Central nervous system melioidosis in the pediatric age group: review

Prasad, G. Lakshmi; Kini, Pushpa; Divya, S

doi:10.1007/s00381-017-3422-1

Cited by 5 publications

(6 citation statements)

References 34 publications

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“…Only 1.5 to 5 percent of the melioidosis cases have been reported to have neurological involvement [1,2]. Although there is a systematic review conducted in pediatric patients [3], most CNS melioidosis cases occur in adults. Therefore, the knowledge of the CNS melioidosis including epidemiology, clinical manifestations, laboratory findings, treatment, and outcome are still limited and solely depend on data of case reports.…”

Section: Introductionmentioning

confidence: 99%

Central nervous system melioidosis: A systematic review of individual participant data of case reports and case series

Wongwandee

Linasmita

2019

PLoS Negl Trop Dis

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Background Central nervous system (CNS) melioidosis is rare. However, delayed diagnosis and treatment could lead to fatality. To identify knowledge of CNS melioidosis, we systematically review case reports and case series. Methodology/Principal findings We searched through PubMed, Web of Science and Thai-Journal Citation Index databases as well as Google Scholar with the last date on July 10, 2018. The diagnosis of CNS melioidosis had to be confirmed with culture, serology or polymerase chain reaction. We excluded the animal cases and the studies that the clinical data were not available. We identified 1170 relevant studies, while 70 studies with a total of 120 patients were analyzed. Ninety-three percent of patients were reported from the endemic area of melioidosis. Median age was 40 years (IQR 18–53), and 70% were men. A total of 60% had one or more risk factors for melioidosis. The median duration from clinical onset to diagnosis was ten days (IQR 5–25). Fever (82%), headache (54%), unilateral weakness (57%) and cranial nerve deficits (52%) are among the prominent presentation. Most patient (67%) had at least one extraneurological organ involvement. The CSF profile mostly showed mononuclear pleocytosis (64%), high protein (93%) and normal glucose (66%). The rim-enhancing pattern (78%) is the most frequent neuroimaging finding in encephalomyelitis and brain abscess patients. Both brainstem (34%) and frontal lobe (34%) are the most affected locations. Mortality rate was 20%. Conclusions/Significance This study is the most extensive systematic review of case reports and case series of CNS melioidosis in all age groups. However, the results should be cautiously interpreted due to the missing data issue. The propensity of brainstem involvement which correlates with prominent cranial nerve deficits is the characteristic of CNS melioidosis especially encephalomyelitis type. The presenting features of fever and neurological deficits (especially cranial nerve palsies) along with the mononuclear CSF pleocytosis in a patient who lives in the endemic area and also has the risk factor for melioidosis should raise the CNS melioidosis as the differential diagnosis.

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Section: Introductionmentioning

confidence: 99%

Central nervous system melioidosis: A systematic review of individual participant data of case reports and case series

Wongwandee

Linasmita

2019

PLoS Negl Trop Dis

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“…The adoptive transfer of B. pseudomallei -loaded CD11b cells via the tail vein resulted in abscesses in the brain that were predominately localized in the regions surrounding the cerebral superior sagittal sinus 10 . In children with melioidosis who develop meningoencephalitis, brain abscesses are more often localized in the parietal lobe than in other regions 2 . In this study, the endotoxemic mice often exhibited abscesses localized in the dural venous sinus of the parietal lobe.…”

Section: Discussionmentioning

confidence: 99%

“…Melioidosis is a fatally infectious disease caused by Burkholderia pseudomallei infection that usually occurs in endemic areas, such as northern Australia and Southeast Asia. The clinical spectrum of melioidosis varies and can present as localized cutaneous suppuration, multiple organ abscesses and neurological disorders 1 , 2 . The severity of melioidosis depends on the invasive route, the size of inoculation, the virulent characteristics of the bacterium and host defense factors 1 , 3 – 5 .…”

Section: Introductionmentioning

confidence: 99%

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Burkholderia pseudomallei-loaded cells act as a Trojan horse to invade the brain during endotoxemia

Hsueh¹,

Lin²,

Liu³

et al. 2018

Sci Rep

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Neurologic melioidosis occurs in both human and animals; however, the mechanism by which the pathogen Burkholderia pseudomallei invades the central nervous system (CNS) remains unclear. B. pseudomallei-loaded Ly6C cells have been suggested as a putative portal; however, during melioidosis, lipopolysaccharide (LPS) can drive disruption of the blood-brain barrier (BBB). This study aims to test whether the Trojan horse-like mechanism occurs during endotoxemia. The expression levels of cerebral cytokines, chemokines and cell adhesion molecules; the activation of astrocytes, microglia and endothelial cells; and the increased vascular permeability and brain-infiltrating leukocytes were evaluated using B. pseudomallei, B. thailandensis, B. cenocepacia and B. multivorans LPS-induced brains. Accordingly, different degrees of BBB damage in those brains with endotoxemia were established. The B. multivorans LPS-induced brain exhibited the highest levels of disruptive BBB according to the above mediators/indicators. Into these distinct groups of endotoxemic mice, B. pseudomallei-loaded Ly6C cells or free B. pseudomallei were adoptively transferred at equal bacterial concentrations (103 CFU). The bacterial load and number of cases of meningeal neutrophil infiltration in the brains of animals treated with B. pseudomallei-loaded Ly6C cells were higher than those in brains induced by free B. pseudomallei in any of the endotoxemic groups. In particular, these results were reproducible in B. multivorans LPS-induced brains. We suggest that B. pseudomallei-loaded cells can act as a Trojan horse and are more effective than free B. pseudomallei in invading the CNS under septic or endotoxemic conditions even when there is a high degree of BBB disruption.

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“…Pediatric neuro melioidosis is very rare and meningoencephalitis is the most common presentation. Cranial nerve palsies and fever are prominent presenting features,[ 7 ] the commonest cranial nerve affected being the facial nerve. [ 8 ] Our patient initially presented as unilateral facial palsy along with low-grade fever.…”

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confidence: 99%