Clinical Study
Balamuthia mandrillaris encephalitis in a child: case report and literature review

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Abstract

Balamuthia mandrillaris encephalitis is a rare disease with high mortality in the children. Due to the lack of specificity in clinical manifestations, laboratory tests, and neuroimaging, the diagnosis of the disease is difficult, especially the diagnosis of etiology. Currently, the evidence shows that the diagnosis of the disease depends on local brain biopsy or autopsy, and it is difficult to detect the pathogens by traditional etiological detection methods in blood and cerebrospinal fluid. We report a 9-year-old Chinese girl with B. mandrillaris encephalitis who was diagnosed with metagenomic next-generation sequencing (mNGS). The technology of mNGS can provide rapid, early etiological diagnosis without the need for a local brain biopsy, which can buy time for the early treatment of patients. We also provide a comprehensive literature review on this disease.

Introduction

Balamuthia mandrillaris amoeba encephalitis (BAE) is an extremely rare and severe central nervous system (CNS) disease with a mortality rate of 95% (Mittal and Alsinaidi, 2017). A research (Ong et al., 2017) analyzed the causes of death mainly in the following 4 aspects: lack of awareness of the disease, lack of effective detection methods, lack of extensive awareness of public health, and risk factors (especially in developing countries). In addition, its clinical symptoms are similar to those of other CNS infection diseases (such as viral encephalitis, bacterial meningitis, etc.) and lack of specificity. Therefore, early diagnosis and intervention are not possible. A BAE case identified rapidly by metagenomic next-generation sequencing (mNGS) is reported as follows.

Section snippets

Case presentation

A 9-year-old girl was admitted to Jixangxi Provincial Children’s Hospital for sudden-onset consciousness disorder. Half a month before admission, she began to lose her appetite with vomiting 2–3 times a day. Her parents took her to the local hospital, but her condition did not improve. A week later, she began to have fever, which was irregular and had a highest temperature of 39 °C, without any other symptoms such as chills, convulsions, cough, and diarrhea. Three days before admission, she

Metagenomics sequencing

Etiological examination by mNGS in the CSF showed that 16 parasite sequences (Table 1) were found, among which the coverage rate, absolute abundance, and relative abundance of B. mandrillaris species were 33.76%, 68.32%, and 98.76%, respectively (Fig. 2).The absolute abundance of genus was 68.32%, and the relative abundance was 98.76%. The sequence number of genus was 3756, and the sequencing depth was 1 (Fig. 3).

Literature review

We carried out database searches of PUBMED® and domestic Chinese database (CNKI, VIP, and WanFang) for relevant studies conducted during the time period 1993–2019 by using the keywords “Balamuthia mandrillaris encephalitis.” There were no Chinese relevant literature and 188 English relevant literature. Sixty-seven articles were selected for detailed review. A total of 94 patients with BAE were reported, including 52 males and 42 females. The age range was from 4 months to 89 years old, 41 cases

Discussion

Amoeba protozoa can be divided into endamoeba and free-living amoebae according to the living environment. The latter, which live in water and soil, have 6 major genera: Naegleria, Acanthamoeba, Balamuthia mandrillaris, Hartmannella, Vablkampfia, and Sappinia pedata. In 1965, Acanthamoeba was reported to infect human beings and became pathogenic (Fowler and Carter, 1965), which was later confirmed as Naegleria. Up to now, 6 amoebae have been found to infect humans or animals, including

Conflicts of interest

The authors declare that they have no conflicts of interest.

Financial disclosure

The authors have indicated that they have no financial relationships relevant to this study to disclose.

Acknowledgment

We would like to express our gratitude to the patient and her family for their participation in this study. This work was supported by Beijing Hospital Authority “Dengfeng” Talent Training Plan (DFL 20181201) and the National Science and Technology Major Project of China (no. 2018ZX10305409).

References (14)

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