LETTERS TO THE EDITOR
|Year : 2022 | Volume
| Issue : 6 | Page : 1208-1210
Myelitis with myositis - An unusual neurological complication of COVID-19
Amanda L Pereira1, Prashant Makhija2, Behram S Pardiwalla2, Raahul J Vakharia3
1 Department of Medicine, Wockhardt Hospital, Mumbai Central, Mumbai, Maharashtra, India
2 Department of Neurology, Wockhardt Hospital, Mumbai Central, Mumbai, Maharashtra, India
3 Department of Radiology, Wockhardt Hospital, Mumbai Central, Mumbai, Maharashtra, India
|Date of Submission||28-Aug-2022|
|Date of Decision||03-Oct-2022|
|Date of Acceptance||05-Oct-2022|
|Date of Web Publication||17-Nov-2022|
Amanda L Pereira
Department of Medicine, Wockhardt Hospitals Ltd., Unit-Adams Wylie Memorial, 1877, Dr. Anand Rao Nair, Marg, Mumbai Central, Mumbai, Maharashtra
Source of Support: None, Conflict of Interest: None
|How to cite this article:|
Pereira AL, Makhija P, Pardiwalla BS, Vakharia RJ. Myelitis with myositis - An unusual neurological complication of COVID-19. Ann Indian Acad Neurol 2022;25:1208-10
|How to cite this URL:|
Pereira AL, Makhija P, Pardiwalla BS, Vakharia RJ. Myelitis with myositis - An unusual neurological complication of COVID-19. Ann Indian Acad Neurol [serial online] 2022 [cited 2023 Feb 6];25:1208-10. Available from: https://www.annalsofian.org/text.asp?2022/25/6/1208/361410
Globally, as of February 2022, the World Health Organization has recorded 400 million confirmed cases of COVID-19, including five million deaths. COVID-19 is associated with neurological manifestations including encephalopathy, seizures, strokes, peripheral neuropathy, and anosmia. There are still sparse data on the occurrence of transverse myelitis or myositis in COVID-19. Although there are few reports of acute myelitis and myositis occurring individually, the combination of myelitis and myositis in the same patient is unreported yet to the best of our knowledge.
A 40-year-old gentleman presented in January 2021 with a history of painful urinary retention and constipation followed by weakness involving both legs since two days. No comorbidities. For around one week prior to admission, he had severe bodyache, with no associated fever, cough, or breathlessness. The urinary retention and constipation was acute in onset. He could not pass urine or stool, although he had the urge. He went to a local nursing home where urinary catheterization was done and per-rectal enema was given to relieve the painful distension of his abdomen. Next day he developed severe weakness in both legs which was symmetrical. He could neither move his legs in bed nor get out of bed. There was no upper limb weakness. No preceding history of drug or toxin exposure or recent vaccination.
On examination, he was conscious and alert. Cranial nerves including fundii were normal. All four limbs were tender on palpation. Power, tone, and reflexes were normal in upper limbs. Both lower limbs showed hypotonia, with the power of 0/5 and absent deep tendon reflexes. Plantars were mute. There was reduced sensory perception to pinprick. The sensory level was at T3.
Provisional diagnosis of acute transverse myelitis with myositis secondary to an infective/inflammatory cause was made. On investigation, creatine phosphokinase (CPK) and urine myoglobin levels were significantly elevated to 89,000 IU/L and 14,585 IU/L, respectively, with raised serum creatinine. Magnetic resonance imaging of cervico-dorsal spine with whole spine screening showed a T2/FLAIR hyper intense signal extending from C3 to D12, suggestive of long segment myelitis. The visualized surrounding muscles showed diffuse hyperintensity on STIR images, indicating myositis [Figure 1]a and [Figure 1]b Cerebrospinal fluid showed significantly raised proteins and modestly elevated white blood cell count with lymphocytic predominance. Cerebrospinal fluid analysis for possible infective etiology was negative. Connective tissue and vasculitic profile screening (ANA, dsDNA, cANCA, pANCA, and antiphospholipid antibodies) was also negative.
