A nine year-old girl from Myittkyina was admitted to Yankin Children’s Hospital and referred to the Rheumatology department, Yangon Specialty Hospital for specialist opinion and further management.The girl was suffered from a two week history of moderate fever (102F) and one week history of shortness of breast.
Her past medical history included: diagnosis of juvenile rheumatoid arthritis (JRA), based on history of fever, multiple joint pain, generalized lymphadenopathy, ANA +, anti DsDNA –ve and high ESR .She had no history of evanescent rash or uveitits. Since that time, she has had bicytopenia : Hb-9.1,WCC-3.29, Plt- 350.
Her medication included: Methotrexate (2.5mg 3 tablets/week ), folic acid, ibuprofen and methyl prednisolone. Her blood C&S reported Brevundimonas diminute which was treated with co-amoxiclav and her symptoms seemed to improve with the prescribed treatment. She has been symptoms free for one and half years.
On this admission, clinical signs of a massive pleural effusion were detected which was confirmed by CXR (PA) and USG (chest). Pleural fluid aspiration was done and RE results showed that exudative pleural effusion with parapneumonic picture as evidenced by protein- 47 g/l , total cell count-3550 cells/ cumm,PMN- 80%, lymphocytes- 20% . The T test was found to be negative but she was already on one type of DMARDS: methotrexate for over one and half year for JRA.
Other immunological investigation revealed : ANA-(+) 1/800 dilution (homogenous and speckled pattern), Anti DsDNA (+) 108.0 IU/ml , low complement level C3-1.2 g/l, C4- 0.15 g/l and anti ENA profile (Antibodies to SSA,SSB,Ro 52,nucleosome). Thus, her diagnosis changed to SLE. In SLE and JRA in paediatric population, the two diseases often overlap and is not uncommon. Results of other tests showed: inflammatory markers: ESR- 95mm/1sthr, CRP- 103.98 mg/l, urine RE NAD, serial urine albumin test (neg), 24 hour urine protein-121mg/ dl, CBC-Hb-8.4, WCC-3.52, ANC-1.64, lymphocytes-1.54. indicating Bicytopenia, in spite of ANC and lymphocytes being on the low side of normal limit. Serology tests were negative with normal liver and renal function and T&DP not being in the reverse pattern.
Treatment included : IV CS1 and Flumox together with high dose methylprednisolone for three days. Her fever subsided after 48 hours and her symptoms improved.
Her fever subsided after 48 hours and symptoms improved.
Biochemical markers improved on the 9th day : CBC-Hb-10, WCC-10.8, ANC-9, L – 1.7, plt -518, CRP was normal and USG (chest) recheck showed mild pleural effusion with left basal consolidation. Her sputum C&S indicated: Leuconostoc mesenteroides sensitive to benzyl penicillin, cefepime, linezolid and vancomycin. Because of the irregular coccoid morphology, misdiagnoses was made as Lactobacillus, streptococcus (espviridans group), pepdicoccus, enterococcus since all share several biochemical properties.
Before 1985, Leuconostoc was considered non-pathogenic but with rising case reports, it is now considered an opportunistic infection with five subspecies which can be identified by additional physiological tests. The reported infections include: bacteremia, meningitis, breast abscess, peritonitis and abdominal abscess. Leuconostoc are sometimes found in healthy individuals and in nosocomial infections related to catheters, blood, etc.
In the designated case, the young girl presented in an immunocompromised state with leukopenia . Although neutrophil and lymphocyte counts are within normal range, in conditions like ‘functional neutropenia’ and lymphopenia, the white cells are unable to function normally.
In our clinical setting, the incidence of Leuconostoc infection has also increased and there have been seven cases identified in a single laboratory since 2015. The cultures were from sputum, urine and wound. Like this present case, one of the cases presented was from an immunocompromised SLEpatientwhohadaUTI and a urine culture which revealed Leuconostoc. The girl had a culture history with Brevundimonas diminuta which was non-lactose fermenting, gram negative bacilli belonging to the genus Pseudomonas.These organism are infrequently isolated in clinical microbiology laboratories but found in cancer patients. Anti CD20 treatment was planned for the girl.
