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|Year : 2012 | Volume
| Issue : 2 | Page : 231-234
Multidrug-resistant tuberculous subdural empyema with secondary methicillin-resistant Staphylococcus aureus infection: An unusual presentation of cranial tuberculosis in an infant
Mazda K Turel, Ranjith K Moorthy, Vedantam Rajshekhar
Department of Neurological Sciences, Christian Medical College, Vellore, Tamil Nadu, India
|Date of Submission||22-Jan-2012|
|Date of Decision||22-Jan-2012|
|Date of Acceptance||22-Jan-2012|
|Date of Web Publication||19-May-2012|
Ranjith K Moorthy
Department of Neurological Sciences, Christian Medical College, Vellore - 632 004, Tamil Nadu
Source of Support: None, Conflict of Interest: None
Despite tuberculosis (TB) being endemic in many parts of the world, its prevalence in infancy is low. Neurotuberculosis in this age is even rarer and presents either as meningitis or intracranial tuberculoma on the background of exposure to the disease. We report occurrence of multidrug-resistant tuberculous subdural empyema in a three-month-old girl as the initial presenting manifestation of TB in the absence of any exposure to the disease. She was successfully managed with surgery and drugs with good outcome at 18 months.
Keywords: Infant, methicillin-resistant Staphylococcus aureus, multidrug-resistant tuberculosis, tuberculous subdural empyema
|How to cite this article:|
Turel MK, Moorthy RK, Rajshekhar V. Multidrug-resistant tuberculous subdural empyema with secondary methicillin-resistant Staphylococcus aureus infection: An unusual presentation of cranial tuberculosis in an infant. Neurol India 2012;60:231-4
|How to cite this URL:|
Turel MK, Moorthy RK, Rajshekhar V. Multidrug-resistant tuberculous subdural empyema with secondary methicillin-resistant Staphylococcus aureus infection: An unusual presentation of cranial tuberculosis in an infant. Neurol India [serial online] 2012 [cited 2019 Dec 8];60:231-4. Available from: http://www.neurologyindia.com/text.asp?2012/60/2/231/96412
| » Introduction|| |
Neurotuberculosis in infancy is very rare with tuberculous meningitis being the most common manifestation.  Other infrequent presentations include tuberculous brain abscess and intracranial tuberculomas. Subdural empyema of tuberculous etiology has been reported only twice before in children , and once in an adult.  Management of patients with multidrug-resistant (MDR) TB is difficult and is associated with high morbidity and mortality. We report an unusual case of a MDR tuberculous hemispheric subdural empyema in a three-month-old infant with superadded methicillin-resistant Staphylococcus aureus (MRSA) infection who underwent surgery followed by combined antibiotic and second-line antituberculous therapies resulting in a good outcome at 18 months follow-up.
| » Case Report|| |
A three-month-old girl, full-term delivery with no perinatal complications, was brought with multiple episodes of right upper and lower limb focal-onset motor seizures. She had no fever and was feeding well but had become increasingly irritable for two days. There was no history of or exposure to TB. She had received Bacillus Calmette-Guérin (BCG) vaccination. There was no other relevant history. Her sensorium was normal with no focal neurological deficits and signs of meningeal irritation. The anterior fontanelle was full. Magnetic resonance imaging (MRI) of brain showed a thick left fronto-temporo-parietal multiloculated subdural collection with an enhancing wall, that was hypointense on T1-weighted images, hyperintense on T2-weighted images and showed restriction on diffusion- weighted images [Figure 1]. There were areas of focal cerebritis. The possibility of an acute pyogenic subdural empyema was considered.
