Case Reports / Journal of Clinical Neuroscience 14 (2007) 1213–1215 19. Odake G, Tenjin H, Murakami N. Cyst of the choroid plexus in the lateral ventricle: case report and review of the literature. Neurosurgery 1990;27:470–6. 20. Wong CW, Ko SF, Wai YY. Arachnoid cyst of the lateral ventricle manifesting positional psychosis. Neurosurgery 1993;32:841–3. 21. Lee KS, Bae HG, Yun IG. Intraventricular arachnoid cyst. J Neurosurg 1989;70:154–5. 22. Okamura K, Watanabe M, Inoue N, et al. Intraventricular arachnoid cyst – on the origin of intraventricular arachnoid cysts. No To Shinkei 1996;48:1015–21 (Abstract).
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23. Yeates A, Enzmann D. An intraventricular arachnoid cyst. J Comput Assist Tomogr 1979;3:697–700. 24. Hirano A, Hirano M. Benign cysts in the central nervous system: neuropathological observations of the cyst walls. Neuropathology 2004;24:1–7. 25. Inoue T, Matsushima T, Fukui M, et al. Immunohistochemical study of intracranial cysts. Neurosurgery 1988;23:576–81. 26. Tubbs RS, Smyth MD, Wellons 3rd JC, et al. Alternative uses for the subgaleal shunt in pediatric neurosurgery. Pediatr Neurosurg 2003;39: 22–4.
doi:10.1016/j.jocn.2006.04.016
Spectra of central nervous system melioidosis Kalai Arasu Muthusamy
a,b,*
, Vicknes Waran b, Savithri D. Puthucheary
c
a
b
University Laboratory of Physiology, Oxford University, Parks Road, Oxford OX1 3PT, United Kingdom Division of Neurosurgery, Department of Surgery, University of Malaya Medical Centre, Kuala Lumpur, Malaysia c Department of Medical Microbiology, University of Malaya Medical Centre, Kuala Lumpur, Malaysia Received 2 February 2006; accepted 14 March 2006
Abstract Burkholderia pseudomallei infection of the central nervous system (CNS) is rare with less than 50 cases reported over the last 30 years. The retrospective melioidosis study at University Malaya Medical Centre has documented three cases of CNS melioidosis out of more than 160 cases of melioidosis since 1978. There were two patients with brain abscess and one with spinal epidural abscess. The predisposing factors were: one patient was an aboriginal farmer and the other two were diabetic. Their age ranged from 17 to 45 years. Prominent neurological features were limb weakness, cranial nerve palsy (6th and 7th) and visual disturbance. CT brain scan and MRI spine showed abscess formation, subdural collection, and spinal epidural collection, osteomyelitis of vertebra and occipital bone and also sagital sinus thrombosis. All these patients underwent surgical drainage leading to bacteriological diagnosis as well as appropriate long-term antibiotic therapy. All had good recovery at 6 months after completion of treatment. 2006 Elsevier Ltd. All rights reserved. Keywords: Meliodosis; Abscess; Brain; Spinal epidural
1. Introduction
2. Case reports
Melioidosis of the central nervous system (CNS) is a rare disease. It is caused by the Gram-negative bacillus Burkholderia pseudomallei. There have been less than 50 cases of melioidosis of the CNS reported over the last 30 years. At the University Malaya Medical Centre we have documented three cases of CNS melioidosis. These were: one patient with a spinal epidural abscess; one with a brain abscess; and one with skull osteomyelitis with subgaleal collection and subdural empyema and cerebral abscess. All cases had B. pseudomallei identified in the pus.
2.1. Case 1
*
Corresponding author. Tel.: +44 7909595757. E-mail address:
[email protected] (K.A. Muthusamy).
