Relapse of chronic melioidosis in a paediatric cystic fibrosis patient: first case report from Malaysia

Abstract

BackgroundBurkholderia pseudomallei is the causative agent of melioidosis, which is a potentially life threatening disease endemic in Southeast Asian countries. In Malaysia, cystic fibrosis (CF) is an uncommon condition. The association between CF and B.pseudomallei infections has been reported previously. However, this is the first case report of a pediatric melioidosis relapse and co-infection with other Gram-negative bacteria in Malaysia.Case presentationA 14-year-old Chinese Malaysian boy presented with a history of recurrent pneumonia, poor growth and steatorrhoea since childhood, and was diagnosed with CF. B. pseudomallei was cultured from his sputum during three different admissions between 2013 and 2016. However, the patient succumbed to end stage of respiratory failure in 2017 despite antibiotics treatment against B.pseudomallei. The isolates were compared using multilocus-sequence typing and repetitive-element polymerase chain reaction (PCR), and confirmed that two of the isolates were of same sequence type, which may indicate relapse.ConclusionsCF patients should be aware of melioidosis in endemic regions, as it is an emerging infectious disease, especially when persistent or recurrent respiratory symptoms and signs of infection occur. The high prevalence rates of melioidosis in Malaysia warrants better management options to improve quality of life, and life expectancy in patients with CF. Travel activities to endemic regions should also be given more consideration, as this would be crucial to identify and initiate appropriate empiric treatment.

The Brief Case: A Traveler’s Tale—Burkholderia pseudomallei Infection in a Cystic Fibrosis Patient

“…Infection is acquired through direct inoculation, inhalation, or ingestion of contaminated soil or water with an incubation period of 1 to 21 days (4-10). The typical presentation with melioidosis in adults is pneumonia, which ranges from mild disease to subacute pulmonary disease mimicking tuberculosis to fulminant septic shock (4)(5)(6)(7)(8)(9)(10). Extrapulmonary presentations such as cutaneous infections, septic arthritis, genitourinary infections, and encephalitis have been reported (4)(5)(6)(7)(8)(9)(10).…”

“…This is followed by an eradication phase, which consists of 3 to 6 months of oral antimicrobials. Trimethoprim-sulfamethoxazole is the drug of choice for eradication, but amoxicillin-clavulanic acid and doxycycline are alternatives (4)(5)(6)(7)(8)(9)(10). Mortality is high in melioidosis associated with sepsis (50% to 90%) despite adequate treatment (10).…”

Towards an Integrated Approach to Improve the Understanding of the Relationships Between Water-Borne Infections and Health Outcomes: Using Malaysia as a Detailed Case Study

“…There was also a report of a diabetic patient who worked as a palm oil worker in Perak, being infected with melioidosis, tuberculosis and Salmonella at the same time, suggesting diabetes as a predisposing factor to multiple infections (Sulaiman et al, 2013). Recorded risk factors for melioidosis include co-morbidities, immune-compromission, cystic fibrosis (Mariappan et al, 2018), thalassemia, kwashiorkor, albinism and patent ductus arteriosus (Fong et al, 2015). Residing areas also play an important role in acquiring melioidosis, Abu Hassan et al (2019) showed that living in areas with large-scale irrigation-based agriculture and mixed agriculture/pastoral environments had a higher prevalence rate of melioidosis (Abu Hassan et al, 2019).…”