulea dolens (PCD) of the left lower limb, so he benefited from aponeurotomies of the internal and anterolateral muscular compartments.

Clinical Discussion:

The severe acute respiratory syndrome coronavirus 2 involves an inflammatory process in thrombotic events in COVID-19 patients, including a cytokine storm. PCD evolves in three semiological phases: venous stasis, weakening of the pulses, and the constitution of major ischemia. In the literature, the authors find many reports that have been published regarding increased thrombus formation in COVID-19 patients; these include DVT formation, pulmonary embolism, and stroke. Nevertheless, publications concerning PCD in COVID-19 patients remain rare.

Conclusion:

Although the severe acute respiratory syndrome coronavirus 2 remains a thrombogenic pathology, systematic anticoagulation is the subject of hypothesis. Hence the importance of regular monitoring of markers of vascular thrombosis.

Highlights

• The coronavirus disease 2019 is a worldwide pandemic of systemic infection that is responsible for serious coagulopathies.

• Publications concerning phlegmasia cerulea dolens in coronavirus disease 2019 patients remain rare.

• Systematic anticoagulation is the subject of a hypothesis. Hence the importance of regular monitoring of markers of vascular thrombosis.

Introduction

Coronavirus disease 2019 (COVID-19) is a worldwide pandemic and systemic infection responsible for ~6.6 million deaths all over the world (https://www.worldometers.info/coronavirus/).

It affects the respiratory system specifically1,2, but it can affect many functional systems of the human body, including the vascular system, and be responsible for serious coagulopathies similar to disseminated intravascular coagulation2.

We report the case of phlegmasia cerulea dolens (PCD) in a patient diagnosed with COVID-19.

Our case report was written according to SCARE guidelines3.

Presentation of case

A 57-year-old male patient with no relevant past medical history, especially no history of diabetes, hypertension, familial thrombophilia, or heart dysfunction, and who did not smoke tobacco, was admitted to the emergency room for swelling with sudden onset of the left leg (Fig. 1) and discoloration up to the thigh.

Figure 1.

Axial section showing significant edema of the limb compared to the right one.

The diagnosis of PCD was retained in front of a clinical picture made of a positive Homans sign with limb coldness and cyanosis. His capillary refill was delayed, with abolition of popliteal, anterior and posterior tibial pulses, without motor and sensitive deficit, on vascular examination. Thus, the patient was put on therapeutic low molecular weight heparin.

Ambient air saturation was estimated at 98%; the other vital signs included a blood pressure of 140/85 mm Hg, heart rate estimated at 102 beats per minute, and a respiratory rate of 18 breaths per minute.

Blood analysis showed: white blood cells at 14 000, C-reactive protein at 85 mg/l, hemoglobin at 12 g/dl, serum creatinine at 12 g/dl, urea at 0.45 g/dl, and a creatine phosphokinase level of 990 U/l.

The patient underwent a computed tomography angiography of the aorta and the two lower limbs, indicating a delay in the opacification of the leg axes due to an important edema of the soft parts, which caused compartment syndrome (Fig. 2).

Figure 2.

Frontal section showing a delay in the opacification of the left leg axes, which are hail due to compartment syndrome.

Indeed, depending on the emergency condition, the patient received amputation relief of the limb by aponeurotomies of the medial and anterolateral muscle compartments. The postoperative evolution was marked by a relief of the limb, which gradually recolored.

During his hospitalization, the patient developed acute kidney injury, stage 2 of KDIGO, associated with hyperkalaemia without electrical signs on the ECG. The creatine phosphokinase were at 6 normal. This clinical presentation suggests rhabdomyolysis was treated by medical treatment of his hyperkalaemia by measures including insulin and serum glucose, correction of his hypovolemia with 0.9% NaCl, and using diuretics for a diuresis higher than 2 l/24 h. The evolution was marked by an improvement in renal function as well as the correction of hyperkalaemia without extrarenal purification methods.

Thus, the patient was discharged under anticoagulant treatment and dressing at home for controlled healing of his discharge incisions (Fig. 3).

Figure 3.

Directed healing of discharge aponeurotomies.

Discussion

The SARS-CoV-2 (severe acute respiratory syndrome coronavirus 2) involves an inflammatory process in thrombotic events in COVID-19 patients, in whom an increase in thromboembolic events has been noted compared to the general population4,5. This inflammatory process is due to damage to endothelial cells, which leads to a thrombotic cascade6.

In this infection, there is a cytokine storm and activation of the extrinsic pathway of coagulation, with synthesis of fibrinogen and thrombopoietin by the liver, mediated by interleukin-6 which plays a key role in this activation7.

Two to ten percent of deep venous thrombosis is complicated by PCD8. We also note, in the case of PCD, a high morbidity and mortality rate, with an amputation rate of 50% and a death rate of 40%9–11.

PCD evolves in three semiological phases: venous stasis, weakening of the pulses, and the constitution of major ischemia. In the literature, we find many reports that have been published regarding increased thrombus formation in COVID-19 patients; these include DVT formation, pulmonary embolism, and stroke.

Anwar et al. 12 report the case of arterial thrombus formation in a COVID-19 patient despite being on full anticoagulation. We have previously reported the case of a patient who presented twice with acute limb ischemia at two different stages, despite being on anticoagulation13.

This is the second case of PCD during the SARS-CoV-2 pandemic in our institute since the first one was published14. To our knowledge, Michael et al.15 have reported the first case of PCD in a COVID-19 patient.

Routine anticoagulation in COVID-19 patients remains a matter of debate, although Tang et al.16 have reported that anticoagulation with low molecular weight heparin is associated with a better prognosis in 99 patients among 499 COVID-19 patients.

Conclusion

Although SARS-CoV-2 remains a thrombogenic pathology, systematic anticoagulation is the subject of hypothesis. Hence the importance of regular monitoring of markers of vascular thrombosis.

Ethical approval

Applicable.

Consent

Written informed consent was obtained from the patients for the publication of this study and its accompanying images.

Sources of funding

There is no financial support.

Author’s contribution

Y.B., H.B., and S.B.: conception, literature review, analysis, data collection, writing –- review and editing. I.H., Y.B., A.R., A.B., and O.E.M.: conception, methodology, supervision.

Conflicts of interest disclosure

There is no conflicts of interest between the authors.

Research registration unique identifying number (UIN)

This is not an original research project involving human participants in an interventional or an observational study, but a case report. This registration is not required.

Guarantor

Dr Youssef Banana (corresponding author), Department of Vascular Surgery, Mohammed VI University Hospital of Oujda, Mohammed First University of Oujda, Morocco. Tel: +212 658 641 363, e-mail: yousef.bana18@gmail.com.

Provenance and peer review

Not commissioned, externally peer-reviewed.