# Leclercia adecarboxylata Cholecystitis with Septic Shock in Immunocompetent Patient

Authors: Nooraldin Merza; John Lung; Ahmed Taha; Ahmed Qasim; Jill Frost; Tarek Naguib
Journal: Case Reports in Critical Care
 (2019)

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## Abstract

L. adecarboxylatais a Gram-negative rod previously namedEscherichia adecarboxylata, isolated as normal flora in the gut of animals including human stool. Most reported cases refer to immunocompromised patients with polymicrobial infections and water environments. Here we present a case of 51-year-old immunocompetent female presented with nausea, vomiting, malaise, and subjective fever for few days. On examination, she was drowsy but arousable and oriented to person, place, time, and situation. Her abdomen was tender globally and more tender in the epigastric area. Vitals showed a temperature of 37°C, pulse of 110 beats/min, blood pressure of 75/50 mmHg, and oxygen saturation of 91% on room air. An HIV panel and hepatitis panel were negative. Liver and gallbladder ultrasound was performed, revealing multiple nonmobile stones with shadowing noted within the gallbladder sac, a thickened gallbladder wall, and a moderate amount of pericholecystic fluid. Broad spectrum antibiotics, crystalloid fluids, and vasopressors were initiated. A few hours after admission she developed respiratory failure for which she underwent endotracheal intubation. An ultrasound guided gallbladder drain was performed. Culture of the biliary fluid yielded pure growth of pan-sensitiveL. adecarboxylata; antibiotics were narrowed accordingly. The patient was on the maximum doses of vasopressin, norepinephrine, and epinephrine with a blood pressure of 75/45 and a mean arterial pressure of 51. She passed away on the fourth day of admission.


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## Body

## 1. Background

Leclercia adecarboxylata is a unique Enterobacteriaceae member frequently isolated from water with infrequent case reports in the literature. Originally described by Leclerc in 1962 asEscherichia adecarboxylata, these facultative aerobic, Gram-negative bacilli were reclassified by the development of more accurate identification methods as a member of the Enterobacteriaceae family [1]. This bacterium is usually associated with polymicrobial infection in immunocompromised individuals, although there are recent observations of finding this microorganism in healthy individual. We hereby report a unique case of cholecystitis leading to septic shock caused byLeclercia adecarboxylata in an immunocompetent individual without significant water exposure.


