Atormac
brintellex
Neurology India
menu-bar5 Open access journal indexed with Index Medicus
  Users online: 881  
 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
 »Related articles
  »  Article in PDF (1,601 KB)
  »  Citation Manager
  »  Access Statistics
  »  Reader Comments
  »  Email Alert *
  »  Add to My List *
* Registration required (free)  

 
  In this Article
 »  Abstract
 » Introduction
 » Case Report
 » Discussion
 »  References
 »  Article Figures
 »  Article Tables

 Article Access Statistics
    Viewed3838    
    Printed92    
    Emailed1    
    PDF Downloaded85    
    Comments [Add]    
    Cited by others 2    

Recommend this journal

 


 
Table of Contents    
CASE REPORT
Year : 2013  |  Volume : 61  |  Issue : 4  |  Page : 414-418

Severe form of radiculo - myelo - neuropathy with meningo - encephalitis secondary to Angiostrongylus cantonensis infection: Unusual corpus callosal lesions and serial magnetic resonance imaging findings


1 Department of Neurology, National Institute of Mental Health and Neurosciences, Bengaluru, Karnataka, India
2 Department of Helminthology, Faculty of Tropical Medicine, Mahidol University, Bangkok, Thailand
3 Department of Neuromicrobiology, National Institute of Mental Health and Neurosciences, Bengaluru, Karnataka, India
4 Department of Neuroimaging and Interventional Radiology, National Institute of Mental Health and Neurosciences, Bengaluru, Karnataka, India

Date of Submission23-Apr-2013
Date of Decision27-May-2013
Date of Acceptance21-Jul-2013
Date of Web Publication4-Sep-2013

Correspondence Address:
Atchayaram Nalini
Department of Neurology, National Institute of Mental Health and Neurosciences, Hosur Road, Bangalore - 560 029, Karnataka
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.117613

Rights and Permissions

 » Abstract 

A 43-year-old man presented with the symptoms of recurrent lower abdominal pain, malaise and loss of appetite of 3-week duration, followed by acute onset of generalized paresthesias, fever and headache which progressed over few days to quadriparesis, altered sensorium, urinary and fecal incontinence. He had consumed raw tongue, liver, gall bladder and testicles of monitor lizard (Varanus bengalensis). Blood picture showed eosinophilia and cerebrospinal fluid (CSF) analysis revealed elevated protein and eosinophilia. Serum and CSF serology was positive for angiostrongyliasis. Magnetic resonance imaging showed focal hyperintense lesions in the corpus callosum and brainstem and an enhancing lesion in the cerebellum. Post-contrast T1-weighted axial images of spine showed evidence of cervical cord hyperintense lesions and root enhancement. Susceptibility weighted images/phase images showed unusual feature of multiple hemorrhagic lesions in the posterior fossa and supratentorial areas. Diffusion showed no restriction of corpus callosal lesions. Patient was treated with the high dose parenteral steroids with albendazole and at 6-month follow-up and had a remarkable recovery.


Keywords: Albendazole, angiostongyliasis, angiostrongylus cantonensis, meningoencephalitis, myelo-radiculo-neuropathy


How to cite this article:
Nalini A, Ramakrishna A, Dekumoy P, Kumar RR, Pakdee W, Saini J, Hegde VS. Severe form of radiculo - myelo - neuropathy with meningo - encephalitis secondary to Angiostrongylus cantonensis infection: Unusual corpus callosal lesions and serial magnetic resonance imaging findings. Neurol India 2013;61:414-8

How to cite this URL:
Nalini A, Ramakrishna A, Dekumoy P, Kumar RR, Pakdee W, Saini J, Hegde VS. Severe form of radiculo - myelo - neuropathy with meningo - encephalitis secondary to Angiostrongylus cantonensis infection: Unusual corpus callosal lesions and serial magnetic resonance imaging findings. Neurol India [serial online] 2013 [cited 2021 Jan 24];61:414-8. Available from: https://www.neurologyindia.com/text.asp?2013/61/4/414/117613



 » Introduction Top


Angiostongyliasis is a parasitic disease caused by human infection with the nematode larvae of Angiostrongylus cantonensis and is an endemic disease in Southeast Asia, China, the Pacific Basin and the Caribbean. The main clinical feature of the disease is eosinophilic meningoencephalitis [1],[2] and occasionally paralysis of the cranial nerves. [3],[4] Radicular pain, coma and respiratory failure have rarely been reported. [5] Imaging often reveals multiple micronodular enhancing brain lesions and linear enhancement in the pia mater. [6],[7],[8] We report a patient with the severe form of radiculo - myelo - neuropathy with meningoencephalitis secondary to A. cantonensis infection. This is probably the first report from India to be confirmed by immunoblot studies.


