Abstract

Background: Intradural extramedullary tuberculoma of the spinal cord (IETSC) is an exceedingly rare manifestation of tuberculosis (TB) affecting the central nervous system.

Case Description: A 33-year-old immunocompetent female with disseminated TB, including pulmonary involvement and leptomeningeal tuberculomas, developed progressive paraplegia and urinary incontinence over 2 months. Magnetic resonance imaging revealed diffuse intradural extramedullary soft tissue from C7 to L2 vertebral levels, indicative of abscess formation and severe spinal cord compression. The surgical intervention involved posterior decompression and abscess removal from D7 to L2 vertebral levels, resulting in partial resolution of granulomatous lesions.

Conclusion: This case emphasizes the importance of considering IETSC in the differential diagnosis of spinal cord compression in TB-endemic regions. Further research is warranted to elucidate optimal management strategies, including the role of surgical intervention, in improving long-term neurological outcomes for patients with this rare but debilitating form of spinal TB.

Keywords: Atypical tuberculosis, Disseminated tuberculosis, Intradural tuberculoma, Surgical decompression

INTRODUCTION

Spinal intradural abscesses are an extremely rare condition.[ 19 ] Intradural spinal tuberculosis (TB) consists of <5% of CNS tuberculoma.[ 11 ] Intradural abscesses have been described as a consequence of a hematogenous spread of an infection; however, they may also result from iatrogenic causes (e.g., epidural injections) or spondylotic phenomena.[ 9 ] Magnetic resonance imaging (MRI) is a vital component in diagnosis, which reveals key pathologic features within the dural sac and the vertebral column.[ 9 ] Studies show that intradural abscesses are successfully treated with drainage and antitubercular drugs.[ 1 ]

TB is considered disseminated when Mycobacterium tuberculosis is isolated from blood or bone marrow or specimens from two or more noncontiguous organs in a single patient.[ 5 ]

In this article, we report a patient with intradural extramedullary tuberculoma of the spinal cord (IETSC) who underwent surgical resection.

CASE DESCRIPTION

A 33-year-old immunocompetent female with disseminated TB, including pulmonary TB and leptomeningeal tuberculomas, who had been on anti-tubercular treatment (ATT) with rifampicin, isoniazid, pyrazinamide, and ethambutol for the past 5 months, presented to our institute with paraplegia and urinary incontinence. This neurological deficit had an insidious onset and progressively worsened over the past 2 months, leading to complete paraplegia, absence of sensation below the D10 level, with positive Upper Motor Neuron signs and American Spine Injury Association (ASIA) A classification, rendering the patient completely bedridden. The preoperative MRI scan revealed diffuse, enhancing T2 and Short tau inversion recovery (STIR) hyperintense soft tissue in the intradural extramedullary compartment of the spine, extending from C7 to L2 indenting the spinal cord granulation tissue and abscess formation from the C7 to L2 vertebral levels [ Figures 1 and 2 ]. A loculated, peripherally enhancing collection with thick internal septations was noted posteriorly from D8 to D10 and from L1 to L2 vertebral levels, causing severe spinal canal stenosis compressing the dorsal spinal cord anteriorly [ Figures 1 - 3 ]. The leptomeningeal enhancement surrounding the lower end of the spinal cord and cauda equina nerve roots was reduced, suggestive of arachnoiditis. An MRI of the brain showed multiple tiny ring and disc-enhancing granulomas in the left parietal and occipital lobes, with perilesional edema. A computed tomography (CT) scan of the thorax revealed a few consolidated patches and firm nodules in the bilateral upper lobes, with multiple mediastinal lymph nodes showing calcification, suggestive of pulmonary Koch’s. A CT scan of the abdomen and pelvis suggested multiple enlarged lymph nodes in the para-aortic region at its bifurcation.

