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Acute acalculous cholecystitis in paediatrics. A not so rare pathology

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1130-0108/2014/106/7/486-489
Revista Española de Enfermedades Digestivas Rev Esp Enferm Dig (Madrid
Copyright © 2014 Arán Ediciones, S. L. Vol. 106, N.º 7, pp. 486-489, 2014

CLINICAL NOTES

Acute alithiasic cholecystitis: A not so rare disease

Javier Blasco-Alonso1, Eloísa Santiago-García-Caro2, Raquel Gil-Gómez3, Carolina Jiménez-Alcántara2,
Pilar Sánchez-Yáñez2 and Guillermo Milano-Manso3
1
Department of Gastroenterology and Children Nutrition, UGC Pediatrics. 2UGC Pediatrics. 3UGC Critical Care
and Children Urgencies. Hospital Materno-Infantil. Hospital Regional Universitario Carlos Haya. Málaga, Spain

ABSTRACT INTRODUCTION
Introduction: Acute acalculous cholecystitis (AAC) occurs Acute acalculous cholecystitis (AAC) represents an
more frequently in critically ill patients, in the immediate
postoperative period, after trauma or extensive burns. It has a high emerging disease (1) that can represent 30 to 50 % of cho-
rate of morbidity and mortality. Ischemia, infection and vesicular lecystitis in children (2). It usually occurs within system-
stasis are determinants in its pathogenesis. ic bacterial (Gram-negative or anaerobic) or viral (EBV,
Material and method: Retrospective study including all cases hepatotropic virus...) infections (3), as well as secondary
of AAC diagnosed in our pediatric intensive care unit between
January 1997 and December 2012.
to dehydration or prolonged parenteral nutrition, appearing
Results: We included 7 patients, all associated with viral or rarely as a complication of severe medical-surgical diseas-
bacterial infection. All of them suffered from abdominal pain, mainly es (multiple trauma, burns, postoperative...), more com-
localized in the right upper quadrant, jaundice and dark urine. mon etiologies in adult. Regarding clinical manifestations
Abdominal ultrasound showed thickening and hypervascularity (4,5), it is required a high suspicion, since the onset of
of the gallbladder wall in all cases. The outcome was satisfactory
without surgery in all patients.
unexplained fever, jaundice or vague abdominal discom-
Conclusions: The clinical presentation is oligosymptomatic fort in a critically ill patient, often intubated and sedated,
within severe systemic diseases. The AAC should be suspected in may be the only track (6,7).
the appearance of any abdominal pain with jaundice/dark urine and AAC handling depends on the time of diagnosis, and
hypertransaminasemia in patients suffering from critical or serious thus in early stages of the disease exclusive medical treat-
infections.
ment may be sufficient (8), reserving cholecystectomy for
Key words: Acute acalculous cholecystitis. Ultrasound. patients with vesicular gangrene or perforation (9). Mor-
Childhood. Critical Care. Hypertransaminasemia. Jaundice. tality depends on the underlying medical condition, rang-
ing from 90 % in critically ill patients, up to 10 % in the
outpatient (10), but always greater than 1 % corresponding
to gallstone cholecystitis.
Our goal is to review the cases of AAC in our pediatric
Blasco-Alonso J, Santiago-García-Caro E, Gil-Gómez R, Jiménez- center and compare with the existing literature.
Alcántara C, Sánchez-Yáñez P, Milano-Manso G. Acute alithi-
asic cholecystitis: A not so rare disease. Rev Esp Enferm Dig
2014;106:486-489.
CASE REPORTS

Retrospective review of hospital records of pediatric

patients under 14 years diagnosed with AAC in our hos-
pital from January 2001 to December 2013. We collected
Received: 15-11-2013 epidemiological (age and sex), clinical (underlying disease
Accepted: 24-03-2014
and clinical characteristics), diagnostic (special emphasis
Correspondence: Javier Blasco Alonso. Department of Gastroenterology on ultrasound) and therapeutic (drugs employed, compli-
and Children Nutrition, UGC Pediatrics. Hospital Materno-Infantil. Hos- cations) data after informed consent was obtained.
pital Regional Universitario Carlos Haya. Avda. Carlos Haya, s/n. 29010 The AAC diagnosis was established by clinical suspi-
Málaga, Spain
e-mail: [email protected] cion (pain in the upper right quadrant, fever, leukocytosis,
Vol. 106, N.º 7, 2014 ACUTE ALITHIASIC CHOLECYSTITIS: A NOT SO RARE DISEASE 487

