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Clinical and Imaging Findings of Musculoskeletal Melioidosis in the Right Hip: A Case Report

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J Korean Soc Radiol 2018;78(3):212-219 https://doi.org/10.3348/jksr.2018.78.3.212

INTRODUCTION

Melioidosis is an infectious disease caused by Burkholderia pseudomallei. The disease is endemic in tropical regions, partic- ularly in Southern Asia and Northern Australia, but is rare in Korea. Until 2014, only 11 melioidosis patients were reported by the Center for Disease Control and Prevention (CDC) in Korea and all patients had a history of travel to Southeast Asia (1).

Melioidosis can affect any organ and cause various clinical man- ifestations. The imaging findings of musculoskeletal melioidosis manifest as osteomyelitis or septic arthritis with soft tissue ab- scess or can mimic bone tumor (2, 3). The current study de- scribes the clinical and imaging findings of melioidosis involv- ing the right femur and hip joint in a 64-year-old man.

Case RepORT

A 64-year-old male presented with complaint of right hip and inguinal pain. He reported a history of diabetes mellitus of fifteen years duration, as well as a history of esophageal cancer. The esophageal cancer was in complete remission after radiochemo- therapy about eight years ago. Until recently, he had resided in Cambodia for five years.

About one month ago, the patient was admitted to the hospi- tal with pneumonia and fever as the only presenting symptom.

Pneumonia was diagnosed on chest X-ray and computed to- mography was performed to identify the cause of the fever.

About three weeks after hospital discharge, he complained of right hip and inguinal pain that had suddenly and markedly in- creased in the absence of any history of identifiable trauma or

Clinical and Imaging Findings of Musculoskeletal Melioidosis in the Right Hip: A Case Report

우측 대퇴부에서 발생한 근골격계 유비저의 임상적, 영상학적 소견: 증례 보고

Myung Hyun Kim, MD, Tong Jin Chun, MD*

Department of Radiology, Eulji University Hospital, Daejeon, Korea

Melioidosis is an infectious disease caused by a Gram-negative bacterium thought to be caused Burkholderia pseudomallei. This disease is endemic in tropical regions, but is not endemic and is rarely encountered in Korea. Nevertheless, the importance of early diagnosis of melioidosis is drawing substantial attention, due to its prolifer- ation and the high mortality caused by the disease. Melioidosis can attack any or- gan, and manifests with a variety of symptoms. In particular, musculoskeletal meli- oidosis is rare and presents with nonspecific musculoskeletal symptoms. The imaging features of musculoskeletal melioidosis manifest a form of osteomyelitis or septic arthritis with soft tissue abscess or can mimic a bone tumor. This study de- scribes the clinical and imaging findings of melioidosis involving the right femur and hip joint in a 64-year-old man.

Index terms Melioidosis

Burkholderia Pseudomallei Osteomyelitis

Femur Infection

Received April 11, 2017 Revised September 4, 2017 Accepted September 29, 2017

*Corresponding author: Tong Jin Chun, MD Department of Radiology, Eulji University Hospital, 95 Dunsanseo-ro, Seo-gu, Daejeon 35233, Korea.

Tel. 82-42-611-3564 Fax. 82-42-611-3590 E-mail: tchun@eulji.ac.kr

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0) which permits unrestricted non-commercial use, distri- bution, and reproduction in any medium, provided the original work is properly cited.

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Fig. 1. A 64-year-old melioidosis patient with right hip pain.

A. Initial and 3 weeks follow-up radiography images including the right hip show no abnormal findings on soft tissue or bony structure.

B. Whole body bone scan shows multifocal increased uptake in the greater and lesser trochanter of the right femur, and weak uptake in the fem- oral head.

B A

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arduous physical labor. He reported that he had experienced pain for several months, but assumed it was “osteoarthritis caused by aging” because it was a nominal pain similar to that in his other joints. On physical examination he had mild tender- ness on the anterior thigh just below the inguinal region. The range of motion in his right hip was normal, but the log rolling test was positive, suggesting inflammation on the right femoral

head or acetabulum.

