Atormac
Neurology India
menu-bar5 Open access journal indexed with Index Medicus
  Users online: 4572  
 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 (551 KB)
    Citation Manager
    Access Statistics
    Reader Comments
    Email Alert *
    Add to My List *
* Registration required (free)  

 
  In this Article
   References
   Article Tables

 Article Access Statistics
    Viewed74    
    Printed0    
    Emailed0    
    PDF Downloaded2    
    Comments [Add]    

Recommend this journal

 


 
Table of Contents    
LETTERS TO EDITOR
Year : 2019  |  Volume : 67  |  Issue : 2  |  Page : 563-565

Chronic meningitis with persistent hypoglycorrhachia: An unusual presentation of Lyme's disease


Child Neurology Division, Department of Pediatrics, All India Institute of Medical Sciences, New Delhi, India

Date of Web Publication13-May-2019

Correspondence Address:
Dr. Sheffali Gulati
Child Neurology Division, Department of Pediatrics, Room No. 3056, Third Floor, Teaching Block, All India Institute of Medical Sciences, New Delhi
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.258039

Rights and Permissions



How to cite this article:
Farmania R, Jauhari P, Chakrabarty B, Gulati S. Chronic meningitis with persistent hypoglycorrhachia: An unusual presentation of Lyme's disease. Neurol India 2019;67:563-5

How to cite this URL:
Farmania R, Jauhari P, Chakrabarty B, Gulati S. Chronic meningitis with persistent hypoglycorrhachia: An unusual presentation of Lyme's disease. Neurol India [serial online] 2019 [cited 2019 May 24];67:563-5. Available from: http://www.neurologyindia.com/text.asp?2019/67/2/563/258039




Sir,

Chronic meningitis with hypoglycorrhachia is generally suggestive of tuberculosis, fungal infections, mumps, protozoal infection, and malignancy; Lyme's meningitis is a rare differential diagnosis. The cerebrospinal fluid (CSF) in Lyme's meningitis typically presents with mononuclear cell predominance, a high level of proteins, and nearly normal sugar; hypoglycorrhachia can rarely be seen.[1] We report a child with chronic meningitis with persistent low CSF sugars despite clinical improvement, who was diagnosed with Lyme's meningitis and treated successfully.

A 7-year old, developmentally normal girl presented with fever, headache, and occasional nonprojectile vomiting for 2 months. There was no history of seizures, loss of consciousness, abnormal movements, or weakness of limbs. The child was diagnosed as having pyogenic meningitis at an outside hospital based on the CSF findings and received multiple antibiotics for 2 months. She was also started on antituberculous therapy for the last 1 week. At presentation in our hospital, the child was conscious and oriented, with brisk deep tendon reflexes, and signs of meningeal irritation. No other neurologic signs were present. A repeat CSF analysis was performed at our center and workup for various etiologies that may be responsible for chronic meningitis was done [Table 1]. The diagnosis of Lyme's meningitis was made on the basis of positive CSF immunoglobulin M (IgM) for Borrelia by enzyme linked immunosorbent assay (ELISA) in high titers (10.9, positive >3.5). The serum IgM assay for the same organism was negative. The workup for other infective etiologies was negative. The child was started on intravenous ceftriaxone (100 mg/kg/day). All previous medications were stopped. The child showed a clinical recovery over the next 7 days; however, on day 14, the CSF showed persistence of cells and hypoglycorrhachia [Table 2]. A 4-week therapy with intravenous ceftriaxone and a three-week therapy with oral doxycycline was planned. The child stayed asymptomatic throughout. A repeat CSF evaluation after 8 weeks still showed persistent low sugar levels. The child remained asymptomatic after a 1-year follow-up period.
Table 1: Etiological workup for chronic meningitis

