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CASE REPORT
1
Department of Pediatrics, 2Department of Microbiology, Faculty of Medicine Siriraj
Hospital, Mahidol University, Bangkok, Thailand
low grade, usually at night, and accompa- showed bilateral interstitial infiltrates.
nied by arthralgia, myalgia, and frequent Additional laboratory investigations on
epistaxis. Three days prior to admission admission included serology for Epstein-
at Siriraj Hospital, Mahidol University, Barr virus, human immunodeficiency vi-
Bangkok, he developed high fever and rus, cytomegalovirus, Burkholderia pseudo-
pallor which prompted his parents to seek mallei, and Orientia tsutsugamushi, as well
medical attention. as blood smears for malaria, a tuberculin
He was admitted at Siriraj Hospital skin test, and a sputum acid-fast stain. The
because of prolonged fever. On admission, results were later found to be negative. To
he appeared ill and pale but was alert and investigate the cause of the anemia and
oriented. His body temperature was at thrombocytopenia, a bone marrow biopsy
38.5ºC, his respiratory rate was 20/min, was performed, which revealed normal
his heart rate was 112/min, and his blood cellularity and maturation, infrequent
pressure was 92/64 mmHg. His weight hemophagocytosis, no clusters of blasts
and height were 30 kg (10th-25th percen- or abnormal lymphoid cells, a slight in-
tile), and 149.3 cm (50th-75th percentile), crease in the number of megakaryocytes,
respectively. He had a left cervical palpa- a decrease in iron deposition, and no ring
ble lymph node 1 centimeter in diameter. sideroblasts. The bone marrow examina-
He had non-tender hepatosplenomegaly tion was also negative for acid-fast bacilli,
with a liver palpable 4 centimeters below fungi, malignancy or granulomas. A blood
the right costal margin, and the spleen culture was obtained on admission and he
palpable 3 centimeters below the left was put on cefotaxime 100 mg/kg/day as
costal margin. No other abnormalities empirical treatment.
were detected. He had been living on a He continued to have high grade
pineapple farm in Sam Roi Yot District, fever to a maximum of 39.8ºC. On day
Prachuap Khiri Khan Province, southern 5 of hospitalization he developed ab-
Thailand. Additional history revealed dominal pain with guarding and rigidity
there were many goat farms in the area, suggesting peritonitis. A computerized
and he had contact with goats approxi- tomogram revealed bilateral pleural ef-
mately 2 months prior to the onset of fever fusions, ascites, hepatosplenomegaly and
through holding goats for immunization mesenteric lymphadenopathy. Abdominal
at his uncle’s farm. paracentesis revealed 50 ml of clear yel-
The complete blood count (CBC) low fluid. Peritoneal fluid examination
revealed a hemoglobin of 8.5 g/dl, a revealed a red blood cell count of 1,600/
hematocrit of 26.8%, a MCV of 73.8 fl, mm3, a WBC count of 110/mm3 with100%
a MCH of 23.4 pg, a MCHC of 31.7 g/ lymphocytes, a glucose level of 101 mg/
dl, a RDW of 16.8%, a white blood cell dl, a LDH of 724 U/l and an albumin of 2.1
count (WBC) of 4,230 cells/mm3 with 38% g/dl. A Gram stain and acid-fast stain of
neutrophils, 58% lymphocytes, and 3% the peritoneal fluid were both negative for
monocytes and a platelet count of 90,000/ organisms, and a culture of the fluid failed
mm3. His urinalysis was normal. Blood to detect bacteria, fungi or mycobacteria.
chemistry revealed normal electrolytes, A polymerase chain reaction (PCR) of the
renal function and liver function with an peritoneal fluid for tuberculosis was nega-
alanine transaminase of 73 U/l and aspar- tive. A repeat CBC revealed a hemoglobin
tate transminase of 31 U/l. A chest x-ray of 9 g/dl, a hematocrit at 29.3%, a WBC
count of 4,100/mm3 with 43% neutrophils, with many clinical presentations (Hati-
57% lymphocytes and 1% monocytes, and poglu et al, 2004). Brucellosis in children
a platelet count of 87,000/mm3. A pro- is frequently mild, self-limited, and less
thrombin time and partial thromboplastin likely to be chronic compared to adults
time were normal. A fibrinogen level was (Shaalan et al, 2002). Brucellosis in adults
231.3 mg/dl (normal 200-400) and the usually causes fever (75.5%), night sweats
D-dimer was 5,379.8 µg/l (normal < 500 (69.7%) and arthralgia (75.5%) (Hatipoglu
µg/l). The antibiotic regimen was empiri- et al, 2004). The most common symptoms
cally changed to piperacillin/tazobactam in children are fever, malaise/myalgia
300 mg/kg/day to improve coverage of an and arthralgia (al-Eissa and al-Nasser,
intra-abdominal infection. 1993; Mantur et al, 2004; Giannakopoulos
On day 8 of hospitalization, the pa- et al, 2006). The most common sign in
tient remained febrile and had no signs of children is hepatomegaly, followed by
improvement. Blood cultures taken on ad- splenomegaly and lymphadenopathy
mission examined with the automate sys- (Giannakopoulos et al, 2006); all were
tem (BacT/Alert) revealed growth of small found in our case. The most common
gram-negative aerobic bacilli, suspected complications involve bone and joint,
to be Brucella sp. This finding prompted a particularly peripheral arthritis in adult
change in therapy to oral doxycycline 100 (Mousa et al, 1987) and spondylitis in the
mg twice daily and intravenous gentami- older age (Colmenero et al, 1996). The os-
cin 5 mg/kg/day. The patient responded teoarticular involvement in children was
well to the treatment. The signs of ab- monoarticular predominantly affecting
dominal peritonitis disappeared within 48 hips or knees (al-Eissa et al, 1990; Ben-
hours and the fever subsided by 72 hours jamin et al, 1992; Bosilkovski et al, 2013).