An 18-year-old native farmer presented with fever, headache and neck stiffness of 1-week duration. He was previously well and had no risk factors including diabetes, renal failure or excessive alcohol consumption. On examination he had signs and symptoms of meningitis with drowsiness and right hemiperesis, seizures and VI and VII cranial nerve palsy. Blood investigation showed leucocytosis and raised erythrocyte sedimentation rate (ESR). His chest X-ray was normal. Blood culture showed no growth. CT brain scan reveal a left occipital abscess and surrounding cerebral edema. He underwent burr hole and drainage of the abscess. A culture sample grew B. pseudomallei. The patients was lost to follow-up.
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Case Reports / Journal of Clinical Neuroscience 14 (2007) 1213–1215
2.2. Case 2 A 45-year-old doctor presented with backache of 2 weeks duration with acute onset of lower limb weakness and urinary retention. He was diabetic. He had no history of recent travel. Blood investigation showed leucocytosis and raised ESR. Blood culture had no growth. His chest X-ray was normal. MRI spine showed heterogenous signal of the T8 vertebra body and extradural collection from T3 to T10 with osteomyelitis of T8 with epidural abscess (Fig. 1a and b). He had a laminectomy and drainage of the abscess. The culture sample grew B. pseudomallei. He was placed on long-term antibiotic treatment and recovered from his lower limb weakness. 2.3. Case 3 A 33-year-old man presented with fever of 3 weeks duration, seizures, altered conscious level and neck stiffness, with signs and symptoms of meningitis. He had no risk factors for melioidosis. He had a small ulcer over his occipital region. The swab culture of the ulcer had no growth. Blood investigation showed leucocytosis, raised ESR and blood culture grew B. pseudomallei. CT brain scan showed a subgaleal collection with osteomylitis of the occipital bone and occipital abscess (Fig. 2). His chest X-ray was normal. The MRI brain showed a welldefined rounded lesion with ring enhancement in the
Fig. 2. (Case 3): A hypodense area is seen in the occipital lobe. There is a small subdural collection (arrow) with rim enhancement on the leftside of the posterior flax cerebri suggesting empyema. The delta sign is seen in the superior sagittal sinus indicating thrombosis.
occipital region with a parafalcine subdural collection and osteomyelitis of the occipital bone. He had a craniectomy and drainage of the abscess. A culture sample grew B. pseudomallei. He was placed on long-term antibiotic treatment. He is well 1-year post-treatment. 3. Discussion
Fig. 1. (a) (Case 2): MRI spine reveals heterogenous signal of the T8 vertebral body with a posterior extradural collection (arrow) extending from T3–T10 on T1-weighted images. (b) (Case 2): The extradural collection (arrow) demonstrates high intensity heterogenous signal on T2weighted images. There was minimal enhancement with contrast.
Melioidosis of the CNS is rare. However, with increasing worldwide travel, this diseases which is predominantly in the tropics, may be exported to temperate countries. Human melioidosis was first reported by Whitmore and Krishnaswami1 in Burma in 1912. Melioidosis is caused by the Gram-negative bacillus soil saprophyte B. pseudomallei previously known as Pseudomonas pseudomallei. The disease is endemic in Southeast Asia, India and Northern Australia.2 The presentation ranges from localized abscess, to severe pneumonia, leading to septicemia and death.2 In a review of 252 cases of melioidosis over 10 years in northern Australia by Currie et al.,3 the authors showed that 85% of cases presented during the rainy season and the predisposing factors included diabetes, excessive alcohol consumption, chronic lung disease, chronic renal failure disease, kava consumption, malignancy, steroid therapy and rheumatic heart disease. There were 20% of patients with no risk factors. The association with diabetes mellitus is particularly strong and increases the risk of infection by up to 100-fold.4
Case Reports / Journal of Clinical Neuroscience 14 (2007) 1215–1219