## 2. Case Report

A 51-year-old female presented to the emergency department with nausea, vomiting, malaise, and subjective fever for few days. She reported difficulty with ambulation as well as right hip and back pain for several months, with nonsignificant past medical or surgical history. She denied alcohol, drug, or tobacco use. She also denied any recent travel, sick contacts, or exposure to standing water. On physical examination, she was drowsy but arousable and oriented to person, place, time, and situation. Her abdomen was tender globally and more tender in the epigastric area. Vitals showed a temperature of 37°C, pulse of 110 beats/min, blood pressure of 75/50 mmHg, and oxygen saturation of 91% on room air. Labs are shown in Table1. An HIV panel, hepatitis panel, ceruloplasmin, antinuclear (ANA) panel, smooth muscle antibody, anti-centromere antibody, antichromatin antibody, anti-DNA antibody, anti-Jo-1 antibody, anti-Scl-70 antibody, anti-SSA antibody, and anti-SSB antibody were all negative. Alpha-1 antitrypsin was slightly above normal limits at 204 mg/dl (reference 90-200). Imaging revealed a normal chest X-ray. Electrocardiogram showed sinus tachycardia with no ischemic changes. A CT scan of the abdomen showed a gallbladder wall thickening and multiple gallstones. To exclude cholecystitis, a liver and gallbladder ultrasound. (Figure 1) was performed, revealing multiple nonmobile stones with shadowing noted within the gallbladder sac, a thickened gallbladder wall, and a moderate amount of pericholecystic fluid.Table 1
The patient’s lab on days 1, 2, 3, and 4.
Day 1 Day 2 Day 3 Day 4 WBC(x10e3/mcL) 9.1 7.6 12.1 13.8  Neutrophils (x103/mcL) 6.6 5.6 10.1 12.4  Hemoglobin (gm/dL) 11.4 8.2 8.2 7.7  Platelets (x10e3/mcL) 136 114 45 54  INR 1.52 2.2 2.7 3.0  Fibrinogen (mg/dL)  172 61 75  D-Dimer (mg/L) >35 >35 >35 >35  LDH(units/L) >4000 7898 9738  Troponin (ng/mL) 0.088  Glucose (mg/dL) 45 71 117 220  Na (mmol/L) 134 144 145 139  K (mmol/L) 6.9 6.4 4.5 3.9  BUN(mg/dL) 73 63 40 14  Creatinine (mg/dL) 2.4 2.4 2.1 1.2  CO2(mmol/L) 8 4 11 22  Anion Gap (mmol/L) 22 29 30 13  Lactate (mmol/L) 11.5 17.5 20 10  AST(units/L) 1650 10623 17523 5846  ALT(units/L) 553 1953 2561 1823  ALP(units/L) 260 257 333 409  Bilirubin 0.98 2.1 2.6 4.8  Albumin 2.5 3 2.5 2.2Figure 1
Ultrasound of gallbladder showing inflamed gallbladder with multiple nonmobile stones with shadowing in the gallbladder body. The gallbladder wall is thickened to 0.99 cm. A moderate amount of pericholecystic fluid is identified.Broad spectrum antibiotics (IV vancomycin and piperacillin/tazobactam), crystalloid fluids, and vasopressors were initiated. A few hours after admission she developed respiratory failure for which she underwent endotracheal intubation. Due to her shock state she was not a candidate for surgery. An ultrasound guided gallbladder drain (Figure2) was performed. Culture of the biliary fluid yielded pure heavy growth of pan-sensitiveL. adecarboxylata; antibiotics were narrowed accordingly to IV meropenem.Figure 2
IR-guided cholecystostomy tube placement and drainage and irrigation of the gallbladder with 60 cc of black bile aspiration.On the second day of admission to the intensive care unit the patient developed signs of disseminated intravascular coagulopathy from septic shock and acute liver failure with profound lactic acidosis and deteriorated rapidly. The patient developed skin mottling of all four extremities due to limb ischemia likely due to peripheral artery vasoconstriction secondary to shock and vasopressor use. The patient had no Doppler signals in the bilateral radial, ulnar, dorsalis pedis, and posterior tibial arteries. Indium-111 tagged WBC scan was performed (Figure3) to make sure we have not missed another source of infection. This showed diffuse pulmonary uptake which we felt was due to acute respiratory distress syndrome and there was no abscess. The patient received disseminated intravascular coagulation (DIC) treatment including multiple packed red blood cells (PRBC), cryoprecipitate and fresh frozen plasma (FFP) transfusions, and treatment for septic shock including antibiotics and vasopressors.Figure 3
Indium-111 white blood cell scan showing no localization of abscess and atypical diffuse pulmonary uptake of white blood cells, which can be associated with an opportunistic infection, pulmonary drug toxicity, and sepsis.The patient also received hemodialysis due to acute kidney injury secondary to septic shock. Consults with gastroenterology and general surgery resulted in recommendations of no surgical intervention because of the critical condition of the patient with intubation, mechanical ventilation, hemodialysis, and vasopressors. The patient developed pulseless electrical activity (PEA) cardiac arrest but recovered after one round of cardiopulmonary resuscitation and 1 dose of epinephrine. The patient was on the maximum doses of vasopressin, norepinephrine, and epinephrine with a blood pressure of 75/45 and a mean arterial pressure of 51.The patient developed another PEA cardiac arrest 11 hours later and could not be resuscitated despite aggressive resuscitation measures that followed advanced cardiac life support protocol. She passed away on the fourth day of admission.


