A 53-year-old lady with no known comorbidity had undergone cisternography for persistent rhinorrhea. Rhinorrhea resolved, and the report was normal. However, a week later, she complained of low back pain, followed by progressive quadriparesis and sphincter dysfunction. A detailed imaging revealed evidence of leptomeningitis around the brain stem, intradural cervical and dorsal collection with nodular enhancement, and clumping of cauda equina roots. Cerebrospinal fluid (CSF) showed pleocytosis with increased protein and hypoglycorrhachia. CSF cell count was 6000 (all lymphocytes), CSF proteins were 1138 mg%, and CSF glucose was 30 mg% (corresponding blood glucose level of 110 mg%). We did not get any systemic clue about the illness except progressive weight loss. Our team concluded either it is directly a deadly sequel of the dye-related complication of cisternography or activation of a chronic infection in the spine. We treated her with antitubercular drugs and steroids along with physiotherapy. We kept in mind the chronicity of the illness and endemicity of tuberculosis. She responded well and was mobilizable with minimal support. Hence, we continued the same regimen, keeping in mind the responsiveness of the patient to the same. We report this case to realize the possibility of this type of complication even with advanced dye and modern techniques of cisternography. Our case also underscores the requirement of prior imaging (magnetic resonance imaging of the spine) before undertaking such a procedure (cisternography). Last but not the least, sometimes we also need to make decisions based on our clinical knowledge as neurological investigations may be inconclusive in many circumstances.
Keywords: Cauda equina syndrome, cisternography, central nervous system tuberculosis
|How to cite this URL:|
Chakraborty D, Bhaumik S, Pramanick G, Biswas D. Story of a cisternography: Did we awaken a sleeping “Giant?”. J Appl Sci Clin Pract [Epub ahead of print] [cited 2023 Mar 29]. Available from: http://www.jascp.org/preprintarticle.asp?id=358985
| Introduction|| |
Cisternography is accepted to be a safe investigation for suspected cerebrospinal fluid (CSF) rhinorrhea. However, rarely, it may cause complications. We report a case where chronic meningitis and cauda equina syndrome happened just after cisternography. It created the dilemma whether it is a sequel to the procedure or due to an infection.
| Case Report|| |
A 53-year-old lady with no comorbidity was at her baseline 7 months before presentation to us. She had an insidious onset and intermittent nasal discharge (watery) which used to aggravate on bending forward with no other associated symptom. Otorhinolaryngology consultation showed no positive result. The fluid tested negative for CSF cisternography was within the normal limits. Postcisternography, she complained of persistent low back pain and 7 days later, she had an episode of fever lasting for 5 days (continuous, moderate grade with no associated symptom). Seven days after it subsided, she developed acute-onset low back pain, followed by progressive weakness of the left followed by the right lower limb (distal followed by proximal aspects). Thereafter, she complained of similar weakness of both upper limbs. All these disabilities progressed, and she was bedridden within few days. Three weeks before her presentation, she experienced progressive symptoms of urgency, hesitancy, incomplete evacuation of urine, and final retention. She had constipation for few weeks before presentation.
During the presentation to our institute (7 months from onset of symptoms), she was with stable vitals. Her higher mental function and cranial nerve examination were within normal limits. She had a power (Medical Research Council grading) of 3-4-/5 in upper limbs with normal tone and normal reflexes. Lower limbs had decreased tone, power of 2/5 proximally, 4/5 (left lower limb was weaker) with decreased deep tendon reflexes. Neck and trunk muscles were weak. Left-sided plantar was extensor and right side nonresponsive. She was having decreased joint position sense below the ankle on both sides. Patchy sensory deficits in limbs and trunk without sensory level. Her finger-nose-finger and heel-sheen tests were abnormal. She had urinary retention without other features of autonomic dysfunction. There was no perianal anesthesia with normal sphincter tone. There was no spine or skull swelling, but tenderness at L3, L4, and L5 vertebral levels.
We started her with supportive care and underwent immediate imaging (magnetic resonance imaging [MRI] of the whole spine with contrast along with MRI of the brain). It showed intradural collection extending from C7-T12 vertebral level with multiple patchy areas of T2 hyperintense lesion in the spinal cord and nodular enhancing lesion at D9 and D10 vertebral levels. There was a CSF loculated at the anterior part of the C2 vertebral level [Figure 1]. There was evidence of marked enhancement of cauda equina roots with clumping. MRI of the brain showed mild leptomeningeal enhancement around the brain stem [Figure 2]. Hence, we suspected diffuse spinal arachnoiditis, meningitis, and myelitis. An immediate CSF study showed a cell count of 6000 (lymphocytes), proteins of 1138 mg%, and glucose of 30 mg% (corresponding blood glucose level of 110 mg%). We did a comprehensive central nervous system (CNS) infection panel, which ruled out common CNS infection. Gram stain, acid-fast bacillus stain, and fungal stain were negative. Her CSF culture was sterile and blood limits were unremarkable with normal serology. We suspected chronic infection and kept tubercular etiology as a high possibility because of the chronicity and endemicity of the illness. It also was supported by imaging and CSF picture.
|Figure 1: Intradural collection extending from C7-T12 vertebral level with multiple patchy areas of T2-hyperintense lesion in the spinal cord and nodular enhancing lesion at D9 and D10 vertebral level. There was a CSF loculated area at the anterior aspect at C2 vertebral level. There was also evidence of marked enhancement of cauda equina roots with clumping (marked with blue arrows). CSF: Cerebrospinal fluid|
Click here to view
|Figure 2: Brain MRI revealed mild leptomeningeal enhancement around the brain stem. MRI: Magnetic resonance imaging|
Click here to view
We had started her with antibiotics in meningitis dose (ceftriaxone and vancomycin) and later replaced it with antitubercular drugs with steroids after CSF and imaging reports. Trunk and limb power improved to the extent, and we mobilized her with two-person support within a few days postadmission.
