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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 9  |  Issue : 4  |  Page : 438-441

A rare co-occurrence of optochiasmatic tuberculoma and guillain-barre syndrome as a paradoxical reaction in tubercular meningitis


Department of Medicine, VMMC and Safdarjung Hospital, New Delhi, India

Date of Submission31-Jul-2020
Date of Decision03-Aug-2020
Date of Acceptance06-Aug-2020
Date of Web Publication15-Dec-2020

Correspondence Address:
Mithu Bhowmick
Department of Medicine, VMMC and Safdarjung Hospital, New Delhi - 110 029
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijmy.ijmy_133_20

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  Abstract 


Tuberculosis (TB) is the leading infectious cause of death worldwide. Tubercular meningitis is the most severe form of extrapulmonary TB which carries high morbidity and mortality. Intracranial tuberculoma may develop paradoxically during the treatment of tubercular meningitis, which is a quite rare occurrence. There are many reported cases of paradoxical development of optochiasmatic tuberculoma during the treatment of tubercular meningitis, causing severe visual impairment. Association of Guillain-Barré syndrome with TB is a rare entity. However, the co-occurrence of optochiasmatic tuberculoma and Guillain-Barré syndrome is even rarer and not reported before in literature. Here, we present the first case of co-occurrence of optochiasmatic tuberculoma and Guillain-Barré Syndrome, developing as a paradoxical reaction in a 23-year-old male.

Keywords: Guillain-Barré syndrome, optochiasmatic tuberculoma, paradoxical reaction


How to cite this article:
Bhowmick M, Lal M, Chakravarthy P S, Kant J. A rare co-occurrence of optochiasmatic tuberculoma and guillain-barre syndrome as a paradoxical reaction in tubercular meningitis. Int J Mycobacteriol 2020;9:438-41

How to cite this URL:
Bhowmick M, Lal M, Chakravarthy P S, Kant J. A rare co-occurrence of optochiasmatic tuberculoma and guillain-barre syndrome as a paradoxical reaction in tubercular meningitis. Int J Mycobacteriol [serial online] 2020 [cited 2021 Jan 16];9:438-41. Available from: https://www.ijmyco.org/text.asp?2020/9/4/438/303439




  Introduction Top


Tuberculosis (TB) is the leading infectious cause of death worldwide.[1] Central nervous system (CNS) TB is highly devastating and accounts for approximately 1% of all cases of TB. Tubercular meningitis is the most severe form of extrapulmonary TB, which carries a high morbidity and mortality.[2] Intracranial tuberculoma may develop as a paradoxical reaction during treatment of tubercular meningitis, and may rarely involve the anterior optic pathway including optic chiasma causing severe vision impairment.

Guillain-Barré syndrome is an acute paralytic peripheral neuropathy with an annual incidence of 0.8–1.9 cases/100,000 people per year.[3] Various antecedent infectious (Campylobacter jejuni, Mycoplasma pneumoniae, EB virus, etc.,) and noninfectious (vaccines) have been implicated as a trigger for the disease, probably due to an aberrant immune response.[3] There are numerous reported cases of optochiasmatic tuberculoma developing as a paradoxical reaction, days to months after treatment of tubercular meningitis. However, the co-occurrence of optochiamatic tuberculoma and Guillain-Barré syndrome, developing as a paradoxical reaction in tubercular meningitis is not reported before in literature. Here, we report the first case of this kind in a 23-year-old male.


  Case Report Top


A 23-year-old male taking antitubercular drugs with a tapering dose of steroid for 2 months for tubercular meningitis, presented with complaints of progressive diminution of vision for 20 days. The patient reported full compliance to antitubercular drugs and there was a gradual improvement of his symptoms. However, after an initial improvement of symptoms, he started having blurring of his vision, and over 20 days course, it was progressive to such an extent that he could only perceive light. He also noticed sudden-onset weakness in both distal lower limbs 7 days before admission and the weakness was so progressive that he could barely move his lower limb on presentation to hospital. There was no respiratory distress. Examination of the patient revealed the only perception of light in both the eyes and bilateral pupil were nonreactive to light. Lower limb examination is suggestive of motor weakness with hypotonia, areflexia, and power of 2/5 in both lower limbs. The plantar reflexes were mute. The sensory system examination was unremarkable. The upper limb examination did not reveal any abnormality. His complete blood profile, liver function test, renal function test, and serum electrolytes all within the normal limits. Serology for hepatitis B and C, HIV was negative. The fundus examination revealed that bilateral pale optic disc. Magnetic resonance imaging (MRI) of the brain with contrast revealed multiple peripherally enhancing conglomerated lesions in bilateral frontal and temporal regions in the perisylvian location and in the basal cistern, including suprasellar and interpeduncular cisterns. Anteriorly, the lesions involved the optic chiasma and hypothalamus. Posteriorly, the lesions are seen extending into ventral midbrain and inferiorly until the pituitary fossa. The lesions were isointense on T1WI with hyperintense rim and hypointense with a variable degree of liquefaction on T2WI [Figure 1] and [Figure 2]. On magnetic resonance spectroscopy, lipid lactate peak was seen at 1.3 ppm. Visual evoked potentials were nonrecordable in both the eyes. Electrodiagnostic studies of both lower limbs were suggestive of acute inflammatory demyelinating polyradiculopathy [Figure 3].
Figure 1: (a and b) T1W MRI brain showing multiple peripherally enhancing, variable-sized conglomerated lesions in the basal cistern, including suprasellar and interpeduncular cisterns around optic chiasma. The lesion was seen extending in the bilateral temporal region (right more than left) in perisylvian location

