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Indian Pediatr 2019;56:
504-505 |
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Amebic Liver Abscess and Kawasaki Disease
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Vijay Viswanathan 1,
Ishita Singh1 and
S Sane2
From 1Pediatric Rheumatology Clinic and 2Department
of Pediatrics, Jupiter Hospital, Thane, India.
Correspondence to: Dr Vijay Viswanathan, Pediatric Rheumatologist,
Jupiter Hospital,
Thane 400 601, India.
Email: [email protected]
Received: August 31, 2018;
Initial review: January 04, 2019;
Accepted: April 20, 2019.
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Background: Co-occurrence of
amebic hepatitis and Kawasaki disease has not been reported previously.
Case characteristics: We describe two children (aged 4 y and 5 y)
with Kawasaki disease and coexisting liver abscess. They were treated
with intravenous immunoglobulins with/without percutaneous drainage in
combination with amebicidal agents. Outcome: Both the children
were completely cured of the amebic hepatitis, and had normalization and
regression of coronaries at follow-up. Message: We report the
co-existence of amebic hepatitis with Kawasaki disease.
Keywords: Fever of unknown
origin, Superantigen, Vasculitis.
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K
awasaki disease (KD) results from an interaction
of a host of infections (superantigen theory), or a canonical response
to a conventional antigen with genetic predisposition [1,2]. The search
for the elusive superantigen still continues with streptococcal,
staphylococcal, and more recently, candidal elements being thought of as
possible triggers [3,4]. Coexisting infections like dengue have been
reported [5,6]. We report 2 cases of co-occurrence of amebic hepatitis
with KD.
Case Report
Case I
A 4-year-old boy was referred with persistent fever
for 18 days and abdominal pain. Ultrasonography abdomen done on day 10
and day 14 outside had revealed an abscess (4 cm × 4.8 cm × 3.8 cm)
involving the right hepatic lobe. He had received multiple antibiotics
and amebicidal drugs with persistent pyrexia. Examination revealed
significant mucositis with erythema and fissuring, pedal edema, and
bulbar non-purulent conjunctivitis with perilimbal sparing. No
lymphadeno-pathy or rash was reported. Tender hepatomegaly was present.
Laboratory findings on admission (day 18) revealed anemia (Hb 7.6 g/dL),
leucocytosis (Total counts 16.8 × 10 9/L,
(P44%, L36%), thrombocytosis 1070×109/L),
elevated acute phase reactants (ESR 112 mm/h and CRP 49.5 mg/L) and
hypoalbuminemia (2.5 g/dL). Repeat blood, urine and throat cultures were
sterile. Repeat USG was similar to previous scans with no liquefaction.
With a suspicion of a coexisting incomplete KD, a transthoracic 2D
echocardiogram was done which showed significantly dilated coronary
arteries with a proximal right coronary artery saccular aneurysm (RCA Z
score 4.31), left main coronary (LMCA Z Score 3.22), left anterior
descending artery (LAD-Z score 2.55). Indirect hemagglutination (IHA)
test for IgG Ameba antibody was positive. With no response to
antibiotics and the positive clinical and cardiac findings, IVIG was
administered at 2 g/kg, with complete resolution of pyrexia within 36
hours. Child was discharged on anti amebicidals and low dose aspirin. He
returned after a week with desquamation of finger tips confirming our
diagnosis. USG improvement and normalization of echo-cardiographic
findings were noticed over 10 weeks and 6 months, respectively.