Figure 1:

Contrast-enhanced magnetic resonance imaging sagittal cut highlights a loculated peripherally enhancing pocket of collection with thick internal septations noted posteriorly from D8 to D10 vertebral level and similar morphology of peripheral enhancing lesion seen at L1 - L2 level. Both the arrows point the loculated peripherally enhancing pocket of collection with thick internal septations .

Figure 2:

Magnetic resonance imaging T2 sagittal cut highlights a loculated peripherally enhancing pocket of collection noted posteriorly from D8 to D10 vertebral level and similar morphology of peripheral enhancing lesion seen at L1-L2 level. Both arrows pointing toward collection.

Figure 3:

Magnetic resonance imaging axial cut at D10 level shows hyperintensity lesion compressing the cord. Blue arrow points the hyperintensity lesion and black arrow points toward cord.

Surgical management

An intradural extramedullary abscess was treated with posterior decompression through a midline incision, and adequate exposure was achieved. A midline laminectomy was performed from the D7 to D10 levels. No epidural granulation tissue or features of spondylodiscitis were found, confirming the radiological findings. The dura was found to be thickened and infiltrated by the underlying mass. An incision was made in the dura, revealing an abscess beneath it. All liquefied and caseous material, along with the abscess, was removed [ Figure 4 ]. A separate incision was made for the lumbar lesion, centered over the L1-L2 disc space, and similar steps were performed. The durotomy was closed with Prolene sutures. Immediately postoperatively, the patient was started on bedside mobilization with Taylor’s brace and axillary support, along with continuous sterile intermittent catheterization. Her sensations below the D10 level began to improve, and her neurological status progressed to ASIA B. A postoperative MRI [ Figures 5 and 6 ], done 5 months after surgical management, showed that most of the granuloma had been removed. At her 6-month clinical follow-up, the patient had begun mobilization with a walker and regained bowel and bladder control. Sensations fully recovered below the D10 level, and she demonstrated positive upper motor neuron signs, with her neurological status improving to ASIA D.

Figure 4:

The intraoperative image highlights the pus coming out after opening the dura. The arrow points to the pus seen after opening the dura.

Figure 5:

Postoperative magnetic resonance imaging sagittal T2 image shows resolution of septate collection at D8-D10 and L1 - L2 level. Both arrows point to the laminectomy defect.

Figure 6:

Postoperative magnetic resonance imaging axial cut at D8 level shows resolution of abscess. The orange arrow points to the cord, and the blue arrow points to the laminectomy site.

DISCUSSION

TB presents in the central nervous system about 10% of the time.[ 8 ] There are four typical presentations of spinal TB: Pott’s spine, nonosseous spinal tuberculous (extradural, intradural extramedullary, and intramedullary forms), tubercular arachnoiditis, and tuberculous meningitis[ 4 ] Out of which nonosseous spinal tuberculoma is extremely rare. Dastur reviewed 74 cases of spinal TB and reported that 65% of tuberculomas are extradural, 8% intramedullary, 5% intradural extramedullary, and 20% are arachnoidal.[ 2 ] The most common site of involvement is the thoracic spine.[ 2 , 7 ]

Roca[ 13 ] reviewed and reported 22 cases of IETSC in 2004. In his article, it was found that most cases of IETSC occurred after tuberculous meningitis while patients were taking anti-tuberculous medication. In the literature, it has been mentioned that despite the effective response to anti-tuberculous treatment, an ongoing inflammatory process within the arachnoid appears to persist, eventually resulting in the development of a tuberculoma. This complication of tuberculous meningitis may represent a manifestation of a paradoxical reaction to anti-tuberculous medication.[ 16 ]

