Abstract
Bilateral femoral neuropathy is rare, especially that caused by bilateral compressive iliopsoas, psoas, or iliacus muscle hematomas. We present a case of bilateral femoral neuropathy due to spontaneous psoas hematomas developed during COVID-19 critical illness. A 41-year-old patient developed COVID-19 pneumonia, and his condition deteriorated rapidly. A decrease in the hemoglobin level prompted imaging studies during his intensive care unit (ICU) stay. Bilateral psoas hematomas were identified as the source of bleeding. Thereafter, the patient complained of weakness in both upper and lower limbs and numbness in the lower limb. He was considered to have critical illness neuropathy and was referred to rehabilitation. Electrodiagnostic testing suggested bilateral femoral neuropathy because of compression due to hematomas developed during the course of his ICU stay. The consequences of iliopsoas hematomas occurring in the critically ill can be catastrophic, ranging from hemorrhagic shock to severe weakness, highlighting the importance of recognizing this entity.
A wide range of neurological complications associated with COVID-19 affecting both central and peripheral nervous systems have been reported. Unilateral femoral mononeuropathy is an uncommon condition. Bilateral femoral neuropathy is rare, especially that caused by bilateral compressive iliopsoas, psoas, or iliacus muscle hematomas. In this report, we present the case of a 41-year-old patient with COVID-19-associated acute respiratory distress syndrome (ARDS), who developed bilateral femoral neuropathy as a consequence of bilateral psoas hematomas. We additionally report the findings of a literature review search for bilateral femoral neuropathy cases in association with psoas, iliacus, or iliopsoas hematomas. The management of such cases remains largely unstandardized and understudied.
Case Report
Patient information
A 41-year-old man, a known case of diabetes mellitus type 2, bronchial asthma, and obesity, presented with cough, shortness of breath, and fever. His COVID-19 positive status was confirmed via nasopharyngeal polymerase chain reaction testing. The patient developed ARDS during his hospital stay, and transferring him to the intensive care unit (ICU) was deemed necessary. His condition continued to deteriorate; hence, he was mechanically ventilated and placed on venovenous extracorporeal membrane oxygenation (ECMO) 3 days into his ICU stay. His ICU course was both complicated and lengthy. He developed partial thrombosis of the proximal part of the right internal jugular vein, which was the ECMO access point at that time. Therefore, the access was switched to the left side (left internal jugular and right common femoral veins). A decrease in the hemoglobin level from 9.0 to 7.5 g/L prompted imaging investigations. Computed tomography (CT) scan of the abdomen and pelvis demonstrated the source of the bleeding: right iliopsoas and left psoas hematomas (Figures 1-2). At the time of the bleeding, heparin infusion was being administered. The patient was treated conservatively, and his hemoglobin level subsequently stabilized.
On discharge from the general hospital, although higher brain function was preserved, upper and lower limb weakness and loss of sensation in the ventral side of both lower limbs were evident. Dabigatran 110 mg twice daily was added to his home medications.
Clinical Findings
The patient was referred as a case of critical illness neuropathy for rehabilitation. He was admitted to the inpatient rehab floor 3 months after the discharge. On examination, upper limb tone and power were back to baseline. However, his lower limbs were hypotonic. Hip flexion was 1/5 on the left side and 2/5 on the right side. Both knee flexors and extensors were weak, with powers of 2/5 and 0/5, respectively. His ankle power was 4/5 bilaterally. Knee and ankle reflexes were absent in both limbs. Furthermore, he had reduced sensation in the distribution of the femoral and saphenous nerves.
Diagnostic assessment
Given the above findings, the patient was suspected to have femoral neuropathy in both limbs in addition to the diagnosis of critical illness neuropathy. Therefore, an electrodiagnostic study was performed. The findings were conclusive for mild sensory motor axonal polyneuropathy and bilateral femoral neuropathy with severe denervation in both quadricepses. The iliopsoas on the left side exhibited significant weakness, but needle electromyography was not performed as the patient was on anticoagulants. As he was retrospectively diagnosed, a follow-up CT scan was carried out 8 months after the bleeding, which showed complete resolution of the hematomas.
Therapeutic intervention
A comprehensive inpatient rehabilitation program was offered, including occupational and physical therapy, for 5 months.
Follow-up and outcomes
One year after the injury to the femoral nerve, the patient was independent for all activities of daily living and was able to walk with a walking aid (walker, quadripod) under supervision owing to safety concerns. As his knees continued to buckle, a ground reaction ankle foot orthosis was prescribed to aid his gait. The motor power was 2+/5 in his right quadriceps and 2/5 in his left side; his hip flexors also improved, with a power of 3/5.
Discussion
This case report presents a patient with bilateral femoral neuropathy secondary to compression by bilateral psoas hematomas associated with COVID-19.