## 3. Discussion

L. adecarboxylatais a motile, Gram-negative rod previously namedEscherichia adecarboxylata, isolated as normal flora in the gut of animals including human stool. Most reported cases refer to immunocompromised patients which gives a clear suggestion of the exclusivity ofL. adecarboxylataas an opportunistic pathogen associated with polymicrobial infections and water environments, particularly as a contaminant in polluted water [2, 3]. The paucity of published case reports of human infections with this bacteria may reflect misdiagnosis, as the organism shares many biochemical features withE. coli, rather than a true infrequency of human infection [4].L. adecarboxylatacauses infections at various organs including pharyngeal abscess, pneumonia, cholecystitis, gluteal abscess, and cellulitis.There are at least 74 cases reported in English-language journals since the first case report ofL. adecarboxylatain 1991, including 19 cases in immunocompetent patients and two cases in patients with cholecystitis, one chronic and one acute (Tables 2 and 3) [2, 4–22].Table 2
Clinical case reports ofL. adecarboxylata infections in immunocompetent patients.
Case Age Gender Infection site and clinical information Co-infection Outcome Identification method Temesgen et al. 1997 35 M right foot would culture, s/p crush injury Enterobacter cloacae, Citrobacter freundii, Enterococcus species, Klebsiella pneumoniae, Stenotrophomonas maltophilia, and Corynebacterium, Acremonium, Penicillium, Mucor, Geotrichum Recovery Biochemical testing  Temesgen et al. 1997 23 M left toe wound culture, s/p nail puncture Acinetobacter calcoaceticus, Enterobacter agglomerans Recovery Biochemical testing  Greco et al. 2001 75 M wound culture in right hand Staphylococcus epidermidis Recovery Not mentioned  Pérez-Moreno et al. 2003 60 M synovial fluid, infection arthritis None Recovery Biochemical testing  Hess et al. 2008 40 F right foot wound culture, s/p superficial incision and swimming in pool None Recovery Biochemical testing  Dalamaga et al. 2009 53 M left arm wound culture, s/p chemical injury None Recovery Biochemical testing  Mbamalu and Macariola 2011 11mo M blood bacteremia None  Not mentioned  Bali et al. 2013 32 M peritonsillar and lateral pharyngeal abscess None Recovery Not mentioned  Zapor et al. 2013 25 M pus and necrotic tissue in right buttock, s/p wartime IED explosion None Recovery 16s ribosomal sequencing with 16s rRNA gene  Haji et al. 2014 70 M blood culture, infectious arthritis None Recovery Not mentioned  Keren et al. 2014 46 M left foot soft tissue infection, s/p laceration while surfing Enterobacter cloacae Recovery after 2 weeks VITEK-2 automated microbial- identification system  Anuradha 2014 50 M gluteal abscess None Recovery MicroScan autoSCAN-4 automated microbial identification system  Anuradha 2014 31 F vaginal swab, vaginitis None Recovery after 10 days MicroScan autoSCAN-4 automated microbial identification system  Grantham et al. 2015 9 F left lower extremity soft tissue infection s/p penetrating injury None Recovery after 2 months Not mentioned  Hurley et al. 2015 2 M right thumb soft tissue infection s/p laceration None Recovery VITEK automated microbial identification system  Prakash et al. 2015 32 F tracheal aspirate, hospital-acquired pneumonia None Recovery after 1 week VITEK automated microbial identification system  Allawh, Camp 2015 26 F fingernail soft tissue infection s/p trauma to nail Staphylococcus Not symptomatic after 2 weeks Not mentioned  Riazzo et al. 2017 30 M Subcutaneous trauma injury to foot Klebsiella pneumoniae, Pseudomonas Discharge after 3 months Microscan Walkaway, confirmed by MALDI-TOF  Capretta et al. 2018 9 M right leg soft issue infection, cellulitis s/p fall N/A Recovery after 3 weeks Not mentionedTable 3
Clinical case reports ofL. adecarboxylatainfections in patients with cholecystitis who were also immunocompromised.
Case Age Gender Infection site and clinical information Co-infection Outcome de Baere et al. 2001 78 F Chronic cholecystitis Enterococcus Recovery  Jover-Sáenz et al. 2008 81 F bile, cholecystitis None Recovery after 1 monthThere is one case in the literature of an 81-year-old female (Table3) with a past medical history of metabolic syndrome and chronic atrial fibrillation who presented with acute cholecystitis and had a positiveL. adecarboxylatabiliary fluid culture [10]. The patient in that case report had risk factors for a compromised immune system including old age, cardiovascular risk factors, and metabolic syndrome, while our patient was immunocompetent and did not have any known source for contractingL. adecarboxylata. Our patient tested negative for HIV and hepatitis B and C virus. The team ordered a tagged WBC scan to make sure we are not missing another hidden infection.L. adecarboxylatais not commonly associated with sepsis and rapid deterioration requiring mechanical ventilation and resuscitative measures as in this case scenario. There is one case report of a patient who developedL. adecarboxylatasepsis, required intubation, and then died in the hospital. This patient had a viral infection prior to admission and then developed pneumonia due to a multidrug-resistantL. adecarboxylatainfection [23]. Our patient who developed septic shock had aL. adecarboxylatainfection was pan-sensitive to most of the tested antibiotics, with no immunocompromising risk factors. MostL. adecarboxylatainfections are sensitive to many antibiotic options and frequently respond to treatment without needing to switch antibiotics [24].Most of L. adecarboxylatastrains are naturally sensitive to tetracyclines, aminoglycosides, all but two β lactams, quinolones, folate pathway inhibitors, chloramphenicol, nitrofurantoin, and azithromycin. They are naturally resistant to penicillin G, oxacillin, erythromycin, roxithromycin, clarithromycin, ketolides, lincosamides, streptogramins, linezolid, glycopeptides, rifampicin, fusidic acid, and fosfomycin, with only minor medium-dependent differences in susceptibility to most antibiotics [24, 25].However, multiantibiotic resistantL. adecarboxylatahas been reported in the literature [21, 26], illustrating the need to continue to practice good antibiotic stewardship to prevent development of resistant strains.The uniqueness of this case could be summarized by the presentation of septic shock withL. adecarboxylatain an immunocompetent state. There are only a few reported cases ofL. adecarboxylatainfection in immunocompetent patients [5]. BecauseL. adecarboxylatais typically found in polymicrobial infections, it is rare to encounter the growth of pure cultures as in this case. There are few reported cases of pure cultures in immunocompetent patients, but not with this severity [5, 22].L. adecarboxylatacases in immunocompetent patients are associated with exposed wound infections and contact with water environments [2], while our patient did not have any open wound and lived in a dry, flat area with no open water sources.


## 4. Conclusion

L. adecarboxylatais a rare Gram-negative rod which often infects immunocompromised hosts. We report a case of acute cholecystitis caused byL. adecarboxylataleading to profound septic shock with DIC and liver failure that did not respond to aggressive treatment measures. Our case illustrates an opposite picture of what has long been known as an opportunistic organism and a cause of mild infections. We hope by presenting this case we contribute to the current knowledge about this organism and perhaps increase the reporting and identification of the organism as a potential invasive pathogen.



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