 » Case Report Top


A 43-year-old truck driver was evaluated on July 26, 2012 for recurrent lower abdominal pain, generalized malaise, loss of appetite and high grade fever and chills since mid-June 2012. At 15 days later, he developed a transient pin head sized non-pruritic vesicular rash over the right shoulder, which rapidly spread in the next 48 h to right side of the trunk, which disappeared over the next 3 days. Simultaneously, he developed severe cutaneous hypersensitivity over the entire right upper limb, which rapidly progressed over 48 h to involve the entire body up to the neck along with rapidly evolving are flexic quadriparesis with motor power 2/5 in the upper limbs and 0/5 motor power in the lower limbs, urinary and fecal incontinence, hyper somnolence and mute state followed by altered sensorium. 2 weeks prior to symptom onset he had consumed raw tongue, liver, gall bladder and testicles of monitor lizard. He also gave a history of consuming cooked rat snakes. Investigations performed during the 3 rd week of illness in another hospital had revealed normal hemogram, biochemistry and computed tomography brain. Initial magnetic resonance imaging (MRI) revealed multifocal linear asymmetric hyperintense lesions in the cerebellar white matter, left internal capsule and lobar white matter of both cerebral hemispheres on T2-weighted (T2W) imaging and hypointense lesions in the body of the corpus callosum on mid-sagittal T1-weighted (T1W) imaging [Figure 1].
Figure 1: First magnetic resonance imaging (MRI). T2‑weighted MR images at the level of middle cerebellar peduncle (a), midbrain (b), basal ganglia (c) and centrum semiovale (d) showing multifocal linear asymmetric hyperintense lesions in the left cerebellar white matter, left internal capsule and lobar white matter of both cerebral hemispheres. Hypointense lesions are seen in the body of corpus callosum on mid‑sagittal T1‑weighted images (e). Enhancement of lesions in left cerebellar white matter visualized on post‑contrast (f)

Click here to view


Cerebrospinal fluid (CSF) done at the first hospital was turbid with 300 cells (predominant polymorphs), elevated protein of 84 mg/dl and glucose of 45 mg/dl. He was treated as bacterial meningitis with no response. In the 4 th week of illness, he was evaluated at our Institute and neurological deficits were same as described. MRI revealed multiple scattered small punctuate areas of signal change in both cerebral white matter, majority appearing hyperintense on T2W/fluid attenuated inversion recovery (FLAIR) images and isointense on T1W images. Susceptibility-weighted images (SWI) revealed susceptibility within the lesions, which on phase images showed increased signal intensity suggestive of micro bleeds. Follow-up imaging at 1 and 5 months showed complete disappearance of a few lesions. Post-contrast study showed no significant enhancement of the residual lesions [Figure 2] and [Figure 3]. Nerve conduction studies during the 4 th week of illness revealed evidence of symmetrical motor and sensory axonal neuropathy with active ongoing denervation.
Figure 2: Follow‑up magnetic resonance imaging after 5 months shows patchy hyperintense signals in the bilateral cerebral white matter. (a and b). Lesions are isointense on T1‑weighted (T1W) images (c) post‑contrast T1W image shows absence of enhancement in the focal lesions (d) susceptibility‑weighted image shows multiple focal microbleeds in the left cerebral hemisphere (e) spine lesion has disappeared (f)

Click here to view
Figure 3: Sagittal T2‑weighted image done in the initial phase of the illness showing focal hyperintense lesions involving the corpus callosum and cervicomedullary junction. Magnetic resonance imaging after 5 months showing a decrease in the corpus callosum lesions and disappearance of the cervicomedullary lesion