|Figure 1: (a-c) Axial (a), Coronal (b), and Sagittal (c) gadolinium-enhanced MRI of a three-month-old child showing a peripherally enhancing multi-loculated subdural empyema with mass effect and midline shift. There were a few areas of focal cerebritis|
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She underwent a left parietal twist drill craniostomy to drain copious amounts of liquid pus, followed by an empirical schedule of intravenous crystalline penicillin, chloramphenicol and metronidazole for two weeks. Vancomycin was added when the cultures grew MRSA. A repeat computed tomography (CT) scan showed increase in the size of the empyema with worsening of edema and mass effect. A left fronto-temporo-parietal craniotomy and evacuation of the subdural empyema was done. At surgery the dura was inflamed and the empyema had a thick subdural membrane with liquid pus that drained under pressure in copious amounts. The pus culture grew MRSA and hence she was treated with a four-week course of intravenous vancomycin, oral rifampicin and oral co-trimoxazole. The histopathology was reported as inflammatory granulation tissue with dense infiltrates of neurophils, histiocytes and lymphocytes. However, there were no well-defined granulomas. At discharge the child was active, alert and healthy with a normal sensorium. At eight weeks, Mycobacterium tuberculosis was isolated in the culture, in addition to MRSA and based on this report the child was treated with isoniazid, rifampicin and pyridoxine. When in-vitro resistance to isoniazid and rifampicin was reported on drug sensitivity testing, pyrazinamide, ciprofloxacin, cyclosporine and ethionamide were added and continued for six months. Isoniazid was continued for the benefit of in-vivo sensitivity  as the child had shown a clinico-radiological improvement. She also developed a small scalp abscess at the six-month follow-up, pus from which again grew MRSA and this was treated with a course of linezolid for four weeks. Chest radiograph and sputum examination of both parents did not reveal any evidence of TB.
At 18 months' follow-up, the child was active, alert and had attained normal developmental milestones. The parents had discontinued anti-tuberculous therapy three months ago (after completing one year of treatment) and hence were advised to extend the course for six months. CT scan showed complete resolution of the empyema and minimal gliosis in the areas of previous focal cerebritis and inflammation [Figure 2].
|Figure 2: (a-d) Axial contrast CT scan of the child at 18 months' follow-up showing complete resolution of the abscess with residual gliosis in the areas of previous focal cerebritis|
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| » Discussion|| |
In the largest reported series of TB in infancy, 11 (23.4%) of the 47 cases had central nervous system (CNS) involvement with the majority of patients being diagnosed with meningitis with or without associated tuberculomas.  The development of tuberculous subdural empyema has been reported only twice before in children, both of whom had prior exposure to M. tuberculosis through a primary infection or contact with another patient. , Our patient was unique in that there was neither past history of systemic TB nor was there any history of exposure to TB. It is known that TB in infants represents a sentinel event within a community suggesting recent transmission most commonly from an infectious adult with pulmonary TB.  In the absence of prior exposure to TB, it is possible that our patient may have had primary tuberculous infection of the palatine tonsil with subsequent hematogeneous dissemination to the subdural space. Vallejo et al., were able to identify an adult source case in 68% infants with the majority (94%) being a household or close family contact emphasizing the importance of performing a thorough epidemiological investigation in children with TB.
Subdural empyemas in children are usually pyogenic in nature with staphylococcus and streptococcus being the most common causative organisms. Sinusitis, otitis, meningitis, and cardiac anomalies are the usual underlying pathologies.  The presence of concomitant pyogenic infection in the presence of TB is known. Van Dellen et al.,  reported a 59-year-old man with tuberculous subdural empyema secondary to osteitis of the cranium who later went on to develop a re-collection eight months after the initial evacuation of pus, and was diagnosed to have an associated staphylococcus infection. Tuberculous infections of the CNS with superadded staphylococcal infection have been reported before but our patient had primary MDR mycobacterium as well as MRSA. [Table 1] summarizes the pediatric cases of tuberculous subdural empyema reported in the English literature. ,
|Table 1: Summary of reported cases of pediatric tuberculous subdural empyema in the literature|
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The global resurgence of TB has been fuelled by several factors including HIV infection, breakdown of TB control programs, overcrowding and MDR TB.  During the last decade, MDR TB has burgeoned in India, resulting in an extremely large number of these cases.  In the previously reported cases of tuberculous subdural empyema, the acid-fast bacilli (AFB) were sensitive to first-line anti-tuberculous therapy. ,, We encountered primary MDR M. tuberculosis, necessitating modification of antituberculous therapy resulting in successful treatment of the infection and good neurological outcome.
This report underscores the importance of submitting pus from any subdural empyema for Mycobacterium tuberculosis cultures as it could be the causative agent. As with our experience and that found in the literature, satisfactory radiological and clinical outcomes can be obtained in these patients with appropriate therapy. Sensitivity reports of M. tuberculosis to first line antituberculous drugs need to be scrupulously followed up, since a change to the next level of antituberculous therapy is warranted in case of documented resistance.
| » References|| |
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|4.||van Dellen A, Nadvi SS, Nathoo N, Ramdial PK. Intracranial tuberculous subdural empyema: Case report. Neurosurgery 1998;43:370-73. |
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[Figure 1], [Figure 2]
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