We have documented three patients in our centre: case 1 had a brain abscess; case 2 had a spinal epidural abscess; and case 3 had a spectrum with subcutaneous (subgaleal) and deep infection (brain abscesses). All the patients presented with neurological signs and symptoms such as seizure in two patients, decreased consciousness level in two patients, lower limb weakness in two and cranial nerve palsy in one. All patients had raised ESR and leucocytosis. Two patients had blood cultures positive for B. pseudomallei. All the patients had drainage of abscess and antimicrobial therapy. Pus cultures demonstrated B. pseudomallei in all three patients. One patient had diabetes which is a risk factor for melioidosis and the other was a farmer who worked with soil. None had a history of recent travel and they presented at the same time of the year. Two patients were treated with intravenous (IV) ceftazidime 100–200 mg/kg/day in three divided doses together with cotrimoxazole 40–60 mg/kg/day in two divided doses for 6 weeks. One patient with a brain abscess was treated with IV chloramphenicol 500 mg 4-hourly together with cotrimoxazole 40–60 mg/kg/day in two divided doses for 6 weeks. To date, there have been few cases of melioidotic brain abscess reported in the literature.5,6 The prospective melioidosis study7 at the Royal Darwin Hospital has documented 12 cases (5%) of CNS melioidosis over 9 years out of a total of 232 cases of melioidosis. Features on presentation were unilateral limb weakness (6), cerebellar signs (2), mixed cerebellar and brainstem features with peripheral weakness (2) and flaccid paraparesis (2). The MRI of the two patients with recent onset flaccid paraparesis showed increased signal uptake in the spinal cord extending down to the conus. The pathogenesis was postulated to be due to direct bacterial invasion of the CNS.
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Melioidosis is difficult to diagnosis on clinical signs alone and positive culture is required from an appropriate specimen. In our experience, CNS melioidosis will require at least 6–12 weeks of parenteral antibiotics, and oral maintenance therapy for 3–6 months to ensure complete eradication of the infection. There are no controlled studies regarding the duration of therapy. Life-long follow-up and counseling is an absolute requirement in the prevention of relapse and recurrence.4 Follow-up is by clinical assessment and using ESR and C-reactive protein (CRP) levels. 4. Conclusions Central nervous system meliodosis presents as a simple brain abscess, spinal epidural abscess or subgaleal abscess penetrating via osteomylitis and forming brain abscess. A high index of suspicion and bacteriological examination is needed for diagnosis. Long-term antibiotic therapy is needed to ensure complete eradication of the infection. References 1. Leelarasamee A. Melioidosis in Southeast Asia. Acta Trop 2000;74:129–32. 2. Dance DAB. Melioidosis as an emerging global problem. Acta Trop 2002;74:115–9. 3. Currie BJ, Fisher DA, Howard DM, et al. Endemic melioidosis in tropical northern Australia: a 10-year prospective study and review of literature. Clin Infect Dis 2000;31:981–6. 4. Puthucheary SD, Vadivelu J. Principles of Management in Human Melioidosis. Singapore: Singapore University Press; 2002, pp. 56–63. 5. Lee MK, Chua CT. Brain abscess due to Pseudomonas pseudomallei. Aust NZ J Med 1986;16:75–7. 6. Lath R, Rajshekhar V, George V. Brain abscess as the presenting feature of melioidosis. Br J Neurosurg 1998;12:170–2. 7. Currie BJ, Fisher DA, Howard DM, et al. Neurological melioidosis. Act Trop 2000;74:145–51.
doi:10.1016/j.jocn.2006.03.022
Rapid regrowth of a successfully coiled traumatic pericallosal aneurysm Chun-Man Yuen a, Yeh-Lin Kuo b, Jih-Tsun Ho a, Jui-Jung Liao a
b
a,*
Department of Neurosurgery, Chang Gung Memorial Hospital-Kaohsiung Medical Center, Chang Gung University College of Medicine, Kaohsiung, Taiwan Hospital, 123, Ta-pei Road, Niaosung Hsiang, Kaohsiung Hsien, Taiwan Department of Radiology, Chang Gung Memorial Hospital-Kaohsiung Medical Center, Chang Gung University College of Medicine, Kaohsiung, Taiwan Received 11 February 2006; accepted 16 April 2006
*
Corresponding author. Tel.: +886 7 7317123; fax: +866 7 7318762. E-mail address:
[email protected] (J.-J. Liao).
