Severe Meliodosis With Multisystemic Involvement: A Case Report

Emergency and Critical Care Team, Kauvery Hospital, Cantonment, Trichy, India

Abstract

Melioidosis is a potentially life-threatening infection caused by the Gram-negative bacterium Burkholderia pseudomallei. Diagnosis of melioidosis is difficult due to its diverse clinical manifestations and ability to mimic many other diseases. We report a case of severe melioidosis in a patient known to have diabetes, who developed lung involvement, with multiple cavitating nodules in both lungs, and also mediastinal lymphadenopathy, both seen in thoracic computed tomography. He had an extrapulmonary lesion on the left side of cheek. Lesions were also seen in the infratemporal fossa with extension into left anterior temporal lobe with bony erosion. B. pseudomallei was cultured from blood sample. This case report highlights the importance of recognizing cavitating nodules as a radiological manifestation of melioidosis to facilitate the prompt administration of appropriate antibiotic treatment, thus reducing morbidity and mortality.

Keywords

Melioidosis, Burkholderia pseudomallei, Cavitating nodules

Background

Melioidosis is caused by B. pseudomallei, a Gram-negative bacillus commonly found in soil or contaminated water, and it is an endemic disease in northern Australia and Southeast Asian countries, in particular Thailand and Malaysia. The clinical spectrum of melioidosis is complex and wide-ranging and includes latent infection, subacute pneumonia, local cutaneous lesions, focal organ abscess, musculoskeletal infection, bacteremia, and lethal fulminant pneumonia. Early diagnosis of melioidosis is challenging for clinicians given its ability to mimic many other diseases and its association with a high mortality rate. We report a case of culture-confirmed pulmonary melioidosis with the cavitating nodules identified on computed tomography (CT) thorax.

Case presentation

A 41-years-aged male, known to have hypothyroidism and T2DM, presented with complaints of loss of appetite and fatigue, scattered skin lesions, significant weight loss, dry cough, and low-grade fever. Patient was diagnosed as UTI/DKA in an outside hospital and was treated empirically a with Meropenem and Teicoplanin, and other supportive medications. Following this he developed neck pain, low back ache, and parasthesia left upper and lower limbs.

He was brought to our hospital for further care. The patient was investigated or infective causes of fever. Patient had Type 1 respiratory failure with bilateral lung collapse and consolidation and mild effusion.

Mediastinal lymphadenopathy was noted with central necrosis was noted on radiology.

MRI whole spine screening showed inflammation, contrast enhancement- evidence of involvement of prevertebral layer/ thin rim anterior to the cord from C2 to C4 region.

MRI brain showed altered signal intensities on the left side of cheek, involving the mucosa pterygoid muscle, left Tempero Mandibular, infratemporal fossa, extending to the anterior temporal lobe with thickened adjacent meninges.

Patient was treated with empirical antibiotics, antipyretics, proton pump inhibitors, antiemetics and other supportive medications.

On the second day of admission patient deteriorated and was intubated, put on mechanical ventilation with high fio2 and PEEP.

He developed shock and multiple ionotropes were initiated.

Subsequently blood cultures showed Burkholderia pseudomallei and diagnosis of melioidosis was confirmed. He was continued with Meropenem and Minocycline which were sensitive.

Patient had other blood parameters serially monitored and correction given accordingly.

ECHO showed features of stress cardiomyopathy, NT proBNP was elevated.

Tracheostomy was done to facilitate weaning.

He developed Critical Illness Associated Polyneuropathy.

With aggressive treatment and careful monitoring patient condition gradually improved. Inotropes were tapered, and drugs altered according to clinical condition.

LV functions improved.

Patient shifted to ward with tracheostomy – 2 L of O2 support. Patient clinical condition improved well and Decannulation of tracheostomy was done.

Patient tolerated well in room air. He was mobilized with walker support, and later discharged in stable status on prescribed medication.