Click here to view
Table 2: Serial CSF study in the index case

Click here to view


Hypoglycorrhachia in a case of chronic meningitis is commonly seen in numerous infectious (tuberculosis, fungal, mumps, and protozoal). The noninfectious etiology, including auto immune diseases like systemic lupus erythematosis, neurosarcoidosis, and malignancy, may also result in this manifestation.[2] In tropical countries like India, tuberculosis is the most common etiology for such a type of presentation; Lyme's meningitis is considered as a rare differential diagnosis. The diagnosis of Lyme's meningitis in non-endemic regions is often missed as it has a chronic course with nonspecific symptoms. The disease is indistinguishable from other causes of chronic meningitis. A long duration of symptoms, cranial nerve palsy, predominance of CSF mononuclear cells, and a high level of CSF proteins in the endemic areas predict a high probability of the presence of Lyme's disease.[3] The establishment of a definite diagnosis of neuroborreliosis requires the resence of neurologic features along with CSF pleocytosis and intrathecal Borrelia antibody production.[3] Our case had prolonged fever with meningeal signs with a low neutrophilic response at the first instance with high protein levels. A high titer of CSF IgM antibody against Borrelia by the ELISA technique compared to the serum IgM antibody in the index child was suggestive of the intrathecal synthesis of antibodies, which was consistent with the diagnosis of neuroborreliosis. The CSF in Lyme's meningitis typically has a high cell count with lymphocytic predominance, as well as a high protein, and a normal sugar level. A persistently low CSF sugar in the index case was an unusual finding. There are anecdotal reports of hypoglycorrhachia associated with Lyme's meningitis.[4],[5] Lakos described the CSF finding in Lyme's disease comparing CSF in cases suffering from Lyme and non-Lyme's meningitis and found a low CSF sugar level in 6 out of 59 (10%) cases in combination with pleocytosis and elevated protein in Lyme's meningitis.[6] Another unusual feature of this infection is the persistence of symptoms even after completion of therapy in certain cases.[7] A serological follow-up is considered unnecessary for establishing the diagnosis of Lyme's disease as antibodies to Borrelia (both IgM and IgG) can persist for years even after the clinical cure and treatment completion.[8] Even prolonging the duration of treatment with oral antibiotics has no role in the treatment of neuroborreliosis.[9] A repeat lumbar puncture in our case after 8 weeks of treatment, with complete clinical recovery, showed normalization of the CSF pleocytosis and protein but persistent hypoglycorrhachia. The pathogenic mechanism for hypoglycorrhachia consists of an admixture of increased utilization by the brain cells, a decreased availability of CSF because of decreased transport across the blood–brain barrier, and a reduced blood flow to the choroid plexus leading to a decreased delivery.[10] Since there was persistent hypoglycorrhachia even after complete treatment, we hypothesize that a decreased transport of glucose across the blood–brain barrier was a more plausible mechanism in Lyme's disease in our patient rather than an increased utilization in the brain.

To conclude, Lyme's meningitis forms an important differential diagnosis in the list of etiologies responsible for chronic meningitis with low CSF sugar values. The workup for Lyme's meningits must be considered once the common infections including tuberculosis have been ruled out in such a scenario. Moreover, it must be borne in mind that an improvement of hypoglycorrhachia should not be taken as a reliable indicator of therapy in proven cases of Lyme's disease since it may persist for a long time.

Authors' declaration

This report was presented at Child Neurocon 2017 and was awarded the first prize under the 'infectious disease' category at the conference.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the forms, the patient's legal authorized representative has given consent for her clinical information to be reported in the journal. They 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.
Hildenbrand P, Craven DE, Jones R, Nemeskal P. Lyme neuroborreliosis: Manifestations of a rapidly emerging zoonosis. Am J Neuroradiol 2009;30:1079-87.  Back to cited text no. 1
    
2.
Leen WG, De Wit CJ, Wevers RA, van Engelen BG, Kamsteeg EJ, Klepper J, et al. Child neurology: Differential diagnosis of a low CSF glucose in children and young adults. Neurology 2013;81:e178-81.  Back to cited text no. 2
    
3.
Cohn K, Thompson A, Shah S, Hines EM, Lyons TW, Welsh EJ, et al. Validation of a clinical prediction rule to distinguish Lyme meningitis from aseptic meningitis. Pediatrics 2012;129:46-53.  Back to cited text no. 3
    
4.
Dayan NE, Rubin LG, Di John D, Sood SK. Hypoglycorrhachia in Lyme meningitis. Pediatr Infect Dis J 2004;23:370-1.  Back to cited text no. 4
    
5.
Verma V, Roman M, Shah D, Zaretskaya M, Yassin MH. A case of chronic progressive Lyme encephalitis as a manifestation of late Lyme neuroborreliosis. Infect Dis Rep 2014;6:51-4.  Back to cited text no. 5
    
6.
Lakos A. CSF findings in Lyme meningitis. J Infect 1992;25:155-61.  Back to cited text no. 6
    
7.
Skogman BH, Glimåker K, Nordwall M, Vrethem M, Ödkvist L, Forsberg P. Long-term clinical outcome after Lyme neuroborreliosis in childhood. Pediatrics 2012;130:262-9.  Back to cited text no. 7
    
8.
Hammers-Berggren S, Lebech AM, Karlsson M, Ngsson BS, Hansen K, Stiernstedt G. Serological follow-up after treatment of patients with erythema migrans and neuroborreliosis. J Clin Microbiol 1994;32:1519-25.  Back to cited text no. 8
    
9.
Oksi J, Nikoskelainen J, Hiekkanen H, Lauhio A, Peltomaa M, Pitkäranta A, et al. Duration of antibiotic treatment in disseminated Lyme borreliosis: A double-blind, randomized, placebo-controlled, multicenter clinical study. Eur J Clin Microbiol Infect Dis 2007;26:571-81.  Back to cited text no. 9
    
10.
Viola GM. Extreme hypoglycorrhachia: Not always bacterial meningitis. Nat Rev Neurol 2010;6:637-41.  Back to cited text no. 10
    



 
 
    Tables

  [Table 1], [Table 2]



 

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