Case Report

Septic Arthritis due to Melioidosis: Rare but can be fatal

Summary

A fifty six year-old farmer from Rakhine State presented with high fever with painful left knee swelling for three weeks and confusion and irrelevant talk for one week. The high fever was not associated with chills and rigor. No prior history of operation, knee trauma, cough, chest pain, abdominal pain or mass. He has a three-year history of poorly controlled Type 2 diabetes mellitus.On examination,GCS was 11/15, temperature was 102’C and vital signs were stable. Left knee examination presented joint effusion with signs of inflammation.Investigations showed marked neutrophil leukocytosis with increased CRP. Blood and synovial fluid culture revealed Burkholderia Pseudomallei. He was treated with IV ceftazidine 2G q 8 hoursand changed to IV meropenem 500mg q 6 hours for 4 weeks according to culture and sensitivity results and followed by oral Amoxicillin/clavulanic acid for 3 months to prevent relapse. Wound and joint debridement was done and post operation days were uneventful.After a one month in hospitalization, he was discharged with planned follow up care in his hometown with a physician and orthopedic surgeon.

Background

Without proper medical treatment, melioidosis is an important fatal tropical disease with a high mortality rate (30-47%) and is endemic in Myanmar. From 1913 to 2018, 298 cases of melioidosis was reported in Myanmar but 257 cases were detected before 1948. Awareness is poor among both clinicians and microbiologists and definite epidemiology is unavailable. 1In 60-90% of melioidosis cases, underlying diseases exist: Diabetes Mellitus, chronic renal failure, steroid therapy, alcohol abuse, liver disease, chronic lung disease, cystic fibrosis, cardiac failure, malignant disease, thalassemia, chronic granulomatous disease and pregnancy.However, HIV infection does not appear to increase risks. Melioidosis usually presents with the following: pneumonia, skin lesions without systemic illness, overwhelming sepsis with abscesses disseminated in multiple internal organs, genitourinary disease with prostatic abscesses. Bone and joint involvement in melioidosis is rare but is a well documented problem.2Isolated septic arthritis, a rare but a recognized manifestation of melioidosis, can be the presenting problem in some patients. The knee joint has been identified as the most commonly affected joint in melioidosis, followed by ankle, hip and shoulder joints.3This interesting case highlights the importance of considering melioidosis in susceptible patients presenting with isolated septic arthritis in endemic areas, even if the other manifestations of melioidosis (abscess, lung involvement) are not present.

Case presentation

A 56 year-old farmer from Rakhine state in Myanmar presented with a three-week history of high fever and painful left knee swelling and one-week history of confusion and irrelevant talk. The high fever was not associated with chills and rigor. No history was noted for: loss of weight and appetite, past operation, knee trauma,cough, chest pain or abdominal pain or mass. Severe pain prevented walking and he presented with a one-week history of drowsiness and irrelevant speech.

Past Medical History

History of hypertension controlled with antihypertensive and three-year history of poorly-controlled Diabetes Mellitus.

Epidemiological history

Occupation: rice farmer from Kyeik taw, Rakhine State

No history alcohol intake or intravenous drug use

Physical Examination:

The patient appeared confused with GCS 11/1and eye 4, verbal 3 and motor 4. Vitals included: blood pressure:120/80 mm Hg; pulse:105 beats per minute;temperature:102 o F; and respiration rate: 22 times per minute. Joint effusion with inflammation signs noted on the left knee but no skin ulcers. All other joints were normal and system examinations were normal.

Investigations

RBS was 330 mg% with Hb A1c was 10%. On full blood count revealed Hb was 12 g/dl (11-17.5g/dl), WBC was 27x 109/L, platelet was 230x 109/L and creative protein was 167 mg/L. Renal and liver function were within normal limits. Left knee joint X-ray showed joint effusion with underlying OA changes. CXR(PA) and USG (abdomen) were normal.Synovial fluid analysis revealed yellow turbid fluid with cell count of 3500 cells/mm3. Synovial fluid culture showed Burkholderia Pseudomallei and sensitive to Amoxicillin /clavulanic acid,Cefotaxime,Ciprofloxacin,Cotrimoxazole, Cefoparazone /salbactam Imipenem,Levofloxacin ,Piperacillin/tazobactam and tetracycline.Blood culture also done before antibiotic and turned out to be Burkholderia Pseudomallei. And sensitive to Amoxicillin/clavulanic acid, Ampicillin /sulbactam, Piperacillin / tazobactam, Imipenem, Meropenem , Cefoparazone/sulbactam, Choramphenicol Intermediate to Ciprofloxacin, Levofloxacin and resistant to Amikacin and Gentamicin.

The patient was initially treated with IV ceftazidine 2G 8 hourly as empirical treatment for septic arthritis and change to IV meropenem 500mg 6 hourly for four weeks according to culture and sensitivity results because of septicemiawith very high CRP. Treatment was followed by PO Amoxicillin/clavulanic acid for another three months to prevent relapse or reinfection.Wound and joint debridement was done one day after admission and post operation days were uneventful.