On initial radiographic examination of the right hip (Fig. 1A), no abnormal findings in the soft tissue and bony structures were appreciated; the study revealed evidence of mild degenerative changes only, which was also observed three weeks later on fol- low-up radiography. However, the pain had progressively in- creased and became more intense with time. After only one

Fig. 1. A 64-year-old melioidosis patient with right hip pain.

C. MRI shows abnormal lesion on the right proximal femur. Fat-suppressed T2-weighted MRI shows heterogeneously high signal intensity and T1-weighted MRI and proton density-weighted MRI show heterogeneously low signal intensity in the inferior portion of the femoral head, neck, greater and lesser trochanter, and the proximal shaft of the femur. Some areas that showed high signal intensity on T2-weighted MRI did not show enhancement. These non-enhanced areas are thought to consist of necrotic tissues. Contrast enhancement is observed not only in bone but also in surrounding soft tissue and iliopsoas bursa.

MRI = magnetic resonance imaging C

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week, the patient complained that he was unable to walk due to severe pain in the right hip and inguinal area.

Further evaluation was felt to be necessary, and a whole body bone scan and contrast enhanced magnetic resonance imaging (MRI), were performed. The whole body bone scan (Fig. 1B) revealed multifocal increased uptake in the greater and lesser trochanter of the right femur, and weak uptake in the femoral head. On MRI (Fig. 1C, D), the fat-suppressed T2-weighted MRI showed heterogeneously high signal intensity and T1- weighted MRI and proton density-weighted MRI showed het- erogeneously low signal intensity in the inferior portion of the

femoral head and neck, greater and lesser trochanter, and prox- imal shaft of the femur, with contrast enhancement observed in some areas at the same sites.

In other words, there were some areas that showed high sig- nal intensity on T2-weighted MRI but no enhancement.

These intraosseous non-enhanced areas were thought to con- sist of necrotic tissue. Contrast enhancement was evident not only in the femur, but also in the surrounding soft tissue. In the right hip joint, prominent synovial thickening and enhance- ment with increased joint effusion were found. A distended il- iopsoas bursa (with bursal wall thickening and increased effu-

Fig. 1. A 64-year-old melioidosis patient with right hip pain.

D. Magnetic resonance image shows prominent synovial thickening and enhancement with increased joint effusion in the right hip joint.

D

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sion) suggested an iliopsoas bursitis or secondary change of the inflammation of the femur and hip joint. These MRI find- ings were thought to be consistent with acute osteomyelitis with

bone necrosis.

An orthopedic doctor at our hospital evaluated the lesion un- der the impression of disease, not only inferred disease from the

Fig. 1. A 64-year-old melioidosis patient with right hip pain.

E. Positron emission tomography/computed tomography shows multifocal hypermetabolic lesion on the proximal shaft of the femur, femur neck, femoral head and iliopsoas bursa.

F. Post-excisional biopsy radiography (left) shows fracture of the lesser trochanter of the right femur and an irregular shaped radiolucent area on the right proximal shaft of the femur. This is thought to be due to the excisional biopsy. Post-re-operative radiography (right) shows widened ra- diolucent area on the proximal shaft of the femur where the drainage catheter tip is located. This indicates that an abscess is formed in this ra- diolucent lesion.

F E

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imaging findings but also metastatic bone lesion, because the patient had a history of esophageal cancer. As the next step of evaluation, positron emission tomography/computed tomogra- phy (PET/CT) was performed to rule out malignancy. On PET/

CT (Fig. 1E) a multifocal, hypermetabolic lesion was identified on the proximal shaft of the femur, femur neck, femoral head and iliopsoas bursa.

About one month later, an excisional biopsy of the right fe- mur lesion was performed (Fig. 1F, right). The orthopaedic sur- geon at our hospital ordered the biopsy because it was thought that the probability of metastasis was most likely. Subsequent pathology confirmed the right femoral lesion as a condition in- volving chronic inflammation with necrosis and degenerative bony trabeculae of woven bone without osteoblastic rimming.

