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Table of Contents    
LETTER TO EDITOR
Year : 2018  |  Volume : 66  |  Issue : 4  |  Page : 1185-1187

Chronic pachymenigitis with dural venous sinus thrombosis: An unusual presentation of cranial melioidosis


1 Department of Neurological Sciences, Christian Medical College, Vellore, Tamil Nadu, India
2 Department of Pathology, Christian Medical College, Vellore, Tamil Nadu, India

Date of Web Publication18-Jul-2018

Correspondence Address:
Dr. Krishnaprabhu Raju
Department of Neurological Sciences, Christian Medical College, Vellore - 632 004, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.237015

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How to cite this article:
Nayak R, Patel B, Raju K. Chronic pachymenigitis with dural venous sinus thrombosis: An unusual presentation of cranial melioidosis. Neurol India 2018;66:1185-7

How to cite this URL:
Nayak R, Patel B, Raju K. Chronic pachymenigitis with dural venous sinus thrombosis: An unusual presentation of cranial melioidosis. Neurol India [serial online] 2018 [cited 2018 Aug 17];66:1185-7. Available from: http://www.neurologyindia.com/text.asp?2018/66/4/1185/237015




Sir,

A 23-year old student presented with a 9-month history of progressive diplopia on looking towards the left and a 6-month history of decreased hearing in the left ear. He was initially evaluated for the above complaints at another center where he was diagnosed to be having left sigmoid and transverse sinus thrombosis with left mastoiditis and was treated with antituberculous therapy, steroids, and anticoagulants; however, there was no improvement in the symptoms.

Examination showed the left third cranial nerve (CN) palsy and paresis of the left seventh, eighth, ninth, and tenth CNs. Magnetic resonance imaging (MRI) of the brain with contrast showed pachymeningeal thickening and abnormal enhancement along the left temporal region, the petrous apex, and within the left mastoid air cells [Figure 1]. Magnetic resonance venogram (MRV) showed chronic partial thrombosis of the left transverse-sigmoid sinus and left jugular vein. Differential diagnosis of infective etiologies such as fungal and mycobacterial tuberculosis, inflammatory diseases such as sarcoidosis and immunoglobulin G4 disease, and malignant conditions such as lymphomas were considered. As the patient showed gradual deterioration on steroids, antituberculous, and anticoagulant medications, a surgical biopsy was undertaken, which was suggestive of chronic meningitis [Figure 2]. Cultures from the biopsy grew Burkholderia pseudomallei. He was treated with 2 g intravenous ceftazidime every 6 hours for 6 weeks, followed by an oral cotrimoxazole regimen for 6 months. At the 6-month follow-up, there was a dramatic improvement in the symptoms, and MRI brain showed complete disappearance of meningeal enhancement and clearance of the left mastoid enhancement [Figure 3].
Figure 1: (a) MRI brain showing pachymenigeal thickening and enhancement noted along the left temporal lobe, left-sided posterior fossa, left petrous bone and clivus, extending into the left tentorium cerebelli. There is also involvement of the left cavernous sinus along with the left sphenoid and basifrontal bones. (b) Abnormal enhancement is seen in the left mastoid air cells and dura along the petrous apex. (c) MR venogram shows chronic partial thrombosis of the left transverse-sigmoid sinus and left jugular vein

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Figure 2: Dense infiltrates of plasma cells and lymphocytes along with areas of fibrosis suggestive of chronic pachymeningitis. (a) ×100, (b) ×200

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Figure 3: (a) MRI brain with contrast done at the 6-month follow up showing complete disappearance of meningeal enhancement. (b) MRI brain with contrast done at the 6-month follow up showing clearance of the left mastoid disease

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Melioidosis is a systemic disease associated with abscesses in multiple organs caused by B. pseudomallei,[1] which is a Gram-negative soil-dwelling organism. Although it is endemic in the southeast Asian countries and northern Australia, neurologic involvement is accounted for in only 3% of all the cases.[2]