Click here to view


Repeat CSF analysis [Figure 4] performed 7 weeks of onset of illness showed hazy CSF with 620 cells/cumm, elevated protein and normal glucose. An automated CSF analysis had identified the large number of eosinophils as neurtrophils and cytospin with manual cell counting revealed abundant eosinophils [Figure 5]. Serial CSF findings are given in [Table 1]. A clinical possibility of parasitic infection was considered, A. cantonensis in view of the patient's food habits. Two serum and four serial CSF samples were tested for angiostrongyliasis at the immunodiagnostic unit for helminthic infections, Department of Helminthology, Faculty of Tropical Medicine, Mahidol University, Thailand. Immunoblot of all samples demonstrated the diagnostic 31 kDa band for A. cantonensis antigen. All samples were negative for Gnathostoma spinigerum antigen. Serum and CSF serology was negative for other infections. Erythrocyte sedimentation rate was 45 mm at the 1 st h. Serum C3 and C4 levels were normal. Cluster of differentiation 4 count was 750 cells/cumm. Serum creatine kinase level was 563 IU/L. Visual evoked potentials (VEPs) revealed P100 latency of 99.9 ms in the right eye and 116.4 ms in the left eye. Patient was started on parenteral dexamethasone at 24 mg/day with which he had subtle but dramatic improvement within 48 h. After parenteral steroid for 2 weeks, he was shifted to oral steroid for 4 weeks. One week after steroids, albendazole was started in a small dose and gradually increased to 800 mg/day and continued in this dosage for a total of 4 weeks. Patient showed a steady improvement in the neurological deficits and at the end of 6 weeks had grade 4 power, but with severe foot drop and atonic bladder. He could walk with minimal support. At the last evaluation in January 2013, higher mental functions were normal, ambulant with 4+/5 power in upper limbs and 4/5 in the lower limbs with the minimal foot drop. Normal bladder function was attained at the end of 4 months. A repeat VEP revealed P100 latency of 99.6 ms in the right eye and 114.4 ms in the left eye.
Figure 4: Two serial hazy looking cerebrospinal fluid obtained at 1 weeks interval

Click here to view
Figure 5: Serial cerebrospinal fluid smear showing progressive reduction in the number of eosinophils

Click here to view
Table 1: Serial cerebrospinal fluid analysis findings with eosinophil counts

Click here to view



 » Discussion Top


Increasing globalization, traveling, migration and changing dietary habits of people and with newly emerging delicacies, hitherto, not practiced in certain countries are expanding the range of potential infectious diseases that are encountered in clinical practice. In this patient, multiaxial neurological presentation with eosinophilia in peripheral blood and CSF and history of consumption of raw flesh and organs of monitor lizard and also cooked rat snakes gave a clue for possible infection with A. cantonensis or G. spinigerum. Definitive diagnosis of angiostongyliasis would require identification of larvae or young adults in human tissue, CSF or eye, which is very rarely achievable. Serology remains the main stay for the diagnosis. Our patient met the criteria for diagnosis of eosinophilic meningitis. [9] The diagnosis of angiostongyliasis was confirmed by positive immunoblot analysis for specific 31 kDa band for A. cantonensis from serial CSF and serum samples. Initially, we faced a few challenges. Firstly, we had not suspected eosinophilic meningoencephalitis in this patient with this clinical presentation. Further, an automated CSF cell count initially misidentified a large number of eosinophils as neutrophils. Only a cytospin analysis with manual counting identified the abundant number of eosinophils, which made us to consider eosinophilic meningitis and further work-up.

The most common clinical manifestation of angiostrongyliasis is self-limiting eosinophilic meningoencephalitis and severe myelo-encephalitis form has been reported rarely. [10],[11],[12],[13] In Gnathostoma infection dermatological manifestations such as migrating panniculitis, eruptions and pseudo furunculosis and radiculomyelitis, encephalitis, paralysis and cerebral hemorrhage are common; although, these features have been rarely described with A. cantonensis infections. [14],[15],[16] Our patient had the clinical features described classically in A. cantonensis and overlapped with features of Gnathostoma infection. [15] A. cantonensis infection in our patient was probably acquired by the consumption of raw tongue, liver, gall bladder and testicles of monitor lizard (Iguana). In almost all reports from other countries, consumption of raw snails, slugs and contaminated green leafy vegetables causes A. cantonensis infection.

Nerve conduction studies showed evidence of moderate axonal motor sensory symmetrical neuropathy. Although radiculopathy is reported with A. cantonensis infection, [15] neuropathy has not been described earlier. However, a case of A. cantonensis infection with meningitis and lumbosacral myeloradiculopathy with normal MRI of the brain and spinal cord had been reported. [17] In our patient, unilateral prolonged P100 latency suggests possible optic neuropathy with no visual symptoms. Optic nerve involvement with blurred vision has been reported earlier only once. [18]

