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Central nervous system melioidosis: A systematic review of individual participant data of case reports and case
Monton Wongwandee1, Patcharasarn Linasmita1
1Department of Medicine, Faculty of Medicine, Srinakharinwirot University, Nakhon Nayok, Thailand.
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.
Insights
Central nervous system (CNS) melioidosis is rare but fatal if untreated. This review highlights key symptoms like fever and cranial nerve deficits, emphasizing early diagnosis in endemic areas to improve outcomes for this serious infection.
Area of Science:
- Infectious Diseases
- Neurology
- Microbiology
Background:
- Central nervous system (CNS) melioidosis is a rare but potentially fatal infection.
- Delayed diagnosis and treatment significantly increase mortality risk.
- Understanding CNS melioidosis is crucial for timely intervention.
Purpose of the Study:
- To systematically review case reports and series of CNS melioidosis.
- To identify and consolidate existing knowledge on the epidemiology, clinical presentation, and outcomes of CNS melioidosis.
- To inform diagnostic and treatment strategies for this rare condition.
Main Methods:
- Systematic literature search of PubMed, Web of Science, Thai-Journal Citation Index, and Google Scholar.
- Inclusion of studies reporting confirmed CNS melioidosis cases (culture, serology, PCR) with available clinical data.
- Analysis of 70 studies encompassing 120 patients, with data collected up to July 10, 2018.
Main Results:
- The majority of patients (93%) were from endemic areas, with a median age of 40; 70% were male and 60% had risk factors.
- Common symptoms included fever (82%), headache (54%), and cranial nerve deficits (52%).
- Mortality rate was 20%, with brainstem and frontal lobe most affected; characteristic CSF showed mononuclear pleocytosis.
Conclusions:
- CNS melioidosis requires prompt recognition, particularly in endemic regions with risk factors.
- Brainstem involvement and cranial nerve deficits are characteristic presentations, especially in encephalomyelitis.
- Fever, neurological deficits, and specific CSF findings should prompt consideration of CNS melioidosis in differential diagnoses.
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