Owing to its long course, the femoral nerve is susceptible to compression, which is most common within the psoas muscle, iliopsoas groove, and inguinal ligament.1,2 Other causes for femoral neuropathy include surgical procedures involving the abdomen, pelvis, inguinal area, and hip, such as hip arthroplasties. Procedures that involve catheterization of the femoral artery or vein is another iatrogenic cause. Penetrating or blunt trauma and hip or pelvic fractures can also cause femoral neuropathy. Although diabetes mellitus can sometimes result in isolated femoral neuropathy, involvement of spinal roots, lumbosacral plexus, or other peripheral nerves is also evident on careful clinical or electrodiagnostic assessment in most cases.3,4 In our case, we propose the bilateral psoas hematomas to be the main culprit for the patient’s bilateral femoral neuropathy. Other contributing factors include right femoral vein cannulation for ECMO, diabetes mellitus, and prone positioning.
Iliopsoas hematomas are uncommon, and most cases are caused by anticoagulation/antiplatelet therapy or hemophilia. In the majority of reported cases, anticoagulation was within the therapeutic range, which applied for both unilateral and bilateral cases (Table 1). Some cases due to trauma in patients with no bleeding disorders have also been reported.1 With the increasing use of anticoagulants in clinical scenarios, iliopsoas hematomas are expected to be increasingly encountered.
This case is noteworthy as most cases of iliopsoas hematoma-associated femoral neuropathy are unilateral. There are 8 reported cases with bilateral femoral neuropathy-associated hematoma in the literature (Table 1). Additionally, we highlight the importance of identifying this condition promptly to avoid delays in diagnosis. Our patient was diagnosed in the rehabilitation setting (Figure 3). The ICU course was retrospectively reviewed to pinpoint the cause. Thankfully, the hematoma resolved spontaneously although the patient was maintained on dabigatran. Earlier diagnosis of this compressive neuropathy might have offered the benefit of discontinuing anticoagulants as soon as it was feasible. Whether stopping the anticoagulants could have hastened the resolution, reduced the duration of compression, and improved the prognosis is an essential consideration in future cases. Owing to the rarity of the condition and lack of evidence, the ideal plan is an individualized management approach and shared decision-making with the patient. The association of this condition with COVID-19 complicated the case. The hypercoagulable state imposed by the infection increased the need for anticoagulants. As evident from Table 1, our patient did not have a robust recovery. The masking of a critical illness and the consequent delay in diagnosis likely worsened the outcome. Another factor that might have affected the outcome is the association with COVID-19.
The presentation of the Iliopsoas hematoma starts with groin and hip pain, radiating to the anterior thigh and lumbar region, with hip flexion and external rotation owing to iliacus muscle spasm. Patients can also develop neuropathy, as in our case, and/or massive bleeding and shock. Femoral neuropathy manifests initially in the form of anteromedial thigh and leg pain, followed by anesthesia at the exact locations. Motor weakness and wasting of the quadriceps can also occur, with a decrease in or absence of knee jerk. Moreover, variable hip flexion weakness may develop.1 As evident from the CT scan, the left psoas hematoma was more extensive and more proximal, which explained the more severe deficits on this side in the present case.
Diagnosing iliopsoas hematomas is best done using magnetic resonance imaging owing to its high sensitivity and specificity. However, CT is routinely used as it is more readily accessible, faster, and has good sensitivity.1 Electrodiagnostic studies are valuable in confirming the diagnosis of femoral neuropathy, excluding other neural involvement, and determining the prognosis of femoral neuropathy.
The mainstay of femoral neuropathy treatment is physiotherapy unless the cause of compression can be removed. Early rehabilitation is likely to improve the outcomes and shorten the duration of recovery (Table 1). Initial actions for treating iliopsoas hematomas involve resuscitative measures as well as reversal of coagulopathy if present. The definitive treatment for iliopsoas hematoma in association with femoral neuropathy remains controversial, and the choice between conservative and operative management is not straightforward. Suggested indications for intervention include severe femoral neuropathy, refractory hemodynamic instability, abdominal compartment syndrome, and large hematomas. The disadvantage of surgery is that he removal of the hematoma may increase the bleeding by eliminating the tamponade effect. Moreover, in some cases, abdominal packing is the only surgical option. In addition, many of the patients are poor surgical candidates. Thus, interventional radiology with intra-arterial embolization or stent-grafting is emerging as a preferred treatment modality, especially in case of active bleeding. Poor surgical candidates may also benefit form ultrasound- or CT-guided percutaneous decompression.2
Conclusion
Much attention has been devoted to the thrombotic complications of COVID-19; however, hemorrhagic complications can also be life-threatening and are associated with lifelong disability. Hence, care should be taken to detect and possibly actively manage hematomas, mainly if they occur around the course of nerves. Knowledge of treatment options is critical, and the therapy should be individualized on a case-to-case basis.
Acknowledgment
We would like to thank content concepts (https://contentconcepts.in/) for English language editing.
Footnotes
Disclosure. Authors have no conflict of interests, and the work was not supported or funded by any drug company.
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