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 Table of Contents  
ORIGINAL ARTICLE
Year : 2021  |  Volume : 8  |  Issue : 4  |  Page : 150-157

Clinical and imaging manifestations of pediatric melioidosis in Hainan, China


1 Department of Radiology, The Second Affiliated Hospital of Hainan Medical University, Hainan, China
2 Department of Anaesthesiology, Hainan General Hospital, Hainan, China
3 Department of Radiology, Sanya People's Hospital/West China (Sanya) Hospital, Sichuan University, Sanya, China
4 Department of Medical Imaging, The Seventh People's Hospital of Chongqing, Chongqing, China
5 Department of Radiology, Hainan Women and Children's Medical Centre, Sanya, China

Date of Submission03-Sep-2021
Date of Acceptance23-Nov-2021
Date of Web Publication17-Aug-2022

Correspondence Address:
Hong Lu
Department of Medical Imaging, The Seventh People's Hospital of Chongqing, Chongqing
China
Yuefu Zhan
Department of Radiology, Hainan Women and Children's Medical Centre, No. 15 Long Kun Nan Road, Haikou, Hainan 572500
China
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/RID.RID_4_22

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  Abstract 


OBJECTIVE: The objective of this study was to investigate the clinical and imaging manifestations of melioidosis in children in Hainan Province, China, to improve its understanding.
MATERIALS AND METHODS: We retrospectively analyzed 10 children with melioidosis in Hainan Province, China, from January 2002 to November 2021. We collected clinical and imaging data. These data were analyzed retrospectively by two radiologists with more than 10 years' radiology imaging experience.
RESULTS: The patients' average age was 8.4 years (range: 17 days to 15 years), the male-to-female ratio was 6:4, and the average length of hospital stay was 31.5 days (5–96 days). Only two patients had underlying diseases, which were acute lymphocytic leukocyte hepatitis and hepatitis B. One patient had no onset of fever, and the other nine patients had a fever, with an average temperature of 39.5°C (38.4–40.3°C). Two patients had a low white blood cell count (0.27 × 109/L, 3.6 × 109/L), four had a normal white blood cell count, and two had a slightly high white blood cell count (13.6 × 109/L, 14.2 × 109/L). Two patients aged <1 year had a high white blood cell count >34 × 109/L and died. One patient was automatically discharged from the hospital and stopped treatment because of economic factors, and the rest improved after treatment. The neutrophil count was normal in two patients, but it was increased in the other patients. There were no data of C-reactive protein (CRP) measurements in three patients, and four of the other seven patients showed greatly elevated CRP concentrations. Among these four patients, two died and two were cured. One patient had sepsis, three had septicopyemia, and two had multiple organ dysfunction syndrome; among these six patients, one died and others improved. Four patients with pneumonia showed scattered exudation, consolidation, and nodules in both lungs, which developed into lung abscesses, as well as melioidosis and mumps. Three patients showed parotid swelling and abscess formation multilocular. One patient had liver and splenic abscesses. One patient had neck abscesses and one had perineal skin abscesses. One patient had purulent meningitis (clinical diagnosis) and disseminated intravascular coagulation (DIC), and this patient died.
CONCLUSIONS: For patients in endemic areas, and those who have clinical manifestations of sepsis, pneumonia, mumps, and liver and splenic abscesses on imaging manifestations, the possibility of melioidosis should be considered. Microbial culture should be carried out as soon as possible, and these results of culture should be considered. Antibiotic treatment should be performed before a diagnosis. Mortality is more likely in patients who are aged <1 year and have considerably elevated CRP concentrations, multiple organ dysfunction syndrome, and central nervous system infection/DIC.

Keywords: China, clinical, Hainan, imaging, pediatric melioidosis


How to cite this article:
Fan L, Wu Y, Mai S, Lu H, Zhan Y. Clinical and imaging manifestations of pediatric melioidosis in Hainan, China. Radiol Infect Dis 2021;8:150-7

How to cite this URL:
Fan L, Wu Y, Mai S, Lu H, Zhan Y. Clinical and imaging manifestations of pediatric melioidosis in Hainan, China. Radiol Infect Dis [serial online] 2021 [cited 2022 Oct 6];8:150-7. Available from: http://www.ridiseases.org/text.asp?2021/8/4/150/353896

FNx01Long Fan and Yehua Wu contributed equally to this work





  Introduction Top


The most suitable growth environment for Burkholderiapseudomallei is the tropical and subtropical regions between latitude 20°N from south to north, and it is mainly found in Australia and Southeast Asia.[1]B.pseudomallei is widely distributed in the environment and is found in soil and surface water.[2] Epidemiological evidence shows that this species is likely spread by humans.[1],[3],[4] The main transmission routes of melioidosis include inoculation, ingestion, and inhalation.[5],[6] In areas where melioidosis is widespread, it might be spread by simple daily life and activities, especially in rice fields, exposed wounds, and ingestion of contaminated soil or direct drinking of untreated water.