Outcome and follow up

During the hospital stay, painful joint swelling was reduced and WBC count and inflammatory marker were decreased. After about one month in hospital, he was discharged and planned to attend follow up at his hometown with joint care of physician and orthopedic surgeon.

Discussion

Melioidosis is a fatal disease prevalent in South-East Asia, Northern Australia, and the Indian subcontinent and is caused by Gram-negative saprophyte Burkholderia pseudomallei.4 The disease is known as a remarkable imitator due to the wide and variable clinical spectrum of its manifestations.5 The clinical manifestations of the acute illness vary from mild localized suppurate skin lesions to severe pneumonia or septicemia.6 Septic arthritis is rare but a well-recognized manifestation of this disease. 5The choice for diagnosis is culture and sensitivity.The Royal Darwin Hospital treatment guideline for initial intensive therapy includes:Ceftazidime (wards) 2 g IV, 6-hourly for at least 14 days or Meropenem (ICU) 1 g IV, 8-hourly for at least 14 days. Eradication therapyis required after the initial intensive therapy with Trimethoprim + sulfamethoxazole 40-60kg, 240+1200mg; >60kg, 320+1600 mg orally, 12-hourly for at least 3 months plus Folic acid 5 mg orally, daily for at least 3 months. 9There are many case reports of septic arthritis due to melioidosis from various parts of world. In Venezuela, a 50 year-old diabetic man presented with repeated episodes of arthritis and osteomyelitis with sepsis syndrome. Blood and synovial culture revealedBurkholderia pseudomallei.7 In a case report from India, a 43-year-old male with no morbidities presented with septic arthritis and left pleural effusion and cultures of synovial and pleural fluid revealed Burkholderia pseudomallei.4 Another case report from India stated that a 52 year-old male with uncontrolled diabetes mellitus (DM) presented with a rare combination of septic arthritis and abscesses in the chest wall, liver and subcutaneous tissue. The patient responded to prolonged treatment of intravenous ceftazidime followed by oral co-trimoxazole.5 Moreover, a case report from Sri Lanka reported that a middle aged diabetic female who had been on azathioprine for autoimmune hepatitis,presented with septic arthritis which turned out to be melioidosis.8 Therefore,all these case reports and our case highlight that induced septic arthritis is not a very rare presentation of disease. A report regarding melioidosis in Myanmar stated that there are lack of awareness of this disease among clinicians and there is insufficient laboratory diagnostic capacity in many parts of Myanmar.10

Take home message

In endemic areas, melioidosis needsto be considered in patients presenting with risk factors as diabetes, immunosuppression state or isolated septic arthritis.These case reports have been presented to raise the awareness of an unusualpresentation of melioidosis: isolated septic arthritis.

References:

  1. Mo Mo Win , Elizabeth A. Ashley , Khwar Nyo Zin, Myint Thazin Aung ,Myo Maung Maung Swe, Clare L. Ling , François Nosten ,Win May Thein , Ni Ni Zaw ,May Yee Aung , Kyaw Myo Tun , David A. B. Dance and Frank M. Smithuis Trop. Med. Infect. Dis. 2018, 3, 28;
  2. Bart Currie,Royal Darwin Hospital and Menzies School of Health Research, Darwin. The Northern Territory Disease Control Bulletin Vol 2 4 1 No. 2 June 2014
  3. Kosuwon W, Taimglang T, Sirichativapee W, Jeeravipoolvarn P. Melioidotic septic arthritis and its risk factors. J Bone Jt Surg. 2003;85-a(6):1058–61.
  4. Patil N, Balaji O, Rao KN, Hande MH, Ahmed T, Singla S. A Rare Cause of Septic Arthritis with Pleural Effusion: Burkholderia Pseudomallei. Asian Journal of Pharmaceutical and Clinical Research. 2017;10(1):1-2.
  5. Rajadhyaksha A, Sonawale A, Khare S, Kalal C, Jankar R. Disseminated melioidosis presenting as septic arthritis. J Assoc Physicians India. 2012 Jun;60:44-5.
  6. Borgmeier PJ, Kalovidouris AE. Septic arthritis of the sternomanubrial joint due to Pseudomonas pseudomallei. Arthritis & Rheumatism: Official Journal of the American College of Rheumatology. 1980 Sep;23(9):1057-9.
  7. Redondo MC, Gomez M, Landaeta ME, Rios H, Khalil R, Guevara RN, Palavecino S, Figuera M, Caldera J, Rivera R, Calvo A. Melioidosis presenting as sepsis syndrome: a case report. International Journal of Infectious Diseases. 2011 Mar 1;15(3):e217-8.
  8. Weerasinghe NP, Herath HM, Liyanage TM. Isolated septic arthritis of hip joint: a rare presentation of melioidosis. A case report. BMC research notes. 2018 Dec;11(1):50.
  9. Currie B. Melioidosis: the 2014 revised RDH guideline. NT Disease Control Bulletin. 2014;21(2):4-8.
  10. Win MM, Ashley EA, Zin KN, Aung MT, Swe MM, Ling CL, Nosten F, Thein WM, Zaw NN, Aung MY, Tun KM. Melioidosis in Myanmar. Tropical Medicine and Infectious Disease. 2018 Mar 1;3(1):28.

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