Neurobrucellosis presenting as an intra-medullary spinal cord abscess
© Vajramani et al; licensee BioMed Central Ltd. 2005
Received: 05 June 2005
Accepted: 16 September 2005
Published: 16 September 2005
Of the diverse presentation of neurobrucellosis, intra-medullary spinal cord abscess is extremely rare. Only four other cases have been reported so far. We present a case of spinal cord intra-medullary abscess due to Brucella melitensis.
A forty-year-old female presented with progressive weakness of both lower limb with urinary incontinence of 6 months duration. She was febrile. Neurological examination revealed flaccid areflexic paraplegia with T10 below sensory impairment including perianal region. An intramedullary mass was diagnosed on Magnetic Resonance Image (MRI) scan extending from T12 to L2. At surgery, a large abscess was encountered at the conus medullaris, from which Brucella melitensis was grown on culture. She was started on streptomycin and doxycycline for 1 month, followed by rifampicin and doxycycline for 1 month. At 2-year follow-up, she had recovered only partially and continued to have impaired bladder function.
Neurobrucellosis, if not treated early, can result in severe neurological morbidity and sequale, which may be irreversible. Hence it is important to consider the possibility of neurobrucellosis in endemic region and treat aggressively.
The presentation of neurobrucellosis, which encompass neuro-psychiatric disorders in brucellosis, is varied. It can present in acute form as meningoencephalitis or in chronic form, where there is involvement of peripheral nervous system or central nervous system. Chronic form includes epidural granuloma, demyelination of brain or spinal nerve roots, long tract degeneration etc. Meningitis has been the most frequent presentation, occurring in about 50% of the cases . Extensive intra-medullary spinal cord abscess due to Brucella, is exceedingly rare. Only four other cases have been reported so far [2–5]. We present a case of intra-medullary spinal cord abscess in which Brucella melitensis was cultured from pus.
Pus revealed gram-negative bacilli. It was inoculated aerobically [Brucella agar, chocolate and MacConkey media], and anaerobically [Kanamycin-vancomycin laked sheep blood agar (KVLB) and Bacteroides bile esculin agar (BBE)]. Brucella agar and CA were incubated in CO2 jar and after 2 days minute translucent colonies were seen. Gram stain from culture showed gram-negative bacilli. Oxidase, catalase, and urease test were positive. There was no H2S production and it was resistant to dye inhibition. The organism was confirmed as Brucella melitensis [6, 7]. The organism isolated in blood culture taken preoperatively, also was identified as Brucella melitensis.
Postoperatively she had fever, headaches and vomiting lasting for about 1 week. It subsided once antibiotics were instituted. She was started on injection streptomycin 1 gm once a day for 1 month with oral doxycycline 100 mgm twice a day for 1 month. After one month she received oral rifampicin 450 mgm once a day with oral doxycycline 100 mgm twice a day for 1 month. Dexamethasone was given only perioperatively and was rapidly tapered and stopped in the post-operative period. Post operatively she gradually improved in neurological status. At 2-year follow up she had grade 3/5 power in both lower limbs and was mobilising on a wheel chair. The urinary symptoms did not resolve and she continues to be on Foley catheter. She refused a repeat MRI scan, as she could not afford it.
Brucellosis is a common zoonosis in many parts of the world. Various types of central nervous system (CNS) involvement in brucellosis have been reported [1, 8–11], the estimated incidence varying from 5–25% in different series, with an average of 3–5% [1, 11, 12]. The exact pathogenesis of CNS involvement is not clear. Various mechanisms have been postulated. It is known that Brucella organisms are capable of prolonged intracellular survival within phagocytes. Decreased host immunity may allow the organisms to proliferate . The organism may act directly or indirectly through its endotoxins . Immune mediated demyelination has been proposed to explain certain chronic forms of neurobrucellosis . The cord or nerve root may secondarily be involved due to spondylitis, vasculitis and arachnoiditis .
Cases of neurobrucellosis with intramedullary spinal cord involvement.
Cokca et al, 1994
Regular consumption of cows milk, living in rural area near breeding animals
Intramedullary dermoid cyst (T11-L2)
Brucella abortus biotype 3
Brucella abortus biotype 3
Bingol et al, 1999.
Raising sheep and consuming raw milk
Intramedullary granuloma at T5
Novati et al, 2002.
Consumption of fresh goats cheese
Intramedullary abscess at T3
Helvaci et al, 2002
Consumption of cheese made from raw goats milk
Intramedullary abscess at T11-T12
Present case, 2005
Living near abattoir, consumption of meat and goat's milk.
Intramedullary abscess at Conus
In the present case, there was direct involvement of spinal cord by the Brucella melitensis leading to a chronic progressive neurological impairment. Despite the raised pre-operative titres of antibody to brucella antigen and high prevalence of brucellosis in this region, the possibility of the cord lesion to be of Brucella origin was not considered pre-operatively, probably because of the rarity of brucella abscess in the spinal cord. The post-operative fever and headache could have been due to transient meningitis resulting from contamination of the CSF space during surgery. This, fortunately, responded well to the antibiotics.
There are no specific guidelines regarding the antibiotic regimens and duration of treatment for neurobrucellosis. The duration of treatment varies from 8 weeks to 2 year depending upon individual cases, surgical or medical line of treatment and response to the treatment. Drugs such as doxycycline, rifampicin and trimethoprim-sulfamethoxazole have been found effective due to their good CNS penetration and synergistic actions [10, 14]. Tetracycline and streptomycin are good for systemic brucellosis, although their CNS penetration is poor. However, as most of these patients have systemic brucellosis as well, they should be covered with these antibiotics, especially in initial stages. In the present case, streptomycin and doxycycline was given for 1 month followed by rifampicin and doxycycline for 1 month. As the abscess was emptied of it contents completely under operating microscope, antibiotics were given for only 2 months in the post-operative period.
It is important to consider the possibility of intra-medullary abscess as a presentation of neurobrucellosis, especially in endemic region.
Prompt detection and neurosurgical drainage with antibiotics usually results in resolution of the infection.
The duration of antibiotics is variable and depends upon, a. the type of lesion, abscess or granuloma, b. whether surgically drained or not, and c. response to treatment. Steroids should be considered initially, especially if edema is demonstrated on the scan.
Increased awareness and early intervention could prevent the neurological disability and improve the functional outcome.
We acknowledge Dr Anjali Vajramani for her help with manuscript preparation.
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