of treatment. Blood cultures taken on days The infection may involve several organ
4 and 7 of hospitalization also grew the systems, including hematologic, genito-
same organism, which was later identified urinary, gastrointestinal, hepatobiliary,
as Brucella melitensis using biochemical cardiovascular, nervous, and respiratory
tests and the automated Vitek 2 system. systems (Mantur et al, 2001; Kantarçeken
The organism was also confirmed to be et al, 2005; Mantur et al, 2006; Ozisik et al,
Brucella sp by 16S rDNA sequencing. The 2006). There have been reports of unusual
minimal inhibitory concentrations (MIC) presentations, such as neurobrucellosis,
for trimethoprim-sulfamethoxazole and pericarditis, pancytopenia, epididymo-
gentamicin determined by the E-test were orchitis, uveitis, mixed cryoglobulinemia
0.032 µg/ml and 0.19 µg/ml, respectively. with renal failure, cutaneous vasculitis
He recieved 2 weeks of daily gentamicin and peritonitis (Hatipoglu et al, 2004;
and 6 weeks of doxycycline. By 4 weeks Hermida Lazcano et al, 2005; Dizbay
of treatment, he had complete clinical re- et al, 2007). Fatal outcomes occur due to
covery and a normal CBC. He continued extensive vasculitis (Dizbay et al, 2007)
to do well for 6 months follow-up after often accompanied by encephalopathy
therapy was completed. (Caksen et al, 2003).
Hematologic alterations in brucellosis
DISCUSSION are common (Martin-Moreno et al, 1983;
Crosby et al, 1984; Aysha and Shayib, 1986).
Brucellosis is a multisystemic disease A large prospective study in adults found
has been reported to have higher relapse clinical manifestations and bone marrow
rates and is usually used in triple drug findings. Acta Haematol 1993; 89: 132-6.
regimens (Mantur et al, 2007). We thus al-Eissa YA, Kambal AM, Alrabeeah AA,
opted to treat our patient with gentamicin Abdullah AM, al-Jurayyan NA, al-Jishi
for 2 weeks and doxycycline for 6 weeks. NM. Osteoarticular brucellosis in children.
He responded rapidly and had no signs Ann Rheum Dis 1990 ; 49: 896-900.
of relapse during 6 months of follow-up. Anonymous. Joint FAO/WHO expert commit-
tee on brucellosis. World Health Organ Tech
Goats remain the main source of B.
Rep Ser 1986; 740: 1-132.
melitensis (Corbel, 1997). A review of 7
adult cases of brucellosis caused by B. Aysha MH, Shayib MA. Pancytopenia and
other haematological findings in brucel-
melitensis in Thailand during 1970 to
losis. Scand J Haematol 1986; 36: 335-8.
2005 revealed 29% (2/7) had a history of
consuming non-pasteurized goat milk Benjamin B, Annobil SH, Khan MR. Osteoar-
ticular complications of childhood brucel-
and 57% (4/7) had a history of contact
losis: a study of 57 cases in Saudi Arabia. J
with goats (Paitoonpong et al, 2006). In
Pediatr Orthop 1992; 12: 801-5.
our case, we hypothesize that the mode
Bosilkovski M, Kirova-Urosevic V, Cekovska
of contraction was direct contact with
Z, et al. Osteoarticular involvement in
infected goats.
childhood brucellosis: experience with 133
In conclusion, we report here the first cases in an endemic region. Pediatr Infect
case of brucellosis in a child in Thailand Dis J 2013; 32: 815-9.
whose case included peritonitis. Brucel- Caksen H, Odabas D, Köse D, Anlar O. A fatal
losis has a broad range of signs and symp- case of brucellosis displaying an atypical
toms and is difficult to diagnose based on clinical course. J Emerg Med 2003; 25: 472-4.
clinical findings alone. In non-endemic Colmenero JD, Reguera JM, Martos F, et al.
areas, such as Thailand, brucellosis is a Complications associated with Brucella
diagnostic challenge. Failure to recog- melitensis infection: a study of 530 cases.
nize brucellosis and provide appropriate Medicine (Baltimore) 1996; 75: 195-211.
antibiotic treatment may result in serious Corbel MJ. Brucellosis: an overview. Emerg
complications or death. It is important to Infect Dis 1997; 3: 213-21.
include brucellosis in the differential diag- Corbel MJ. Brucellosis in humans and animals.
nosis of children with prolonged fever and Geneva: World Health Organization in col-
hepatosplenomegaly, especially in those laboration with the Food and Agriculture
with a history of exposure to animals. Organization of the United Nations and
World Organisation for Animal Health,
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