Case II
A 5-year-old girl presented with a history of
remittent fever of 7 days, redness of lips and swelling on left side of
neck. A history of a macular rash and non-purulent conjunctival
congestion on days 3 and 4, respectively was elicited. Clinical
examination revealed mucositis with erythema and fissuring, pedal edema,
and bulbar non-purulent conjunctivitis with perilimbal sparing. An
enlarged left anterior cervical lymph node were also (>1.5 cm), and mild
pedal edema were also noted. Her vitals were stable with mild
tachycardia (Heart rate 102/min) and respiratory rate of 30/min with
normal blood pressure (94/60 mm Hg). Laboratory findings on admission
revealed anemia (Hb 7.6 g/dL), leucocytosis (total counts 21.4 × 10 9/L,
P 83%, severe thrombocytosis (1071× 109/L),
elevated acute phase reactants (ESR 90 mm/h and CRP 380 mg/L) and
hypoalbuminemia (1.5 g/dL). With negative cultures, a trans-thoracic 2D
echocardiography showed LMCA- Z score 0.06, LAD -Z score of 1.91 and RCA
-Z score of 5.97. IVIG was administered at 2 g/kg with a diagnosis of
KD. Post IVIG, with persistent but spaced fever spikes, she developed
increasing respiratory distress and tender hepatomegaly. Ultrasonography
showed a 7.4 × 6.5 × 6.4 cm liquefied abscess with mild pleural
effusion. IHA for IgG ameba antibody was positive. Percutaneous drainage
revealed 200 mL fluid with an ‘anchovy sauce’ like appearance, and
administration of parenteral amebicidal drugs led to complete resolution
of symptoms over the next 3 days. She was discharged on low dose
aspirin, and subsequent desquamation at the end of a week confirmed the
diagnosis of KD. Subsequently she showed a steady regression of her
coronary artery abnormalities over 3 months, and was continued on low
dose aspirin with regular echocardiographic monitoring.
Discussion
Multiple infectious agents have been implicated as
triggring agents for KD, including viruses, bacteria, rickettsiae and
even candidal agents [1]. Adding to the list of coexisting infections
with KD, we describe the presence of amebic hepatitis through this
report. In the first child, a lack of clinical response along with a
sequential evolution of clinical signs suggestive of KD prompted us to
confirm the diagnosis with 2 D echocardiogram. The second child had
classic features of KD from the beginning, diagnosis was confirmed by
echocardiography, but child had only a partial response to IVIG
(IVIG-resistant). New onset abdominal pain and respiratory distress
prompted a USG revealing the liver abscess. Complete resolution of
symptoms was achieved with drainage of the abscess and addition of
amebicidals. Both children had evidence of severe inflammation in the
form of increased acute phase markers and acute hypoalbuminemia.
Serological tests (IHA) confirmed the diagnosis of amebic hepatitis in
both of them.
A proven presence of an infection should not dissuade
the possibility of a co-existing inflammatory condition like KD, if the
response to therapy is inadequate or in the presence of suggestive
signs. Whether amebiasis was a coexisting infection with inflammation
(KD) causing a subclinical response or the potential organism triggering
the development of KD is debatable. However, these cases strengthen the
theory regarding the possible role of microorganisms with a disordered
innate immune system as a possible etiology. While dealing with a
suboptimal response to any infection, possibility of underlying
inflammatory conditions like KD needs to be considered in clinically
suggestive situations.
Contributors: The case was managed by VV, IS and
SS. VV and IS reviewed literature and wrote the draft manuscript which
was edited by SS. All three authors have seen and approved the
manuscript.
Funding: None; Competing interests: None
stated.
References
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2. Kawasaki T. Acute febrile mucocutaneous syndrome
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toes in children. Arerugi. 1967;16:178-222.
3. Takahashi K, Oharaseki T, Wakayama M, Yokouchi Y,
Naoe S, Murata H. Histopathological features of murine systemic
vasculitis caused by Candida albicans extract—an animal model of
Kawasaki disease. Inflamm Res. 2004; 53:72-7.
4. Ohno N. Murine model of Kawasaki disease induced
by mannoprotein-beta-glucan complex, CAWS, obtained from Candida
albicans. Jpn J Infect Dis. 2004;57:S9-10.
5. Agarwal E, Shah D, Gupta P. Dengue fever
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