Atormac
Neurology India
menu-bar5 Open access journal indexed with Index Medicus
  Users online: 368  
 Home | Login 
About Editorial board Articlesmenu-bullet NSI Publicationsmenu-bullet Search Instructions Online Submission Subscribe Videos Etcetera Contact
  Navigate Here 
 Search
 
  
 Resource Links
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
    Article in PDF (1,484 KB)
    Citation Manager
    Access Statistics
    Reader Comments
    Email Alert *
    Add to My List *
* Registration required (free)  

 
  In this Article
   References
   Article Figures
   Article Tables

 Article Access Statistics
    Viewed2389    
    Printed17    
    Emailed0    
    PDF Downloaded30    
    Comments [Add]    

Recommend this journal

 


 
Table of Contents    
LETTERS TO EDITOR
Year : 2018  |  Volume : 66  |  Issue : 2  |  Page : 561-564

Chronic eosinophilic meningitis as a manifestation of isolated spinal neurocysticercosis: A rare case and a review of literature


1 Department of Neurosurgery, NIMHANS, Bengaluru, Karnataka, India
2 Department of Neurology, NIMHANS, Bengaluru, Karnataka, India
3 Department of Neuro-pathology, NIMHANS, Bengaluru, Karnataka, India

Date of Web Publication15-Mar-2018

Correspondence Address:
Dr. Amey R Savardekar
Department of Neurosurgery, NIMHANS, Bengaluru, Karnataka
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.227297

Rights and Permissions



How to cite this article:
Shashidhar A, Savardekar AR, Mundlamuri RC, Netravathi M, Nalini A, Chickabasaviah YT, Arivazhagan A, Rao MB. Chronic eosinophilic meningitis as a manifestation of isolated spinal neurocysticercosis: A rare case and a review of literature. Neurol India 2018;66:561-4

How to cite this URL:
Shashidhar A, Savardekar AR, Mundlamuri RC, Netravathi M, Nalini A, Chickabasaviah YT, Arivazhagan A, Rao MB. Chronic eosinophilic meningitis as a manifestation of isolated spinal neurocysticercosis: A rare case and a review of literature. Neurol India [serial online] 2018 [cited 2019 Oct 19];66:561-4. Available from: http://www.neurologyindia.com/text.asp?2018/66/2/561/227297




Sir,

Neurocysticercosis (NCC), an infection of the human brain caused by the larvae of the parasite Taenia solium, is the most common parasitic infection of the nervous system.[1],[2] The disease is primarily intracranial;[2],[3] and spinal involvement has been described only in 0.3–5.8% of NCC patients.[4],[5],[6] Isolated spinal NCC (without lesions in the brain) is very rare.[5],[6],[7],[8],[9] Spinal NCC predominantly presents with the symptoms and signs of spinal cord compression; however, occasionally, obstruction of the cerebrospinal fluid (CSF) pathways caused by scarring of the subarachnoid spaces may also cause symptoms.[3],[10] Spinal NCC manifesting primarily as recurrent chronic meningitis is highly unusual and raises important issues regarding the pathogenesis of the meningitis.[1],[11]

A 55-year old woman presented with multiple episodes of diffuse, dull-aching, holocranial headaches associated with photophobia and low-grade fever over a 2-year duration. She also gave a history of two episodes of right focal motor seizures over the past 1 year.

With the differential diagnosis of benign intracranial hypertension and chronic tuberculous meningitis (in view of the endemicity of tuberculosis), she underwent a computed tomography (CT) of the brain (plain and contrast), which was normal. She also underwent a diagnostic lumbar puncture (LP), which revealed normal CSF pressure. However, the CSF analysis showed a lymphocyte predominant picture with a low glucose and high protein content, consistent with chronic meningitis. Moderate number of eosinophils suggested the possibility of parasitic infection. Blood serum testing for anti-cysticercal antibody, immunoglobulin (Ig) G, by enzyme-linked immunodeficiency assay (ELISA) was positive. Testing for tuberculosis was negative. Contrast-enhanced magnetic resonance imaging (MRI) of the brain showed an increased obliquity of bilateral optic nerves pointing towards the presence of idiopathic intracranial hypertension, with absence of any intracranial lesion (namely NCC). The patient was treated for chronic meningitis with steroids and albendazole therapy. She showed symptomatic improvement; however, episodic headaches and low-grade fever persisted.