normal liver function) and confirmed by abdominal ultra- and/or dark urine. Blood tests revealed moderate hyper-
sound (92 % sensitivity and 96 % specificity), not being transaminasemia, always greater than three times normal,
necessary to perform any other imaging test. Ultrasound in all patients (maximum levels 1286 U/l AST and 2798
studies reviewed by two radiologists in all children who ALT, with consistently high GGT), with hyperbilirubin-
met clinical criteria. Ultrasonographic diagnostic criteria emia in 4 cases. The ultrasound showed wall thickening
(8,11) were divided into major (gallbladder wall thickening and hypervascularity in the absence of lithiasis in all cas-
over 3.5 mm, gallbladder distended to at least 5 cm in the es without pericholecystic fluid. Treatment was conserva-
longitudinal dimension and no evidence of ascites or hypo- tive with fluid therapy, parenteral nutrition, analgesia and
albuminemia, the presence of pericholecystic fluid or sub- antibiotics, adding vitamin K, lactulose and ursodeoxy-
serosal edema, intramural gas) and minor criteria (presence cholic acid in the patient with ALL and in neonates. The
of echogenic bile (sludge), distension greater than 8 cm in most used combination of antibiotics was third generation
the longitudinal or 5 cm in the transverse dimension). A cephalosporin and antianaerobe agents (mainly metroni-
diagnosis was considered positive if it included either a dazole). The evolution was satisfactory without surgery
minimum of two major criteria or one major and two minor in all patients.
criteria, in the appropriate clinical setting.
Seven patients were included (Table I): 5 of them were
women, ages distributed in two peaks (4 teenagers between DISCUSSION
11 and 13 years and 3 infants between 2 and 24 months),
with the first four cases diagnosed between 2001 and 2005 As for the typical age of presentation, according to Ima-
and the last three ones between 2011 and 2013. Five of moglu (8), it predominates at school age (mean 7.8 years),
them were previously healthy children, one suffered from not fully coinciding in our series (median 11.5 years),
acute lymphoblastic leukemia (ALL) and received a bone although cases have also been reported in neonates, infants
marrow transplant with suspected graft versus hostage and preschool (in our work appearing in two infants, aged
disease (GVHD) and, the other case was a newborn with 2 and 12 months).
sepsis/meningitis by E. coli. The predisposing factor was a AAC has been associated with intercurrent infections,
viral or bacterial infection (EBV, HAV, E. coli, Micrococ- metabolic disorders, vascular problems, burns, injuries
cus, B. melitensis, P. pneumoniae) in 6 patients, highlight- and malignancies in children. Our patients presented
ing positive urine culture only E. faecalis in the seventh within systemic diseases of varying severity (5 of them
case. All of them started with mild abdominal pain, right stayed in the pediatric intensive care unit at some point),
upper quadrant referral, and fever, and only the first four as published in the literature (9), 5 of them with different
cases with cholestatic syndrome presented with jaundice blood cultures and/or seropositive to different infectious

Table I. Patients characteristics

Diagnosis at Length of AST / ALT / TB / DB Leukocytes Vasoactive
Case Age Sex Infection PRISM (%) Treatment
PICU admission stay (days) GGT (U/L) (mg/dl) / CRP (mg/L) drugs
134 / 215 /
1 13 years F Clinical sepsis EBV 6 3.8 / 2.9 14800 / 5.1 1.3 Yes Analgesia / Vit K
223
1286 / 2798 / Analgesia / Vit K /
2 11 years F Clinical sepsis HAV 4 12 / 10.4 4020 / 13 1.2 No
678 UDCA / PN / Ab
Sepsis- 1170 / 895 / Analgesia / Vit K
3 2 month M E. coli 12 15.5 / 11.7 4030 / 54 18.3 No
meningitis 687 PN / Ab / UDCA
ALL / TPH / Micrococcus 886 / 918 / Analgesia / Vit K /
4 12 years M 11 16 / 11 9450 / 8.1 3.1 Yes
GVHD sp 342 Ab / UDCA / PN
Systemic 192 / 179 /
5 12 years F B. melitensis 4 0.3 / 0.1 4690 / 81 1.7 No Analgesia / Ab
Brucellosis 237
Hemolytic-
P. 234 /258 / Analgesia / Ab /
6 2 years F uremic 56 5.1 / 2.3 12346 / 73 3.1 Yes
pneumoniae 356 UDCA / PN
syndrome
228 / 228 / 14840 / Analgesia / Ab /
7 1 year F Clinical sepsis E. faecalis 6 0.2 / 0.1 1.2 Yes
909 125 UDCA / Vit K
PICU: Pediatric intensive care unit; F: Female; M: Male; ALL: Acute linfoblastic leukemia; TPH: Hematopoietic stem cell transplantation; GVHD: Graft versus hostage disease;
TB/DB: Total and direct bilirubin; CRP: C reactive protein; M: Mayor criteria; m: Minor criteria; PN: Parenteral nutrition; Ab: Antibiotics. UDCA: Ursodeoxicolic acid. PRISM:
Pediatric risk of mortality.