|Figure 1: (a) Sagittal T2 weighted image of the cervico-dorsal cord showing T2 hyper intense lesion extending from C3 to D12. (b) Coronal STIR image showing diffuse hyperintense signal in the neck muscles|
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In view of the ongoing COVID-19 pandemic, real time polymerase chain reaction for COVID-19 and High resolution computed tomography of chest were done. Both were negative. However, anti–SARS-CoV-2 qualitative antibody titer was significantly raised, suggesting a recent COVID-19 infection [Table 1].
|Table 1: Relevant investigations including blood, urine, and CSF analysis|
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The patient was treated with intravenous methylprednisolone 1 gram for three days followed by oral steroids. Although CPK levels significantly reduced (from 89,000 to 4,178 IU/L over 10 days), there was minimal improvement clinically in lower limb weakness. He then underwent five cycles of plasmapheresis. Oral steroids were also continued in tapering doses for eight weeks. Following plasmapheresis, there was a reduction in muscle tightness and tenderness with a gradual but significant improvement in motor power. The CPK, aspartate aminotransferase, alanine aminotransferase, and creatinine levels normalized further. On discharge, the patient was ambulating with support.
Simultaneous inflammatory involvement of spinal cord and muscular system is very rare and documented only in few case reports. Topcu et al. described an adolescent female with transverse myelitis, myositis, and Guillain Barre syndrome following Mycoplasma pneumoniae infection. Ray et al. reported a patient of chronic ulcerative colitis who presented with cervical cord transverse myelitis and more than a year later, developed dermatomyositis and fibrosing alveolitis with positive Jo-1 antibodies. Transverse myelitis with myositis has also been reported in antiphospholipid antibody syndrome and tropical infections like Chikungunya.,
Concomitant myelitis with myositis in COVID-19 has not been reported to the best of our knowledge.
It is still unclear whether the various neurological manifestations seen in COVID-19 occur secondary to systemic inflammation or due to direct action of the virus on the central nervous system. Steardo et al. hypothesized that the SARS-CoV-2 virus is neurotropic and enters the central nervous systemvia Angiotensin Converting Enzyme 2 receptors expressed in both neurons and glial cells. Wu et al. proposed a blood circulatory pathway by which the virus directly infects the central nervous system, releasing inflammatory mediators and increasing blood brain barrier permeability. They also suggested that simultaneous immune-mediated and hypoxic injury may be responsible for the neuropathology.
There is paucity of data regarding myelitis and skeletal muscle injury in COVID-19. Sheraton et al. while reviewing neurological complications of COVID-19 found only one case of acute myelitis in a COVID-19–positive elderly male from Wuhan, China. Guan and co-workers showed preliminary evidence of skeletal muscle injury in COVID-19–positive patients. They found the prevalence of myalgias to be 14.9%, with the rate increasing with disease severity. They also reported higher CPK levels (> 200 U/L) among 12.5% of nonsevere and 19% of severe cases. Only 0.2% were caused by rhabdomyolysis. Mao et al. also reported skeletal muscle injury in 10.7% cases, of which a significantly higher number comprised patients with liver and kidney dysfunction. Khedr et al. in their surveillance study of acute neurological manifestations among 439 Egyptian patients of COVID-19 reported two cases of myelopathy and two of myositis.
Our patient did not have typical symptoms of COVID-19 prior to his neurological symptoms. Real time polymerase chain reaction for COVID-19 was negative, suggesting an absence of active infection. However, antibodies to COVID-19 were positive in high titer, suggesting a possible recent asymptomatic infection. The patient did not have any history of COVID-19 vaccination prior to hospitalization which could have contributed to his high antibody titers.
This case emphasizes that COVID-19 should be considered as a possible etiology while evaluating patients with atypical neurological presentations.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
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