At a 2-year follow-up, she presented with severe low backache radiating to the anterolateral aspect of bilateral thighs. On examination, she was neurologically intact but bilateral straight leg raising (SLR) test was positive. MRI of the lumbosacral spine revealed an intradural, extramedullary cystic lesion at the L2–L3 level, compressing the cauda equina [Figure 1]. The cyst wall was brilliantly enhancing on contrast administration. In the context of the previous diagnosis of chronic eosinophilic meningitis, the radiological differentials were spinal NCC or spinal hydatid cyst. She underwent a lumbar (L) 2–L3 laminectomy and excision of the cyst. The cyst was intradural and extramedullary in location with a thickened arachnoid surrounding the true cyst wall [Figure 2]a,[Figure 2]b,[Figure 2]c. The thickened arachnoid was peeled, as shown in [Figure 2]a, and the cyst was delivered. Histopathology revealed a degenerating cysticercal cyst with no active scolex [Figure 2]d. Postoperatively, the patient recovered well and has been symptom-free during a 1-year follow-up period. MRI lumbosacral spine at the 3-month follow-up visit showed no evidence of any residual or recurrent lesion [Figure 3].
Figure 1: MRI of the lumbosacral spine with T1-weighted sequences in sagittal (a) and axial (d) sections; T2-weighted sequences in sagittal (b) and axial (e) sections; and, contrast-enhanced T1-weighted sequences in sagittal (c) and axial (f) sections showing the cystic lesion at the L2-L3 level with the cyst wall being T1-isointense, T2-hypointense and demonstrating an intense contrast-enhancement. The cyst contents are iso-intense with the CSF in all sequences. The contrast enhancement illustrates an intense arachnoidal reaction surrounding the cysticercal cyst

Click here to view
Figure 2: Solid line with closed arrow (a and b, intraoperative images) shows the cysticercal cyst wall (pearly white); while the surrounding inflamed thick arachnoid is shown by the dotted line with open arrow in (a). (c) Figure shows total excision of cyst. (d) Histopathological image (H and E, ×50) of the cysticercal cyst demonstrates the bladder cyst wall thrown into folds with the characteristic external corrugated appearance. Inset (H and E, ×400) shows the external homogenous eosinophilic tegument, the base of which is composed of the tegument cells. Inner pale mesenchyme contains excretory ducts

Click here to view
Figure 3: Postoperative (at 3-month follow-up) MRI of the lumbosacral spine with (a) T1-weighted, (b) T2-weighted, (c) contrast-enhanced T1-weighted sequences showing complete excision of the lesion

Click here to view


Our patient presented with recurrent bouts of aseptic chronic meningitis and refractory headaches. CSF showed lymphocytic predominance and moderate eosinophilia, which prompted us to investigate further for the presence of tuberculous or parasitic pathology. Testing for serum anti-cysticercal antibody IgG by ELISA was positive. However, MRI brain (plain and contrast) did not reveal any lesions. A screening MRI of the spine, at the time, may have revealed the likely pathology. We speculate that the patient's symptoms of recurrent bouts of headache resulted from inflammatory response to the cyst, with possibility of rupture of the NCC into the subarachnoid space. This was the probable cause of chronic meningitis, and may have led to scarring in the subarachnoid space, accounting for the imaging features of idiopathic intracranial hypertension.

Eventually, when the cyst size exceeded the narrow confines of the spinal canal, the patient presented with symptoms of spinal compression. The patient underwent surgery as the cauda equina was under severe compression. Medical management in the form of cysticidal drug therapy leads to complete resolution of cystic lesions in only 40% of previously untreated patients; hence, medical management alone was not an option in this case.[8] Radiologically, the evidence that the spinal cysticercal cyst had ruptured in the past was demonstrated by the intense contrast enhancement seen in the cyst wall and surrounding arachnoid. Intraoperatively, the thickened arachnoid surrounding the cyst wall is illustrated in [Figure 2]a, marked with the dotted line and open arrowhead. This suggested that the cyst was inflamed. As the patient was treated empirically with anti-helminthic drugs due to the positive serum IgG, the histopathology revealed a degenerating cysticercal cyst with no active scolex.