The results also confirmed the soft tissue surrounding the fem- oral head as a suppurative inflammation with granulation tissue and fat necrosis. Other evaluations such as tissue culture were not performed. About one month later (and after the excisional biopsy), surgical re-intervention was performed due to some oozing at the operation site (Fig. 1F, Left). A trochanteric ab- scess was found on the surgical bed, therefore, incision, drainage and a biopsy of the synovium of the hip were performed. The acute suppurative inflammation in the synovium of the hip was pathologically confirmed. A culture of the abscess contents re- vealed Burkholderia pseudomallei. This Gram-negative bacillus was previously detected in blood cultures to either rule in, or exclude, the presence of pneumonia.

After the initial diagnosis of pneumonia, the patient was treat- ed with ceftriaxone (a type of β-lactam antibiotic and clarithro- mycin (a type of macrolide), as the first line of defenset with re- gard to treatment for community-acquired pneumonia. After the culture test results were obtained, the patient was treated with piperacillin/tazobactam, another β-lactam antibiotic, and levofloxacin, a fluoroquinolone, for about three weeks (as a guideline of severe pneumonia caused by Gram-negative bacil- li). After culturing of Burkholderia pseudomallei in the right hip abscess, the same antibiotics therapies were planned, because the same type of antibiotic is used for melioidosis regardless of the location or type of infections.

However, the patient was transferred to another hospital clos- er to his residential area.

DIsCUssION

Melioidosis is a disease endemic to tropical parts of the world in general, and Southeast Asia and Northern Australia in partic- ular. It is thought that the etiology of melioidosis is most likely.

Burkholderia pseudomallei (4). The first patient diagnosed with melioidosis resided in Myahjar, and the case was reported in 1912 In Korea, the first documented melioidosis patient was re- ported in 2003 (5).

This disease is regarded, and managed, as a contagious com- municable disease in Korea, due to its increasing incidence with increased international travel and a high mortality rate, of about 80%, without proper early medical intervention and treatment (6).

To date, a total of eleven melioidosis patients have been re- ported in Korea. These patients were all male, with a mean age of 52.3 years (range, 32–66 years) and had all visited Southeast Asia. The period of symptom development was variable, rang- ing from one to three days (1). Underlying diseases, including di- abetes mellitus, chronic lung disease, renal disease, malignancy, immunosuppressive treatment and heavy alcohol consumption and/or or chronologic age over 50 years, are well-known risk fac- tors. These risk factors are associated not only with susceptibili- ty to melioidosis, but also with the relative severity of the dis- ease (1, 7).

The patient in this report had much in common with with other male Korean patients. The patient was 63-years-old man, and with documented histories of residence in Southeast Asia, diagnosed diabetes mellitus and cancer treatment.

Melioidosis can affect any organ and cause a variety of differ- ent symptoms. Pulmonary involvement, such as pneumonia, is the most common clinical feature and disseminated abscess formation is a characteristic feature of melioidosis (1). Accord- ing to Cheng and Currie (4), musculoskeletal melioidosis is dif- ficult to differentiate from other infectious musculoskeletal dis- eases based on clinical symptoms, but the systemic features are prominent.

Musculoskeletal melioidosis has a relatively low incidence.

Currie et al. (8) reported that only 4% of melioidosis patients presented osteomyelitis or septic arthritis in Northern Austra- lia, over a period of ten years. Another study (9), carried out in Northern Australia, reported a 7.6% incidence of bone and joint infections in melioidosis patients.

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There are two possible infection routes for musculoskeletal melioidosis. One is directly from skin injury or wound infection.

The other is hematogenously from another primary disease, such as pneumonia with a form of septicemia (9). The patient in this case report can be considered to belong to the latter group.

