A 56 year old Sri Lankan female presented with fever, chills and rigors for 2 weeks. She was diabetic and her glycaemic control was fairly good with oral hypoglycaemic drugs: fasting blood sugar was 112 mg/dl and HbA1C was 5.7%. At presentation, fever was associated with constitutional symptoms like malaise and loss of appetite, but she didn't have any other symptoms on systemic review. She is a housewife, not engaged in gardening or farming.
On admission, upon examination, she was febrile (temperature was 101.4 0 F), did not have lymphadenopathy, oral ulcers, uveitis, peripheral signs of infective endocarditis, rashes or hepatosplenomegaly. Rest of her clinical examination was unremarkable.
On the third day of admission, blood culture reported positive with the growth of Burkholderia cepacia. She was started on IV ceftazidime 2 g every 6 h, with the suspicion of Burkholderia pseudomallei and melioidosis serology was ordered. Colony morphology consisted of grey-white small, smooth colonies. Gram stain showed gram negative slender rods with bipolar staining, antibiotic susceptibility pattern gave clues to the microbiological diagnosis of Burkholderia pseudomallei, sensitive for ceftazidime and meropenem and resistant to gentamycin (Figs. 1 and 2). The melioidosis antibody titer Ig M was 1:640, measured by enzyme linked immunosorbent assay (ELISA). The isolate was confirmed as Burkholderia pseudomallei by PCR the following day.
She was continued on IV ceftazidime 2 g every six hours. During her 2nd week of hospital stay (24th day of fever), she developed right hip joint pain, right sided low backache, right buttock pain, difficulty walking and restricted right hip joint movements. She did not have any other joint involvement or history of arthritis. She did not have past history of tuberculosis or significant contact history of tuberculosis. She did not have change of bowel habits, red eyes to suggest inflammatory bowel disease. She didn't have any skin rashes to suggest psoriasis or vasculitis or recurrent oral ulcerations to suggest behcet's disease. She did not have a history of raw milk ingestion to suggest brucellosis. She didn't have family history of arthritis or autoimmune diseases. By that time, her right hip joint movements were restricted due to pain, she had severe tenderness over right sacroiliac joint and sacroiliac stretch maneuvers were positive for a clinical diagnosis of right sacroiliitis. She had a neutrophilic leukocytosis, with high inflammatory markers. Ultrasound Scan of the hip joint detected a large fluid collection measuring 5 cm x 18 cm involving the right iliopsoas, without extension into hip joint. A Contrast Enhanced CT scan (CE CT) of Chest, abdomen and pelvis showed multiple intramuscular abscesses over right psoas, iliacus, gluteus and obturator internus (the largest abscess measured 18 cm x 4 cm over right psoas and iliacus (Fig. 3)). There were no visceral abscesses in the liver or spleen. Initially US guided aspiration and drainage was carried out to drain the iliopsoas abscesses (by the 28th day of fever), followed by a pig tail catheter drainage over 5 days, since it was a large collection. The aspirated fluid culture was also positive for Burkholderia pseudomallei.
One week post aspiration, she had an MRI scan of spine and sacroiliac joints: which showed resolving infection in right iliacus muscle, with a thin collection of fluid and muscle oedema. It also showed right sacroiliitis with surrounding marrow oedema and mild reactive bone changes (Fig. 4). We didn't aspirate fluid from sacroiliac joint, since an alternative cause for sacroiliitis was unlikely, melioidosis was the likely cause for her right sacroiliitis.
Fever, multiple abscesses with sacroiliitis, pointed towards the musculoskeletal involvement of melioidosis. Her sacroiliitis was infectious in nature, so inflammatory causes like psoriatic arthritis, ankylosing spondylitis and inflammatory bowel disease were unlikely. Anyway we wanted to exclude co-infection of melioidosis with tuberculosis/ brucellosis/ fungal infections and immunosuppressed states like AIDS (Acquired Immuno Deficiency Syndrome).
Her Mantoux test was negative, chest X ray was normal, sputum for acid fast bacilli were negative and pus drained from abscesses didn't yield any growth on bacterial cultures, AFB cultures or fungal cultures. Brucella and HIV antibodies were also negative.
Despite treatment with IV ceftazidime, her fever spikes continued attributable to the complications with intramuscular abscesses and sacroiliitis. Her Melioidosis antibody titers obtained 2 weeks apart increased from 1/640 to 1/10240. Thus IV ceftazidime (given for 12 days) was changed over to IV meropenem 2 g every 8 h with oral co-trimoxazole 3 tablets (each tablet = 400 mg of sulfamethoxale and 80 mg of trimethoprim) every 12 h was added on the 28th day of fever.
Results of some of the relevant investigations are shown below, given in a Table 1.
According to the recent international guidelines, in deep seated abscesses, duration of intensive phase of melioidosis is extended and retimed from the date of most recent drainage of Burkholderia pseudomallei.. Therefore our patient's IV therapy was extended additional 14 days of abscess drainage. She developed pancytopenia, during the second week of treatment with co-trimoxazole. Her haemoglobin was 7.9 g/dL, white cell count was 2.8 x 109/L and platelet count was 113x 109 /L. This was changed into oral doxycycline 100 m grams every 12 h to avoid bone marrow suppression. Her intensive phase was continued with IV meropenem 2 g every 8 h another 6 weeks together with oral doxycycline. During the 6th week of intensive phase treatment, she also developed Syndrome of Inappropriate Secretion of ADH (SIADH), with persistent hyponatraemia (120-135 mmol/L), urinary sodium of 92 mmol/L, and serum osmolality 264mosm/kg and urine osmolality of 389mosm/l. She responded within 1 week with fluid restriction therapy alone, without change of medical management.
She was restarted on oral co-trimoxazole 4 tablets every 12 h, at the end of intensive phase treatment, (each tablet = 400 mg of sulfamethoxazole and 80 mg of trimethoprim) and discharged her on eradication phase (since cotrimoxazole is the best drug in eradication phase, it was restarted and she didn't develop pancytopaenia this time). She was followed up monthly for 6 months and she made an uneventful recovery at the end of eradication phase. Follow up MRI Scan of sacroiliac joints done after 2 months showed marked reduction of infection, oedema and sacroiliitis and by that time, she was completely asymptomatic (Fig. 5).