The reports of MRI features in A. cantonensis infection are limited, particularly in the myelitis form. [19],[20],[21] In most reported studies, MRI brain was normal. [8],[14],[16],[18] MRI features reported by Kanpittaya et al. included prominence of the Virchow-Robin spaces, subcortical enhancing lesions and abnormal high T2 signal lesions in the periventricular region. [21] Among 13 Thai laborers, in nine patients MRI showed non-specific meningeal enhancement and increased bilateral signals in the globus pallidus on T1W MRI. Relative hyperintense signal on Serum C3 and C4 levels were normal. CD4 count was 750cells/cumm. in the cerebral peduncles were noted in two patients. [8] In our patient, MRI revealed multiple scattered small punctuate areas of signal change in both cerebral and cerebellar white matter. SWI and phase images showed evidence of microbleeds. However, this extent of microbleeds is unusual in A. cantonensis infection and corpus callosal lesions have not been described. In addition, our patient had spinal cord involvement with radicular affection, which has been previously reported in only one paper. [7] It is noteworthy that our patient had persisting parenchymal hemorrhagic lesions probably secondary to migrating worms which has not been reported in A. cantonensis infection. [15] MRI of spinal cord may show scattered or focal signal abnormalities, which may enhance with contrast administration. Nerve root enhancement is also known. [7] Our patient had both cervical cord lesion and nerve root enhancement. Neurological disease caused by G. spinigerum is an important differential diagnosis and MRI patterns of the two diseases is also similar; however, G. spinigerum shows greater intracerebral hemorrhage and causes myelitis more often. [21] In an another study contrast, MRI had revealed a multiple round or oval enhancing nodules, seen on T1W image and a few were stick-shaped lesions along with mild ventricular enlargement in two patients. Follow-up MRI had shown severe lesions between 5 th and 8 th weeks and it took at least 4-8 weeks for a lesion to resolve completely while the larger lesions needed more than 22 weeks. [7] In our case, follow-up MRI at 1 month showed complete disappearance of few lesions while some lesions unchanged. No significant change in the morphology of lesions was noted. Post-contrast study showed no significant enhancement of the lesions. MRI performed at 5 months follow-up revealed partial resolution of the signal changes however, corpus callosum and cerebral white matter lesions could still be well-appreciated.

The serial CSF had shown hazy fluid with polymorphic pleocytosis, raised protein and normal glucose levels. This is the typical CSF findings described earlier. [8] In an earlier report, of the 10 patients with eosinophilic meningoencephalitis secondary to A. cantonensis, eight patients had consumed raw flesh of monitor lizard. [22] It has been established that the yellow tree monitors (Varanus bengalensis) carry the infective stage of A. cantonensis due to their snail eating habit. [23] Our patient had consumed raw tongue and organs of monitor lizard as a native treatment for diabetes mellitus. In these clinically challenging scenarios and the presence of emerging global infections, physicians and neurologists need to be aware of these rare neuroinfections.

 
 » References Top

1.Pascual JE, Bouli RP, Aguiar H. Eosinophilic meningoencephalitis in Cuba, caused by Angiostrongylus cantonensis. Am J Trop Med Hyg 1981;30:960-2.  Back to cited text no. 1
[PUBMED]    
2.Legrand G, Angibaud G. Eosinophilic meningitis due to Angiostrongylus cantonensis. Rev Neurol (Paris) 1998;154:236-42.  Back to cited text no. 2
[PUBMED]    
3.Kuberski T, Bart RD, Briley JM, Rosen L. Recovery of Angiostrongylus cantonensis from cerebrospinal fluid of a child with eosinophilic meningitis. J Clin Microbiol 1979;9:629-31.  Back to cited text no. 3
[PUBMED]    
4.Punyagupta S, Juttijudata P, Bunnag T. Eosinophilic meningitis in Thailand. Clinical studies of 484 typical cases probably caused by Angiostrongylus cantonensis. Am J Trop Med Hyg 1975;24:921-31.  Back to cited text no. 4
[PUBMED]    
5.Sawanyawisuth K, Tiamkao S, Kanpittaya J, Dekumyoy P, Jitpimolmard S. MR imaging findings in cerebrospinal gnathostomiasis. AJNR Am J Neuroradiol 2004;25:446-9.  Back to cited text no. 5
[PUBMED]    
6.Hwang KP, Chen ER. Clinical studies on Angiostrongyliasis cantonensis among children in Taiwan. Southeast Asian J Trop Med Public Health 1991;22 Suppl: 194-9.  Back to cited text no. 6
[PUBMED]    
7.Jin E, Ma D, Liang Y, Ji A, Gan S. MRI findings of eosinophilic myelomeningoencephalitis due to Angiostrongylus cantonensis. Clin Radiol 2005;60:242-50.  Back to cited text no. 7
[PUBMED]    
8.Tsai HC, Liu YC, Kunin CM, Lee SS, Chen YS, Lin HH, et al. Eosinophilic meningitis caused by Angiostrongylus cantonensis: Report of 17 cases. Am J Med 2001;111:109-14.  Back to cited text no. 8
[PUBMED]    
9.Kuberski T. Eosinophils in cerebrospinal fluid: Criteria for eosinophilic meningitis. Hawaii Med J 1981;40:97-8.  Back to cited text no. 9
[PUBMED]    
10.Koo J, Pien F, Kliks MM. Angiostrongylus (Parastrongylus) eosinophilic meningitis. Rev Infect Dis 1988;10:1155-62.  Back to cited text no. 10
[PUBMED]    
11.Enzenauer RW, Yamaoka RM. Eosinophilic meningitis and hydrocephalus in an infant. Arch Neurol 1982;39:380-1.  Back to cited text no. 11
[PUBMED]    
12.Prociv P, Tiernan JR. Eosinophilic meningoencephalitis with permanent sequelae. Med J Aust 1987;147:294-5.  Back to cited text no. 12
[PUBMED]    
13.Ko RC, Chiu MC, Kum W, Chan SH. First report of human angiostrongyliasis in Hong Kong diagnosed by computerized axial topography (CAT) and enzyme linked immunosorbent assay. Trans R Soc Trop Med Hyg 1984;78:354-5.  Back to cited text no. 13
[PUBMED]    
14.Ramirez-Avila L, Slome S, Schuster FL, Gavali S, Schantz PM, Sejvar J, et al. Eosinophilic meningitis due to Angiostrongylus and Gnathostoma species. Clin Infect Dis 2009;48:322-7.  Back to cited text no. 14
[PUBMED]    
15.Graeff-Teixeira C, da Silva AC, Yoshimura K. Update on eosinophilic meningoencephalitis and its clinical relevance. Clin Microbiol Rev 2009;22:322-48.  Back to cited text no. 15
[PUBMED]    
16.Lo Re V 3 rd , Gluckman SJ. Eosinophilic meningitis. Am J Med 2003;114:217-23.  Back to cited text no. 16
    
