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         CASE SNIPPET Year : 2023  |  Volume : 69  |  Issue : 4  |  Page : 239-240

A rare presentation of acute transverse myelitis

KG Koshy, SS Nashi, GB Kulkarni, A VR Taalapalli
Department of Neurology, NIMHANS, Bengaluru, Karnataka, India

Date of Submission26-Jun-2022Date of Decision10-Aug-2022Date of Acceptance27-Aug-2022Date of Web Publication10-Mar-2023

Correspondence Address:
Dr. S S Nashi
Department of Neurology, NIMHANS, Bengaluru, Karnataka
India

Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/jpgm.jpgm_520_22

How to cite this article:
Koshy K G, Nashi S S, Kulkarni G B, Taalapalli A V. A rare presentation of acute transverse myelitis. J Postgrad Med 2023;69:239-40

A 23-year-old engineer with a history of human immunodeficiency virus (HIV) infection for 1 year, on treatment with tenofovir, lamivudine, and efavirenz, presented with tingling sensation and weakness of the left lower limb for 4 days, difficulty in passing urine for 3 days, and weakness and numbness in the right lower limb for 2 days. He had pain and redness in the right eye for 1 month. There was no cranial nerve involvement or symptoms pertaining to the upper limbs.

General examination was normal. There was no lymphadenopathy or organomegaly. The cardiac examination was unremarkable. Pupils were equal in size and reactive to light and accommodation. Motor examination revealed spastic paraparesis with sensory impairment to pain, fine touch, and vibration below the T6 segmental level. Deep tendon reflexes were exaggerated in the lower limbs and plantar responses were bilaterally extensor. There were no signs of meningeal irritation. Ophthalmological evaluation showed uveitis in the right eye. There was no evidence of retinitis or papilledema.

His CD4 count was 186 cells/mm3. Magnetic resonance imaging (MRI) of the spinal cord showed a longitudinally extensive T2 hyperintensity involving the cervicodorsal spine [Figure 1]a and [Figure 1]b from the C6 to D9 level with cord expansion. Post-contrast T1 images showed subtle foci of patchy enhancement within the dorsal cord [Figure 1]c. A few foci of blooming were seen in the dorsal cord on gradient echo (GRE) imaging [Figure 1]d. The cerebrospinal fluid (CSF) study showed normal opening pressure, 42 cells per high-power field with elevated protein (91.2 mg/dl) and low sugar (47 mg/dl). Hence, an infective or inflammatory cause of myelopathy was considered. CSF India ink staining for Cryptococcus, cryptococcal antigen, CBNAAT (cartridge-based nucleic acid amplification test), Gram stain, and cultures were negative. Serum NMO/MOG (neuromyelitis optica/myelin oligodendrocyte glycoprotein) were negative. Serum VDRL (venereal disease research laboratory), Serum TPHA (Treponema pallidum hemagluttination) and CSF VDRL were positive. CSF polymerase chain reaction (PCR) tests for HSV (herpes simplex virus) and CMV (cytomegalovirus) were negative. CSF cytology did not show any abnormal cells. Hence, a provisional diagnosis of acute longitudinally extensive transverse myelitis due to syphilitic etiology was made.

Figure 1: (a) MRI (T2 sagittal section) of the cervicodorsal spinal cord showing a longitudinally extensive T2 hyperintensity (blue arrow) with cord expansion; (b) MRI (T2 axial section) at the lower cervical spinal cord showing T2 hyperintensity involving the whole cord (blue arrow); (c) MRI (T1 post-contrast sagittal image) of the cervicodorsal cord showing subtle foci of patchy enhancement in the dorsal cord (blue arrow); (d) MRI (GRE axial image) showing blooming focus in the dorsal cord

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He was treated with intravenous ceftriaxone 1 gram twice daily for 14 days. At the time of discharge, his power in both lower limbs had improved from MRC (Medical Research Council) grade 1 to MRC grade 3. Following discharge, he was continued on antiretroviral therapy, cotrimoxazole prophylaxis, and physiotherapy. At follow-up, 3 months after discharge, he had only minimal weakness in his lower limbs and could walk without support. Repeat MRI at this stage showed a significant reduction in the long segment T2 hyperintensity [Figure 2]. There were no foci of blooming or enhancement in the repeat MRI. Repeat serum and CSF VDRL tests showed a 4-fold fall in titer.

Figure 2: Follow-up MRI (T2 sagittal image) at 3 months showing significant resolution in the dorsal cord T2 hyperintensity

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Longitudinally extensive transverse myelitis (LETM) is defined as spinal cord inflammation extending beyond three vertebral segments. It may be due to demyelinating, inflammatory, infective, or neoplastic causes. A retrospective study of 64 patients in northern India revealed that the NMO spectrum disorder was the most common single cause of LETM, accounting for about 32% of cases.[1] The other causes were multiple sclerosis, acute disseminated encephalomyelitis, subacute combined degeneration, tuberculous myelitis, and systemic lupus erythematosus. The common causes of myelopathy in HIV infection include vacuolar myelopathy, tuberculosis, CMV polyradiculomyelitis, toxoplasmosis, and lymphoma.[2] Syphilis is a sexually transmitted disease that has reduced in incidence in the antibiotic era but is re-emerging in connection with the AIDS pandemic. There are three patterns of spinal cord involvement in syphilis – meningomyelitis, meningovascular disease, and tabes dorsalis.[3] The subacute onset, CSF pleocytosis, increased CSF protein, and near complete recovery with treatment in the present case favors a diagnosis of meningomyelitis. Uveitis and optic perineuritis are the most common eye findings in tertiary syphilis, of which uveitis was seen in the present case.[4] The two radiological signs described in syphilitic myelitis are the flip flop sign (T2 hypointensity with gadolinium enhancement at sites of active inflammation) and candle guttering appearance (peripheral strip-like enhancement at the surface of the spinal cord).[5] As the imaging findings are not specific or sensitive, it is important to maintain a high index of suspicion for syphilitic myelitis, especially in immunocompromised patients as it is a readily treatable cause and in view of the danger posed by inadvertent steroid therapy in such cases.

Declaration of patient consent

The authors certify that appropriate patient consent was obtained.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 

 :: References 
1.Jain RS, Kumar S, Mathur T, Tejwani S. Longitudinally extensive transverse myelitis: A retrospective analysis of sixty-four patients at tertiary care center of north-west India. Clin Neurol Neurosurg 2016;148:5-12.  
    2.Chong J, Di Rocco A, Tagliati M, Danisi F, Simpson DM, Atlas SW. MR findings in AIDS-associated myelopathy. AJNR Am J Neuroradiol 1999;20:1412-6.  
    3.Chilver-Stainer L, Fischer U, Hauf M, Fux CA, Sturzenegger M. Syphilitic myelitis: Rare, nonspecific, but treatable. Neurology 2009;72:673-5.  
    4.Parker SE, Pula JH. Neurosyphilis presenting as asymptomatic optic perineuritis. Case Rep Ophthalmol Med 2012;2012:621872.  
    5.Dong H, Liu Z, Duan Y, Li D, Qiu Z, Liu Y, et al. Syphilitic meningomyelitis misdiagnosed as spinal cord tumor: Case and review. J Spinal Cord Med 2021;44:789-93.  
    
  [Figure 1], [Figure 2]