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Figure 2: (a and b) T2W MRI of the brain showing rounded lesion around optic chiasma. Posteriorly, the lesion was seen extending into ventral midbrain and inferiorly until pituitary fossa

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Figure 3: Nerve conduction study suggestive of acute inflammatory demyelinating polyradiculopathy

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Based on clinical, imaging, and nerve conduction studies a presumptive diagnosis of optochiasmatic tuberculoma and Guillain-Barré syndrome as a paradoxical response to treatment for tubercular meningitis was made. The patient was treated with intravenous immune globulin, intravenous dexamethasone, and antitubercular drugs continued. On day 7 of hospital stay, he had improvement in the power of both lower limb, but visual acuity remains unchanged. The patient refused for neurosurgical intervention. Based on the response to an intravenous immune globulin, an immunological basis of the phenomenon is thought. The patient was discharged on day 10 with the power of 4/5 in both lower limb, and with the advice of continuation of steroid and antitubercular drugs. The patient was kept under the rehabilitation program.


  Discussion Top


Paradoxical reaction in tubercular meningitis is characterized by either worsening of preexisting tubercular lesion or the appearance of the new tubercular lesion in a patient who shows initial improvement following antitubercular treatment.[4] The exact pathogenesis of this phenomenon is poorly elucidated. The probable hypothesis for this inexplicable phenomenon is related to an aberrant immunological response to mycobacterial antigen.[4],[5] Based on the same pathophysiology, Dev et al. also described a case of tuberculous encephalopathy without meningitis in a patient with disseminated TB.[6]

Vision loss due to optochiasmatic tuberculoma is a rare occurrence in tubercular meningitis. The vision loss may be attributed to gradual compression of optic chiasma and optic nerve.[7] Contrast MRI of the brain helps in the early detection of this condition. In most cases resolution of the lesion usually occurs with continued treatment of antitubercular drugs and prolonged course of steroid. In the context of tuberculoma compromising optic pathway, prompt surgical intervention is required.

Guillain-Barré syndrome is an immune-mediated acute paralytic neuropathy. Association of Guillain-Barré syndrome with CNS TB is a rare entity. The pathophysiology is linked to molecular mimicry between microbial and host antigen leading to aberrant immunological response targeting spinal roots and peripheral nerves.[3] The disease runs a monophasic course and presents with acute flaccid paralysis usually starts in distal lower extremities and ascends proximally. Respiratory failure can develop in 20%–30% of patients requiring invasive ventilation.[3] The diagnosis of Guillain-Barré syndrome is established using the Brighton criteria which include clinical assessment, nerve conduction studies, cerebrospinal fluid (CSF) findings, and exclusion of other causes.[8] CSF picture commonly reveals albumin cytological dissociation but CSF may be normal if obtained in 1st week of illness.[7] Our case meets Brighton criteria-level 2 of diagnostic certainty. Intravenous immune globulin and plasma exchange are currently considered the standard of care in Guillain-Barré syndrome.

Our patient developed this clinical picture after an initial improvement of symptoms which supports a diagnosis of paradoxical reaction. Clinical improvement of symptoms following immunomodulatory therapy further supported the diagnosis.

This case report enlightens the treating physician about this rare condition which is very crucial, as the paradoxical reaction may lead to the erroneous diagnosis of treatment failure or drug toxicity. Early diagnosis is imperative to minimize the neurological complications and morbidity.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understand that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
MacNeil A, Glaziou P, Sismanidis C, Maloney S, Floyd K. Global Epidemiology of Tuberculosis and Progress Toward Achieving Global Targets-2017. MMWR Morb Mortal Wkly Rep 2019;68:263-6.  Back to cited text no. 1
    
2.
Cherian A, Thomas SV. Central nervous system tuberculosis. Afr Health Sci 2011;11:116-27.  Back to cited text no. 2
    
3.
Willison HJ, Jacobs BC, Van Doorn PA. Guillain-Barré syndrome. Lancet 2016;388:717-27.  Back to cited text no. 3
    
4.
Singh AK, Malhotra HS, Garg RK, Jain A, Kumar N, Kohli N, et al. Paradoxical reaction in tuberculous meningitis: Presentation, predictors and impact on prognosis. BMC Infect Dis 2016;16:306.  Back to cited text no. 4
    
5.
Nicolls DJ, King M, Holland D, Bala J, del Rio C. Intracranial tuberculomas developing while on therapy for pulmonary tuberculosis. Lancet Infect Dis 2005;5:795-801.  Back to cited text no. 5
    
6.
Dev N, Bhowmick M, Chaudhary S, Kant J. Tuberculous encephalopathy without meningitis: A rare manifestation of disseminated tuberculosis. Int J Mycobacteriol 2019;8:406-8.  Back to cited text no. 6
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7.
Poon WS, Ahuja A, Li AK. Optochiasmatic tuberculoma causing progressive visual failure: When has medical treatment failed? Postgrad Med J 1993;69:147-9.  Back to cited text no. 7
    
8.
Sejvar JJ, Kohl KS, Gidudu J, Amato A, Bakshi N, Baxter R, et al. Guillain-Barré syndrome and Fisher syndrome: Case definitions and guidelines for collection, analysis, and presentation of immunization safety data. Vaccine 2011;29:599-612.  Back to cited text no. 8
    


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  [Figure 1], [Figure 2], [Figure 3]



 

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