IETSC can occur unpredictably, typically a time frame of 3 weeks to 1 year.[ 10 , 12 , 18 ] The mechanism driving this paradoxical reaction remains uncertain. However, it is believed to stem from interactions between the host’s immune response and mycobacterial products. As chemotherapy controls active TB, the host’s immune suppression subsides, potentially leading to hypersensitivity to mycobacterial protein derivatives and triggering a paradoxical immune response. This immune reaction perpetuates ongoing inflammation in the arachnoid membrane, ultimately resulting in the development of tuberculoma.[ 10 , 12 , 18 ] A combination of surgical intervention and medical therapy has demonstrated impressive results in managing intradural extramedullary spinal tuberculomas. While some researchers contend that anti-tuberculous treatment alone may suffice when a paradoxical reaction leads to tuberculoma formation, a review of the literature indicates that medical therapy alone is often ineffective. The reasons for the inadequate response to anti-tubercular chemotherapy in these cases are not fully understood.[ 3 ] Consequently, we argue that surgical intervention is essential when intradural extramedullary spinal tuberculoma occurs as a paradoxical reaction to treatment. Intradural tuberculous granulomas and chronic tuberculous arachnoiditis are considered atypical forms of TB and are generally secondary to tuberculous meningitis, which was also observed in our case. The disease presents insidiously with paraparesis, hypoesthesia with a sensory level, and bladder dysfunction due to cord involvement or compression by the inflammatory process, which in our case occurred after 5 months of ATT. The possible causes of this abscess could include drug resistance, poor compliance, and nutritional deficiencies. It has been stated in literature[ 15 ] that the growth of tuberculomas during treatment is thought to be linked to changes in the patient’s immune response. Infected individuals often exhibit hypersensitivity to various mycobacterial proteins. Tuberculostatic medications disrupt mycobacterial structures, releasing these proteins and resulting in localized inflammation and swelling. This process can trigger a delayed hypersensitivity reaction. Intradural tuberculomas tend to expand slowly and may become encapsulated after a latent period, leading to the paradoxical worsening of existing lesions. Such changes can impair the effectiveness of tuberculostatic drugs in penetrating the affected areas. Adjunctive steroid administration can also be considered beneficial for reducing interstitial edema and perilesional granulomatous vasculitis. However, delaying surgery, as in our case where there was a 2-month delay after the onset of complete paraplegia, can lead to worse outcomes.[ 10 , 16 ]

It is important to note that, in many patients, the diagnosis of IETSC was only established after symptoms had been present for a substantial period. Such delay was probably associated with a poorer response to therapy.[ 10 , 17 ] Regarding factors that influence the prognosis, in the developing world, due to affordability and accessibility, they are commonly misdiagnosed. The mainstay of treatment is surgical decompression to relieve the cord and an appropriate chemotherapy regimen [ Table 1 ]. A delay in surgery might be associated with worse outcomes.[ 14 ] Ju et al. suggested that antitubercular medications should be prescribed for at least 3 weeks before surgery to prevent the TB bacilli from spreading during surgery.[ 6 ] The role of surgical decompression needs to be evaluated by a long-term prospective study, as there is a possibility of irreparable damage done from TB infection due to end artery damage or irreversible damage to the cord.

Table 1:

A literature review of similar studies.

CONCLUSION

Although IETSC is an extremely rare form of spinal TB, it must be considered among the differential diagnoses for patients with a known history of TB and spinal cord compression. In cases where patients present with significant deficits due to cord compression, early diagnosis and aggressive surgical decompression, along with prolonged antibiotic therapy, are important predictors of a successful neurological and clinical outcome. Further research is needed to evaluate the role of surgical decompression in such cases, as there is a possibility of end artery damage or irreversible damage to the spinal cord.

Ethical approval

Institutional Review Board approval is not required.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

Use of artificial intelligence (AI)-assisted technology for manuscript preparation

The authors confirm that there was no use of artificial intelligence (AI)-assisted technology for assisting in the writing or editing of the manuscript and no images were manipulated using AI.

Disclaimer

The views and opinions expressed in this article are those of the authors and do not necessarily reflect the official policy or position of the Journal or its management. The information contained in this article should not be considered to be medical advice; patients should consult their own physicians for advice as to their specific medical needs.

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