Limmathurotsakul et al.[6] predicted that melioidosis could become a global burden. The data of human and animal melioidosis cases that have been recorded from 1910 to 2014, as well as the presence of areas with suitable environments for melioidosis, support this prediction.[1],[7] Approximately 165,000 cases (95% confidence interval: 68,000–412,000) of human melioidosis and 89,000 (36,000–227,000) deaths worldwide each year have been estimated. The mortality rate of melioidosis is as high as 53.9%. The currently reported cases of melioidosis are much <1% of their predicted number. Some researchers[1],[8] have suggested that by 2030, the major risk factor for infection of melioidosis will be diabetes; in the 25 counties with the highest incidence of melioidosis, the incidence rate of diabetes has increased to varying degrees by 2030,[8] which presents a risk for the spread and prevalence of melioidosis in future.

There have been fewer reports of melioidosis in children than those in adults. In Australia, the central nervous system is more affected in children than in adults, with a rate of 3%–4% in adults and 7%–33% in children.[9],[10],[11] There appear to be fewer reported cases of melioidosis in Asia, with studies in Thailand showing an incidence of 1.5%–3%, and an incidence of 7.5% in Malaysia.[12],[13] Moreover, the clinical manifestations of melioidosis in children differ by region. Among Australian children, primary cutaneous melioidosis (49%), pneumonia (20%), and bacteremia (16%) are relatively rare.[10],[14] In Cambodia and Thailand, children with melioidosis most frequently have skin and soft tissue infections, suppurative mumps, or cervical lymphadenitis,[15] while mumps in child patients in Malaysia account for only 1%. However, there has not been a series of reports on children with melioidosis in China. Therefore, this study retrospectively analyzed the clinical and imaging data of 10 cases of child melioidosis in Hainan Province, China, from 2002 to 2021, to improve the understanding of melioidosis in children.


  Materials and Methods Top


Clinical data

This study was a retrospective analysis of 10 patients with pediatric melioidosis in Hainan Province, China, from 2002 to 2021. The inclusion criteria were as follows: (1) microbial culture for the diagnosis of B.pseudomallei infection, including blood culture, drainage fluid culture, and other secretion cultures; (2) aged ≤16 years;[10] and (3) clinical medical records and imaging data, which were assessed by two doctors with more than 10 years' work experience who reached a consensus.

The collected data included the patients' sex, age, underlying diseases, clinical manifestations, imaging features, treatment, and outcome. The underlying diseases were determined by combining the patient's past medical history and related examinations after hospital admission. The clinical manifestations were obtained from the hospital admission records and thefirst ward round records. The clinical results, based on discharge results of all patients, were divided into death, automatic discharge after stopping treatment, cure, and improvement.


  Results Top


The average age of the 10 patients with pediatric melioidosis was 8.4 years (range: 17 days to 15 years). The male-to-female ratio was 6:4, and the average hospital stay was 31.5 days (5–96 days). Two patients had underlying diseases and eight had no underlying diseases. One patient had no fever, and the remaining nine patients had onset of fever, with an average temperature of 39.5°C (38.4–40.3). A total of 70% of patients had a body temperature of >39. Two patients showed a low white blood cell count, four had a normal white blood cell count, two had a slightly high white blood cell count, and two had a white blood cell count ≥34 × 109/L. The two patients with a white blood cell count ≥34 × 109/L died, and both were younger than 1 year. One patient was discharged automatically because of economic factors and stopped treatment, and the rest showed improvement after treatment. The proportion of neutrophils appeared to have little effect on the clinical outcomes. Among the seven patients who had C-reactive protein (CRP) concentrations measured, four showed greatly elevated concentrations. Of these four patients, two died and two improved. One patient had sepsis, three had septicopyemia, and two developed multiple organ dysfunction syndrome (MODS); among these six patients, one died and others improved. Pneumonia was common in the patients with melioidosis (n = 4), and it manifested as scattered exudation, consolidation, and coagulation in both lungs. Nodules from both lungs developed into lung abscesses [Figure 1] and [Figure 2]. Three patients had melioidosis and mumps, which manifested as parotid gland enlargement and multilocular abscess formation [Figure 3] and [Figure 4]. One patient each had liver and splenic abscesses [Figure 5], neck abscesses, and convulsion perineal abscesses. One patient had purulent meningitis (clinical diagnosis) and disseminated intravascular coagulation (DIC), and this patient died [Table 1], [Table 2], [Table 3].
Figure 1: Computed tomography of the chest in case 1. (a-c) Computed tomographic images taken on July 27, 2021, show diffuse nodular, large, patchy shadows in both lungs, some of which are consolidation lesions, and the lesions in the upper lobe of the right lung form cavities. A small amount of effusion in the right pleural cavity can be seen.