Cranial manifestations of melioidosis also include scalp abscesses, osteomyelitis, meningitis, subdural empyema, encephalitis, brain abscesses, and meningeal irritation.[3] [Table 1] shows the recently reported cases of melioidosis presenting as mass lesions and osteomyelitis.[4],[5],[6],[7],[8]
Table 1: Recent reported cases of cranial melioidosis presenting as mass lesion and osteomyelitis

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Dural sinus thrombosis complicating septic melioidosis was first reported from Thailand where dural venous sinus thrombosis was attributed to the hypercoagulable state secondary to septicemia.[9]

The uniqueness of our case lies in the absence of a mass lesion, osteomyelitis, or septic melioidosis. It is the first reported case, to the best of our knowledge, where melioidosis is presented with only pachymeningitis and dural venous sinus thrombosis.

Treatment of neurological melioidosis includes an initial intensive therapy by parenteral ceftazidime (50 mg/kg up to 2 g intravenously every 6 hours or meropenem (25 mg/kg up to 1 g intravenously every 8 hours) for a period of 6–8 weeks, followed by the eradication therapy by oral cotrimoxazole (trimethoprim–sulfamethoxazole) at a dose of 40/8 mg/kg (up to 1,600/320 mg) every 12 hours, and doxycycline 2 mg/kg, (up to 100 mg) every 12 hours for 6 months.[3],[8],[9],[10]

Melioidosis should be considered in the differential diagnosis of chronic pachymeningitis, even in the absence of brain abscess and predisposing factors, especially when the patient is not responding to steroids and empirical antituberculous drugs. This ensures an early diagnosis and treatment and prevents mortality and morbidity.

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.
Howe C, Sampath A, Spotnitz M: The pseudomallei group: A review. J Infect Dis 1971;124:598-606.  Back to cited text no. 1
    
2.
Currie BJ, Ward L, Cheng AC. The epidemiology and clinical spectrum of melioidosis: 540 cases from the 20 year Darwin prospective study. Trop Dis 2010;4:900.  Back to cited text no. 2
    
3.
Kumar GS, Raj PM, Chacko G, Lalitha MK, Chacko AG, Rajshekhar V. Cranial melioidosis presenting as a mass lesion or osteomyelitis. J Neurosurg 2008;108:243-7.  Back to cited text no. 3
    
4.
Chadwick DR, Ang B, Sitoh YY, Lee CC. Cerebral melioidosis in Singapore: A review of five cases. Trans R Soc Trop Med Hyg 2002;96:72-6.  Back to cited text no. 4
    
5.
Bergin P, Boyes L, Sage M. Cerebral melioidosis. Australas Radiol 2005;49:79-83.  Back to cited text no. 5
    
6.
Kuan YC, How SH, Ng TH, Fauzi AR. The man with the boggy head: Cranial melioidosis. Singapore Med J 2010;51:e43-5.  Back to cited text no. 6
    
7.
Naha K, Dasari S, Kusugodlu R, Prabhu M. Cranial melioidosis with extradural extension after a fall in the bathroom. Australasian Med J 2012;5:455.  Back to cited text no. 7
    
8.
Saravu K, Kadavigere R, Shastry AB, Pai R, Mukhopadhyay C. Neurologic melioidosis presented as encephalomyelitis and subdural collection in two male labourers in India. J Infect Dev Ctries 2015;9:1289-93.  Back to cited text no. 8
    
9.
Niyasom S, Sithinamsuwan P, Udommongkol C, Suwantamee J. Dural sinus thrombosis in melioidosis: The first case report. J Med Assoc Thai 2006;89:242-7.  Back to cited text no. 9
    
10.
Currie BJ, Fisher DA, Howard DM, Burrow JN, Lo D, Selva-nayagam S, et al. Endemic melioidosis in tropical northern Australia: A 10-year prospective study and review of the literature. Clin Infect Dis 2000;31:981-6.  Back to cited text no. 10
    


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