17.Maretiæ T, Peroviæ M, Vince A, Lukas D, Dekumyoy P, Begovac J. Meningitis and radiculomyelitis caused by Angiostrongylus cantonensis. Emerg Infect Dis 2009;15:996-8. Available from: March 2013.  Back to cited text no. 17
    
18.Jin EH, Ma Q, Ma DQ, He W, Ji AP, Yin CH. Magnetic resonance imaging of eosinophilic meningoencephalitis caused by Angiostrongylus cantonensis following eating freshwater snails. Chin Med J (Engl) 2008;121:67-72.  Back to cited text no. 18
[PUBMED]    
19.Noskin GA, McMenamin MB, Grohmann SM. Eosinophilic meningitis due to Angiostrongylus cantonensis. Neurology 1992;42:1423-4.  Back to cited text no. 19
[PUBMED]    
20.Hsu WY, Chen JY, Chien CT, Chi CS, Han NT. Eosinophilic meningitis caused by Angiostrongylus cantonensis. Pediatr Infect Dis J 1990;9:443-5.  Back to cited text no. 20
[PUBMED]    
21.Kanpittaya J, Jitpimolmard S, Tiamkao S, Mairiang E. MR findings of eosinophilic meningoencephalitis attributed to Angiostrongylus cantonensis. AJNR Am J Neuroradiol 2000;21:1090-4.  Back to cited text no. 21
[PUBMED]    
22.Parameswara K. Case series of eosinophilic meningoencephalitis from South India. Ann Indian Acad Neurol 2006;9:217-22.  Back to cited text no. 22
    
23.Radomyos P, Tungtrongchitr A, Praewanich R, Khewwatchan P, Kantangkul T, Junlananto P, et al. Occurrence of the infective stage of Angiostrongylus cantonensis in the yellow tree monitor (Varanus bengalensis) in five Provinces of Thailand. Southeast Asian J Trop Med Public Health 1994;25:498-500.  Back to cited text no. 23
[PUBMED]    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]
 
 
    Tables

  [Table 1]

This article has been cited by
1 In Vivo Brain MR Imaging at Subnanoliter Resolution: Contrast and Histology
Takashi WATANABE,Jens FRAHM,Thomas MICHAELIS
Magnetic Resonance in Medical Sciences. 2016; 15(1): 11
[Pubmed] | [DOI]
2 Angiostrongylus cantonensis: a review of its distribution, molecular biology and clinical significance as a human pathogen
JOEL BARRATT,DOUGLAS CHAN,INDY SANDARADURA,RICHARD MALIK,DEREK SPIELMAN,ROGAN LEE,DEBORAH MARRIOTT,JOHN HARKNESS,JOHN ELLIS,DAMIEN STARK
Parasitology. 2016; 143(09): 1087
[Pubmed] | [DOI]



 

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