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Figure 2: Computed tomography of the chest in case 2. (a-c) Computed tomographic images taken on November 4, 2014, show that both lungs are scattered with patchy fuzzy shadows and nodules. The right lower lobe shows atelectasis, and there is a small amount of effusion on both sides of the chest. (d-f) Computed tomographic images taken on December 4, 2014, after treatment show that exudative lesions in both lungs have been absorbed, the nodules in both lungs are smaller than previously, and the bilateral pleural effusion has been absorbed.

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Figure 3: Plain and enhanced computed tomography in case 3. (a-c) Plain and enhanced computed tomographic images taken on April 24, 2007, show a diffuse, large left parotid gland, uneven density, multilocular cystic imaging performance, strengthening of the wall, and no enhanced capsule. A ring-shaped enhanced nodule under the skin of the left maxillofacial region can be seen.

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Figure 4: Plain and enhanced computed tomography in case 7. (a-c) Plain and enhanced computed tomographic images on September 6, 2016, show both parotid glands, and there are equal and slightly low-density shadows, which are worse on the left side. A few gaseous shadows are seen subcutaneously. An enhanced scan shows obviously uneven enhancement, with nonenhanced, low-density, necrotic areas, and the lesions involve deep and shallow lobes of the parotid glands on both sides.

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Figure 5: Computed tomography in case 5. (a-c) An enhanced computed tomographic scan shows multiple, round-like, abnormal density lesions in the liver, with no enhancement in the center, and peripheral ring enhancement or separation enhancement. S7 lesions are formed by the fusion of multiple lesions and the spleen is enlarged. In enhanced scanning, the spleen shows multiple, small, circular, ring-shaped enhancement foci with no enhancement in the center.

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Table 1: Basic clinical data of children with melioidosis

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Table 2: Complaints, bacteriology, main diagnoses, and outcomes of children with melioidosis

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Table 3: Imaging manifestations of melioidosis in children

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  Discussion Top


McLeod et al.[10] compared cases of melioidosis in adults and children in northern Australia over 24 years. Children are more likely to develop melioidosis without bacteremia. The ten patients in our study showed no melioidosis, but one had sepsis and three had septicopyemia. Two patients had underlying diseases (acute lymphoblastic leukemia and hepatitis B) and eight had no underlying diseases. This finding is similar to that in McLeod et al.'s study.[10] In adult cases, melioidosis is often accompanied by underlying diseases, the most common being diabetes.[8],[16] The risk factors for melioidosis in children include thalassemia and low immunity.[1] In this study, one patient had acute lymphoblastic leukemia as an underlying disease, and it has been 2 years since he was diagnosed with leukemia (from August 2018 to August 2020), accompanied by immunocompromised.

Previous studies have reported that the mortality rate of children with melioidosis is lower than that of adults.[17] However, McLeod et al.[10] showed no difference in the mortality rate of adult versus child cases of melioidosis. This discrepancy between studies may be related to the reduction in the overall mortality rate of melioidosis in Australia to 12%.[18] This finding indicated that melioidosis can be detected and diagnosed at an early stage, and better medicines and medical care can be provided. In our study, two patients died, and one patient was discharged automatically after stopping treatment. The mortality rate was 20%, which is lower than that in adults with melioidosis[1],[7] but higher than that reported by McLeod et al.[10] In our study, in the two patients who died, one had MODS and the other appeared to have purulent meningitis with DIC, which may have been the leading cause of death. Both of these patients were aged < 1 year, and one was a newborn. A previous study showed that the mortality rate was 72.5% in 22 patients with neonatal melioidosis,[19] which may have been due to the fact that the neonatal immune system was not yet fully developed. Of these 22 newborns, there were 10 cases of transmission, 8 cases were attributed to community infection or medical-related infection, 1 case was due to breastfeeding transmission, and the other case was vertical transmission caused by placental microabscesses.