Severe-Meliodosis-1
Severe-Meliodosis-2

Fig. 1a & b. CT-Chest: Bilateral moderate pleural effusion with passive atelectatic changes in bilateral lower lobes. Multiple cavitating nodules in bilateral lung fields.

Discussion

Melioidosis is caused by B. pseudomallei, a facultative intracellular Gram-negative saprophytic bacterium commonly found in soil or contaminated water. It is a multiorgan infectious disease which can present with a wide spectrum of clinical presentations. The Darwin study found pneumonia to be the most common presentation of melioidosis in approximately half of cases. Less common presentations include genitourinary infection (14%), skin infection (13%), bacteremia without evident focus (11%), septic arthritis or osteomyelitis (4%) and neurological melioidosis.

Melioidosis is sometimes called “the great imitator” because it can mimic many other diseases. Therefore, diagnosis of melioidosis remains a challenge for clinicians. There is a substantial body of published case reports on melioidosis mimicking tuberculosis. A study in India showed that 8 out of 22 cases of confirmed melioidosis, were mimicked by pulmonary tuberculosis. In melioidosis, upper lobe involvement with early cavitation and rapid progression were common. In the subacute and chronic forms, the radiographic pattern sometimes mimicked tuberculosis, with upper lobe involvement, patchy alveolar infiltrate with cavities or fibroreticular lesions.

Nodular opacity can be visualized in a pulmonary cavitary process.

CT scan is classically described in pulmonary aspergillosis: either with an aspergilloma in a pre-existing lung cavity historically most often formed by previous tuberculous infection; or during the recovery phase of invasive aspergillosis when air fills the space between the retracting devitalized infected lung tissue and the parenchyma.

However, other pathology can also manifest with the Cavitating nodules most notably tuberculosis itself or a cavitary lung malignancy.

A previous case of melioidosis with the multiple cavitating nodules has also been to possible presentation.

Pneumonia is the presenting feature of over half of all melioidosis cases, with a wide diversity ranging from acute, fulminant sepsis with multifocal lung infiltrates to subacute unilateral pneumonia to chronic infection..

Risk factors for melioidosis include diabetes mellitus, hazardous alcohol use, pre-existing renal disease, immunosuppressive therapy including steroids and thalassemia. Occupational or recreational exposure to contaminated soil or water is the usual infecting event.

Our patient was diagnosed with diabetes mellitus which may have predisposed him to melioidosis infection.

Pulmonary melioidosis presenting with pleural effusion occurred in 12.2% of the total 162 cases reported between 1996 and 2002 in study conducted in Thailand. An interesting observation was that our patient had lymphocyte-predominant pleural effusion, and this finding was in accordance with a case report by Chung et al. whereby lymphocytic pleural effusion was found in all 3 patients with acute melioidosis. Therefore, melioidosis should be considered in the differential diagnosis of lymphocytic pleural effusion, which otherwise was most commonly seen in tuberculosis or malignancy.

Guidelines for melioidosis therapy have been standardized with initial intravenous ceftazidime or meropenem, followed by eradication therapy with sulfamethoxazole/trimethoprim. Our patient developed parapneumonic effusion following inadequate response to amoxicillin-clavulanic acid. B. pseudomallei is susceptible to amoxicillin-clavulanic acid, hence the initial clinical response in this case, but outcomes with amoxicillin-clavulanic acid are inferior to standard therapy.

In conclusion, the cavitatory nodules can be found in several conditions, and it is not always due to fungal infection. It is important for clinicians to be able to recognize the cavitatory nodules as a radiological manifestation of melioidosis to enable prompt administration of empirical melioidosis treatment in suspected cases to reduce the morbidity and mortality. In this present case, appropriate clinical evaluation and use of diagnostic techniques, such as CT thorax and diagnostic blood cultures may facilitate early diagnosis and management.

As an emergency physicians, ever play a significant role in sepsis bundle protocol and ever keep in mind about tropical time bomb.

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