In this report, we describe a patient with melioidosis that in- volved the right femur and hip joint, with recent underlying pneumonia. Burkholderia pseudomallei were cultured from a trochanteric abscess on the right hip joint and previous blood drawn during pneumonia, suggesting hematogenous spread from the lung, presenting as pneumonia. The patient also had a history of diabetes mellitus, radiochemotherapy for esophageal cancer and had lived in Cambodia until recently. On MRI, the inferior portion of the femoral head and neck, the greater and lesser trochanter, and the proximal shaft of the femur showed heterogeneous high signal intensity on T2-weighted MRI and low signal intensity on T1-weighted MRI. On post-contrast MRI, some areas of the right femur that showed high signal intensity on T2-weighted MRI did not show enhancement, suggesting that they contained necrotic tissue. These non-enhanced areas, which were thought to consist of necrotic tissue, can provide strong evidence of melioidosis on imaging evaluation.

In conclusion, melioidosis should be suspected when there is an infectious disease such as pneumonia in patients with chron- ic diseases including diabetes mellitus who have visited an area where the disease is prevalent including Southeast Asia and northern Australia. Furthermore, on initial or follow-up evalu- ation, necrosis or abscess may be strong evidence of melioidosis.

ReFeReNCes

1. Kim SW, Kwon GY, Kim B, Kwon D, Shin J, Bae GR. Imported melioidosis in South Korea: a case series with a literature review. Osong Public Health Res Perspect 2015;6:363-368 2. Lim KS, Chong VH. Radiological manifestations of melioido-

sis. Clin Radiol 2010;65:66-72

3. Muttarak M, Peh WC, Euathrongchit J, Lin SE, Tan AG, Lert- tumnongtum P, et al. Spectrum of imaging findings in mel- ioidosis. Br J Radiol 2009;82:514-521

4. Cheng AC, Currie BJ. Melioidosis: epidemiology, pathophysi- ology, and management. Clin Microbiol Rev 2005;18:383- 416

5. Kwon KY, Yoon SK. Characteristic of melioidosis and current status of infection in Korea. Public Health Wkly Rep 2012;

5:10-12

6. Korea Centers for Disease Control and Prevention. Guideline for melioidosis prevention and control KCDC. Available at:

http://cdc.go.kr/CDC/notice/CdcKrTogether0302.jsp?menuIds=

HOME001-MNU1154-MNU0005-MNU0088&cid=68797/.

Published Jul 12, 2016. Accessed Mar 15, 2017

7. Jain VK, Jain D, Kataria H, Shukla A, Arya RK, Mittal D. Meli- oidosis: a review of orthopedic manifestations, clinical fea- tures, diagnosis and management. Indian J Med Sci 2007;

61:580-590

8. Currie BJ, Ward L, Cheng AC. The epidemiology and clinical spectrum of melioidosis: 540 cases from the 20 year Darwin prospective study. PLoS Negl Trop Dis 2010;4:e900

9. Raja NS, Scarsbrook C. Burkholderia pseudomallei causing bone and joint infections: a clinical update. Infect Dis Ther 2016;5:17-29

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우측 대퇴부에서 발생한 근골격계 유비저의 임상적, 영상학적 소견:

증례 보고

김명현 · 전동진*

유비저는 Burkholderia pseudomallei에 의해 발생하는 감염성 질환이다. 이는 주로 열대 지방에서 발생하는 풍토병으로 한 국에서는 드물다. 하지만 최근 발병률이 점점 증가하고 있고 높은 사망률을 보여 초기 진단의 중요성이 부각되고 있다. 유 비저는 인체의 어떤 장기에서도 발병 가능하며 다양한 증상을 일으킬 수 있다. 특히 근골격계 유비저 환자는 빈도가 매우 드물고 비특이적인 근골격계 증상을 일으킬 수 있다. 현재 알려져 있는 근골격계 유비저의 영상학적 소견은 골수염의 형태 나 농양을 동반한 화농성 관절염 또는 골종양을 모방하는 것으로 알려져 있다. 이에 우리는 본 증례발표에서 64세 남자 환자의 우측 대퇴골과 우측 고관절에서 발생한 유비저의 임상학적 그리고 영상학적인 소견에 대해 다루고자 한다.

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