Rev Esp Enferm Dig 2014; 106 (7): 486-489

488 J. BLASCO-ALONSO ET AL. Rev Esp Enferm Dig (Madrid)

agents, as in the seventh patient, despite being a positive analgesia and use of antibiotics active on Gram negative,
urine culture to E. faecalis, the clinical picture was most anaerobes and Enterococci, which in turn reach therapeutic
consistent with a viral hepatitis untested by serology. This concentrations in the biliary tract. So, all of our patients
demonstrates that most AAC in children occur during the were treated conservatively, adding vitamin K, lactulose
course of serious infectious diseases (10), 10 % of which and ursodeoxycholic acid in the patient with ALL and in
are in the form of sepsis, as published by Shu-Ching Huang neonates who attended E. coli sepsis/meningitis.
in 2011 (11), and which is also reflected in our work with According to the data of our study and the literature
the 2 month-old infant suffering from AAC within E. coli reviewed, we draw several conclusions: Although the AAC
sepsis/meningitis. Furthermore, Shu-Ching Huang also is a rare entity in children, it must be considered by the
said that less than 10 % of the AAC are produced in the pediatrician among the causes of abdominal pain, espe-
course of neoplastic diseases, as our 12 year old patient cially in critically ill children that are often intubated and
with a history of ALL and transplanted with suspected sedated, with detection of vague abdominal discomfort,
GVHD, treated cyclosporine, which developed Micrococ- fever and jaundice besides, and where the source of infec-
cus AAC. Moreover, the latter patient is the first pediatric tion is not entirely clear. Ultrasound is the most reliable
case reported in the literature by Micrococcus CAA, after method for diagnosis (Fig. 1). In children, conservative
a thorough review using MEDLINE + EMBASE (key treatment is effective in solving this disease, although it
words: AND Micrococcus cholecystitis), which showed does require close clinical, analytical and ultrasound mon-
only one previous case of acute gallstone cholecystitis by itoring, able to detect complications.
Kocuria kristinae, belonging to genus Micrococcus, a 56
year old Chinese woman. It must be also pointed out the
case of the AAC as a complication of systemic brucellosis, REFERENCES
since only 21 cases have been described to date after a
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entity. Ann Surg 1982;195:131-6.
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the second pediatric patient (12). titis in children. J Pediatr Surg 1996;31:127-30.
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in critically ill patients, or make it difficult to classify ecystitis. J Clin Gastroenterol 2003;36:166-9.
abdominal pain. Cholestasis is another common sign in 6. Trowbridge RL, Rutkowski NK, Shojania KG. Does this patient have
patients with AAC (5/7 of our patients) and, together with acute cholecystitis? JAMA 2003;289:80-6.
the hypertransaminasemia not justified for other reasons 7. Yokoe M, Takada T, Strasberg SM, Solomkin JS, Mayumi T, Gomi H,
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of CAA (8), besides being a non-invasive and having Acute acalculous cholecystitis in children: Diagnosis and treatment. J
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cholecystectomy, this technique is not free of morbidity genemia predict a fatal outcome in childhood acute acalculous chol-
and mortality, ranging between 9 and 66 %, and it is now ecystitis. JPGN 2011;53: 548-52.
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Rev Esp Enferm Dig 2014; 106 (7): 486-489

Vol. 106, N.º 7, 2014 ACUTE ALITHIASIC CHOLECYSTITIS: A NOT SO RARE DISEASE 489

Patient with data of systemic inflammatory response

Fever, abdominal pain, cholestasis and/or hypertransaminasemia

Yes No

Abdominal Doppler-Ecography

Sonographic criteria (two mayor criteria

Clinical and analytical evaluation
or one mayor and two minor)

– Mayor criteria:
Gallbladder wall thickening over 3.5 mm
Gallbladder distended to at least 5 cm
No evidence of ascites or hypoalbuminemia
Pericholecystic fluid or subserosal edema
No
Intramural gas
– Minor criteria:
Presence of echogenic bile (sludge)
Longitudinal distension > 8 cm
Transversal distensión > 5 cm

Yes

Conservative treatment: Fluid, parenteral nutri- Favourable evolution

tion, analgesia and antibiotics, together with Cholecystostomy
vitamin K, lactulose, ursodeoxycolic acid… Unfavourable evolution
Cholecystostomy

Fig. 1. Diagnostic-therapeutic algorithm.

Rev Esp Enferm Dig 2014; 106 (7): 486-489

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