She had no history of TB but had features of decreased appetite and significant weight loss for a few months before her presentation to our institute. She had episodes of gastrointestinal upset, including periods of increased frequency of bowel movements and constipation. Hence, we searched for an abdominal source of infection. Her contrast-enhanced computed tomography (CT) chest was unremarkable and her CT abdomen showed heterogeneous liver parenchyma with hepatomegaly and splenomegaly. The colonoscopy and endoscopy were within normal limits.
| Discussion|| |
Our patient had symptoms started just after the CT cisternography so we could not rule out the association. Despite the rarity, reports of reversible and irreversible neurodeficit with intrathecal drug administration are there. There was a report of a young lady (underwent regional obstetric anesthesia) having arachnoiditis with cauda equina dysfunction and hydrocephalus after intrathecal bupivacaine. Although the mechanism is not very, reports of conus medullaris syndrome after radionuclide cisternography is there., Although a known (rare) complication of a dye pantopaque, yet to be reported with advanced noniodinated dye (which was used in her case).
We never got evidence of TB either. The blood tests, radiological evaluation, and Mantoux test were noncontributory. We know the sensitivity of CSF GeneXpert is higher with at least 3 ml of CSF sample or higher (82%), but we could not afford to draw enough CSF in our case, due to arachnoiditis and spinal block.
We continued the same regimen along with rehabilitation. After 2 months of follow-up, she could walk with minimal support with better sphincter function. We were not sure about the exact etiology. However, chronicity of the illness, constitutional symptoms, hepatosplenomegaly of unknown etiology, and hypoglycorrhachia hinted toward tubercular etiology. Our case highlights the importance of imaging (MRI of the spine) before cisternography in doubtful cases. As because, this might have changed the approach to our case altogether.
As the tubercular bacillary loads in CSF stay low, the diagnostic tests are enough. CSF Ziehl–Neelsen has sensitivity of 10%–20%. Mycobacterial culture is more sensitive (60%–70%) but takes more than weeks. Clinical diagnostic algorithms are there for TB but they are diverse and untested in many populations. Hence, in life-saving cases, clinical judgment to be the last call. In our case, we concluded possibly a dormant infection in the spine got activated postprocedure or the total catastrophe was a very rare sequel of the procedure itself (cisternography).
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.
We acknowledge the faith patient and her family kept on us during difficult times.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Huang M, Dalm B, Simpson RK. Toxic myelitis and arachnoiditis after intrathecal delivery of bupivacaine via an implanted drug delivery system: Case report and review of the literature. Cureus 2018;10:e2240.
Park BS, Park J, Koh SH, Choi H, Yu HJ, Lee KE, et al.
Conus medullaris syndrome as a complication of radioisotope cisternography. Can J Neurol Sci 2012;39:347-51.
Choi JC. Conus medullaris syndrome following radionuclide cisternography. Case Rep Neurol Med 2014;2014:201745.
Silva JA, Taricco MA, Brito JC, Neves VD, Farias RL. Constrictive arachnoiditis after pantopaque myelography causing syringomyelia and paraparesis: Case report. Arq Neuropsiquiatr 2001;59:619-22.
Bahr NC, Marais S, Caws M, van Crevel R, Wilkinson RJ, Tyagi JS, et al.
GeneXpert MTB/Rif to diagnose tuberculous meningitis: Perhaps the first test but not the last. Clin Infect Dis 2016;62:1133-5.
Ho J, Marais BJ, Gilbert GL, Ralph AP. Diagnosing tuberculous meningitis – Have we made any progress? Trop Med Int Health 2013;18:783-93.
Patel VB, Theron G, Lenders L, Matinyena B, Connolly C, Singh R, et al.
Diagnostic accuracy of quantitative PCR (Xpert MTB/RIF) for tuberculous meningitis in a high burden setting: A prospective study. PLoS Med 2013;10:e1001536.
Nhu NT, Heemskerk D, Thu do DA, Chau TT, Mai NT, Nghia HD, et al.
Evaluation of GeneXpert MTB/RIF for diagnosis of tuberculous meningitis. J Clin Microbiol 2014;52:226-33.
Khadilkar SV, Kadam ND, Kulkarni RV, Meshram CM, Meshram AR, Patel BA, et al.
Guidelines versus ground lines: Tuberculosis of the central nervous system. Neurol India 2019;67:787-91.
] [Full text]
64/4A/9, Beliaghata Main Road, Kolkata - 700 010, West Bengal
Source of Support: None, Conflict of Interest: None
[Figure 1], [Figure 2]