The issues raised in this case report are multifold. Primarily, was NCC indeed the cause of the chronic meningitis? The argument supporting our stance stems from the positive laboratory findings suggestive of cysticercal etiology (e.g., CSF eosinophilia, positive anti-cysticercal IgG antibody, as well as the characteristic histopathology confirming the presence of a cysticercal cyst) along with the exclusion of other important differential diagnosis including tuberculosis, which is endemic in India. The second question that needs consideration is the cause of the patient's initial symptomatology; could the symptoms be attributable primarily to the spinal NCC or to small nondetectable intracranial cysts. Rare cases of spinal cysticercal cysts presenting with meningeal onset of symptoms have been reported.[1],[8],[11] These cases are reviewed in [Table 1]. The spinal neurocysticercal cyst in our patient showed significant perilesional inflammation (as suggested by the intense contrast enhancement of the cyst wall on MRI and intraoperative finding of a thickened arachnoid surrounding the cyst). We hypothesize that the cyst may have ruptured into the surrounding subarachnoid space causing arachnoidal scarring and resulting in the episodes of chronic meningitis (headache and fever). The symptom of right focal motor seizures, however, cannot be attributed to the spinal NCC. Possibility of a ruptured viable brain cyst that was not detected on the initial neuroimaging scans, and which triggered the meningeal reaction, could also be considered.[11] Spinal NCCs in the subarachnoid space have been postulated to result from CSF spread of intracranial NCC.[11],[12] In this context, a ruptured brain cyst may also be considered as the inciting event leading to the genesis of the spinal NCC in our patient.
Table 1: Review of previously published cases of spinal neurocysticercosis with the rare presentation of chronic meningitis

Click here to view


Our case, as also the case reported by Boulos et al., demonstrates that the presentation of NCC can be deceptive, with cervicomedullary junction or spinal lesions presenting with severe headaches and recurrent aseptic meningitis.[1] We agree with the point proposed by Hackius et al., that a normal cranial MRI does not rule NCC in all patients.[8] This case makes a strong point for a spinal MR screening in patients having chronic meningitis with clinical features and CSF picture indicating a possibility of NCC, in the absence of intracranial lesions on MRI brain [Figure 4]. Isolated spinal cysticercosis can be considered as a remote differential diagnosis of chronic eosinophilic meningitis (especially in endemic regions).
Figure 4: Figure showing the proposed neuroimaging algorithm for cases of chronic eosinophilic meningitis depending on the clinical findings

Click here to view


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

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Boulos MI, Aviv RI, Lee L. Spinal neurocysticercosis manifesting as recurrent aseptic meningitis. Can J Neurol Sci 2010;37:878-80.  Back to cited text no. 1
    
2.
Garg RK, Malhotra HS, Kumar N. Disseminated cysticercosis: Many issues are still unresolved. Neurol India 2016;64:1060-2.  Back to cited text no. 2
[PUBMED]  [Full text]  
3.
Alsina GA, Johnson JP, McBride DQ, Rhoten PR, Mehringer CM, Stokes JK. Spinal neurocysticercosis. Neurosurg Focus 2002;12:e8.  Back to cited text no. 3
    
4.
Valsangkar SA, Kharosekar HU, Palande DA, Velho V. Isolated conus-epiconus neurocysticercosis. Neurol India 2015;63:119-20.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
Mewada T, Srivastava AK. Isolated cervical intramedullary cysticercosis. Neurol India 2016;64:188-9.  Back to cited text no. 5
[PUBMED]  [Full text]  
6.
Joob B, Wiwanitkit V. Isolated cervical intramedullary cysticercosis. Neurol India 2016;64:586.  Back to cited text no. 6
[PUBMED]  [Full text]  
7.
Callacondo D, Garcia HH, Gonzales I, Escalante D, Nash TE. High frequency of spinal involvement in patients with basal subarachnoid neurocysticercosis. Neurology 2012;78:1394-400.  Back to cited text no. 7
    
8.
Hackius M, Pangalu A, Semmler A. Neurological picture. Isolated spinal neurocysticercosis. J Neurol Neurosurg Psychiatry 2015;86:234-5.  Back to cited text no. 8
    
9.
Pant I, Chaturvedi S, Singh G, Gupta S, Kumari R. Spinal cysticercosis: A report of two cases with review of literature. J Craniovertebr Junction Spine 2016;7:285-8.  Back to cited text no. 9
    
10.
Colli BO, Assirati Junior JA, Machado HR, dos Santos F, Takayanagui OM. Cysticercosis of the central nervous system. II. Spinal cysticercosis. Arq Neuropsiquiatr 1994;52:187-99.  Back to cited text no. 10
    
11.
Muthukumar N. Neurocysticercosis: Evolution of our understanding. Neurol India 2017;65:885-7.  Back to cited text no. 11
[PUBMED]  [Full text]  
12.
Abarrategui Yague B, Garcia Garcia ME, Orviz Garcia A, Casas Limon J. Lymphocytic meningitis and spinal neurocysticercosis: A case report and literature review. Neurologia 2014;29:574-6.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]
 
 
    Tables

  [Table 1]



 

Top
Print this article  Email this article
   
Online since 20th March '04
Published by Wolters Kluwer - Medknow