In our study, the most common infection site of melioidosis in children was the lungs(4 cases) and parotid gland (3 cases). This is in contrast with no cases of mumps reported in Australia[10] but is similar to other Australia reports of the incidence of pneumonia (20%).[10],[14] Our finding is also similar to the rate of 30%–40% of mumps in children with melioidosis in Southeast Asia,[20],[21] but we found a higher incidence of liver abscesses than that in reports from Thailand.[22] This regional difference in the incidence of melioidosis may be due to bias owing to the small sample size. This difference may also be due to different modes of transmission, such as inhalation and ingestion of unchlorinated domestic water or other contaminated water sources.[23]

In this study, the white blood cell count of the two patients who died was ≥34 × 109/L, and the CRP concentration was high (164.2 and 295 mg/L), which suggested that the patients had a severe inflammatory response. Domthong et al.[24] and other researchers showed that lung performance, white blood cell count, and blood urea nitrogen concentrations were positively correlated with the mortality rate of melioidosis. This previous finding of the effect of melioidosis on white blood cell count is consistent with that in our study. Cheng et al.[25] suggested that the CRP concentration in the hospital is not a sensitive marker for melioidosis. The finding of a normal CRP concentration cannot exclude acute, chronic, or recurrent melioidosis. In multivariate analysis, sepsis and bacteremia are associated with mortality. In this study, three patients had no CRP measurements, but in the other seven patients, CPR concentrations in four patients were greatly elevated (164.2–295.72 mg/L). Of these four patients, two died, which suggested that CRP concentrations may be related to death in patients with melioidosis.

The imaging manifestations of children with pneumonia due to B.pseudomallei are similar to those of adults. Common manifestations are scattered exudation, consolidation, nodules in both lungs, and rapid progression to cavities.[26] Other symptoms, such as lymphadenitis (3%), pleural effusion (5%–15%), and pericardial involvement (1%), have also been reported.[27] Some rare manifestations, such as mediastinal abscess imitating malignant tumors, have been reported.[28] The imaging manifestations of pneumonia in children in this study are consistent with those reported in the literature. In this study, case 6 showed pneumonia accompanied by infection of the anterior mediastinum and thymus. The presence of mumps in our patients with melioidosis is consistent with previous reports,[29] which manifests as a small multilocular abscess with separation, and enhancement of the separation can be seen. The finding of one case of liver and splenic abscesses with scattered multiple small abscesses in this study is consistent with previous reports.[30] Among them, necklace sign is the strongest predictor of a Melioidosis hepatosplenic abscess;[30] Cervical and perineal abscesses of melioidosis are nonspecific in imaging. In this study, one patient had purulent meningitis (clinical diagnosis) with DIC. This patient died, and an imaging evaluation could not be performed because of incomplete imaging data. The imaging manifestations of melioidosis of the central nervous system are multiple small abscesses, peripheral edema, and subdural empyema in the brain. These imaging manifestations are mainly single or multiple nodules in the brain, as well as ring-shaped and flaky enhancement. This disease progresses rapidly, venous and sinus thrombosis is rare,[31] and it rarely manifests as meningitis.


  Conclusions Top


Melioidosis in children is still rare, even in endemic areas. Clinicians need to fully understand the clinical and imaging manifestations of melioidosis in children. We retrospectively analyzed ten children with melioidosis with clinical and radiological data in Hainan Province, China. We found that living in endemic areas or having a history of living in endemic areas, clinical onset of a high fever, age <1 year, a high white blood cell count, elevated CRP concentrations, MODS, and central nervous system infection/DIC result in a higher risk of developing melioidosis. Neonatal melioidosis or children aged ≤1 year may be a particularly high-risk group. Imaging manifestations of pneumonia, mumps, liver and splenic abscesses, and soft tissue abscesses should be considered. The possibility of melioid infection should be considered, and microbial culture should be carried out in a timely manner. Timely diagnosis and treatment affect the outcome of melioidosis. Sensitive antibiotics should be administered in advance in accordance with guidelines before waiting for a definite diagnosis of melioidosis.

Acknowledgments

We thank Ellen Knapp, PhD, from Liwen Bianji (Edanz) (www.liwenbianji.cn/), for editing the English text of a draft of this manuscript.

Financial support and sponsorship

This research was funded by the Hainan Provincial Health Commission Science and Technology Project (18A200008) and Hainan Province Clinical Medical Center.

Conflicts of interest

There are no conflicts of interest.



 
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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]
 
 
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