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Indian Pediatr 2017;54:
595-596 |
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Brucella Causing Liver
Abscess in a Child with Selective IgA Deficiency
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Devdeep Mukherjee, Priyankar Pal and RitabrataKundu
From Department of Pediatric Medicine, Institute of
Child Health, Kolkata, West Bengal, India.
Correspondence to: Dr Devdeep Mukherjee, Uttara
Cooperative Housing Society, 13, Broad Street, Kolkata 700 019, West
Bengal, India.
Email:
[email protected]
Received: August 07, 2016;
Initial review: November 09,2016;
Accepted: April 13, 2017.
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Background: Brucella has been known to cause
pyrexia of unknown origin. Case Characteristics: 9-year-old boy
with fever and abdominal pain; multiple abscesses within the liver on
ultrasonography. Observations: IgM Antibodies against Brucella
were raised in his serum sample, and Brucella serum agglutination test
was positive. Immunological work-up suggested selective IgA deficiency.
Reduction in size following treatment with
trimethoprim-sulphamethoxazole, amikacin and doxycycline. Message:
Brucellosis should be considered as an etiology of liver abscess in
patients not responding to conventional antibiotics.
Keywords: Brucellosis,Immunodeficiency, Management.
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L iver abscess due to
Staphylococcus, Streptococcus and Gram-negative organisms like E.coli,
Klebsiella and Salmonella have been commonly reported in children. We
report liver abscess caused by Brucella in a 9-year-old child with
selective IgA deficiency.
Case Report
A 9-year-old boy presented to us with a history of
low grade fever for 3 weeks with intermittent abdominal pain localized
to the epigastric region. Pain was not related with food intake and
there was no history of vomiting, diarrhea, passage of worm or blood in
stool, or oral ulceration. He had received oral Cefixime and Ofloxacin
prior to admission, without any remission of symptoms.
At the time of hospitalization, the child was febrile
and there was no jaundice or pallor. Liver and spleen were palpable 4 cm
and 2 cm below the costal margin, respectively. He also had generalized
lymphadenopathy. Hemoglobin (Hb) was 12.2g/dL, total leukocyte count
(TLC) 9.2×10 9/L
with neutrophil 69% and lymphocyte 24%. C-reactive protein (CRP) was
37.3 mg/L (Normal <5 mg/L). Blood and urine cultures were sterile. Liver
and renal function tests were normal.Widal and Mantoux test were also
negative. Ultrasonography (USG) of abdomen revealed few iso- and
hyper-echoic lesions in the right lobe. Lesion in segment IV measured
17×28 mm, and lesion in segment VI showed liquefactive component (WebFig.
1a). Fever and pain persisted even after treatment with Ceftriaxone,
Cloxacillin and Metronidazole for 14 days. Repeat USG after two weeks
showed lesions to be of same size with decreased echogenicity,
suggestive of increased liquefaction. Computed tomography (CT) scan
revealed multiple thick walled, septated, hypodense space occupying
lesions with peripheral and septal post-contrast enhancement. They were
close to the biliary tree, and were communicating with each other within
the liver (WebFig. 1b). CT-guided percutaneous aspirate
did not show any organism on Gram stain and Ziehl Neelsen stain;
bacterial and fungal cultures were also negative.
Brucella IgM antibodies were reactive – 27.8
(significant >12). Brucella serum agglutination test titer was 1:320. On
questioning, the parents revealed a history of the child consuming
unpasteurized cow milk. HIV serology, CD4:CD8 ratio and chronic
granulomatous screening were normal. Serum Immunoglobulin A (IgA) level
was 22 mg/100 mL (Normal 34-305 mg/100 mL). Serum levels of IgG, IgM and
IgE were normal. We changed the antibiotics to Trimethoprim-
Sulphamethoxazole (TMP-SMZ), Doxycycline and Amikacin. Gradually over
the next three days, the fever reduced in intensity, and abdominal pain
subsided. He was finally discharged after 10 days of this treatement.
At two weeks follow up, he developed deranged liver
enzymes for which Doxycycline was discontinued. TMP-SMZ was continued
for a total duration of 6 weeks. Largest lesion at 3 months follow-up
measured 12×10 mm. Serial USG done over the next 6 months showed a
gradual reduction in size of the abscess, with no further recurrence of
fever or abdominal pain.
Discussion
Brucellosis is transmitted to humans following intake
of unpasteurized milk or milk products of infected animals. It may also
spread following handling of infected tissue samples [1]. Brucella
melitensis, B. suis, B. canis and B. abortus are the
commonest species known to be pathogenic to human beings [1-3]. Though
we were not able to isolate brucella in this child, our diagnosis was
based on history (consumption of unpasteurized milk), clinical findings
and positive brucella serology. Isolation of brucella by blood culture
is difficult in long-standing cases. It is a slow-growing organism,
often needing 7 to 30 days for incubation with random blind subcultures
[4,5]. Serology is the most useful diagnostic test in the absence of
bacteriological confirmation, although false-positive reactions can
occur.
Hepatic involvement in brucellosis is usually
characterized by elevated transaminases [1,6]. Liver abscesses have been
rarely reported, especially in children [1,2]. Patients infected with
brucella may develop liver abscess as a result of caseation necrosis of
granulomatous tissue. It may manifest acutely or may remain latent for a
long period to manifest later [7].USG and CT scan used to confirm the
diagnosis usually reveal a hypoechoic or hypodense area. Calcifications
are classical in brucella liver abscess, which were not documented in
this child [7,8].
Despite being susceptible to multiple antibiotics,
treatment failure is often seen because of the intracellular location of
brucella, delayed diagnosis, and inappropriate dosage and duration of
antibiotics. Combination of medical therapy with surgical drainage/percutaneous
aspiration is the treatment of choice for localized abscesses caused by
brucella [1,9]. TMP-SMZ for 4-6 weeks in combination with Rifampicin or
aminoglycosides for 7-14 days or Doxycycline have been documented to
reduce the relapse rates considerably [1,2,6,7,9]. As patients with
complicated brucellosis often need prolonged course of antibiotics, we
treated this child with three drugs and later discharged him on two oral
antibiotics once clinical improvement was noted.
This child was also diagnosed with IgA deficiency,
which happens to be the most common primary immunodeficiency in
children. Infections of the respiratory tract (with Haemophilus
influenzae and Streptococcus pneumoniae) and gastrointestinal
(GI) tract are common. Secretory IgA has an essential role in intestinal
homeostasis as most of the commensal bacteria are located in the GI
tract [10]. We could not find any earlier reported occurrence of liver
abscess with brucella in a patient with selective IgA deficiency.
Non-response to conventional antibiotics in a case of
liver abscess should prompt us to consider other atypical organisms such
as brucella. Prolonged treatment with combination antibiotics may reduce
the requirement of surgery and result in complete recovery. Patients
with deep seated abscess with atypical organisms should also be screened
for associated immunodeficiency.
Contributions: DM,PP, RK: were involved in
interpretation of investigations, diagnosis and patient management; DM:
drafted the manuscript which was verified and approved by PP and RK.
Funding: None; Competing Interest:
None stated.
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