Skip to main content
  • Case Reports
  • Open access
  • Published:

Cholecystitis secondary to Salmonella typhi: a rare pathology with an unreported management option—a case report and literature review

Abstract

Background

This report describes the presentation and course of treatment for one of the youngest reported cases of empyema of the gallbladder. Given the rare occurrence of this disease process, we elected to proceed with a systematic review of the literature. This is only the 7th case series discussing pediatric empyema of the gallbladder due to Salmonella typhi in the literature, and the second case ever reported in the USA.

Case presentation

We report a case of a previously healthy 13-month-old girl who presented with diffuse peritonitis and equivocal imaging studies. Diagnostic laparoscopy revealed purulent peritonitis. The gallbladder was distended with intraluminal pus. Laparoscopy was converted to laparotomy to facilitate exposure, and a cholecystostomy tube was placed. Cultures from the fluid were positive for Salmonella typhi. The patient received a 14-day course of intravenous Ceftriaxone followed by 14 days of oral amoxicillin and clavulanate. A cholangiogram performed 8 weeks after surgery confirmed normal biliary anatomy. The cholecystostomy tube was removed. The patient recovered uneventfully and is doing well over 9 months later.

Conclusion

There is no consensus on treatment with options reported ranging from medical management with antibiotic therapy to more invasive procedures such as cholecystostomy tube or cholecystectomy. Less invasive management options are an alternative for Salmonella cholecystitis.

Background

While the exact incidence of acute cholecystitis in children is unknown, both acute and chronic cholecystitis are less frequently seen in the pediatric population than in adults [1]. Chronic cholecystitis is more common than previously thought in the pediatric population, and data suggests upwards of 87% of all patients presenting with cholecystitis have the chronic type [2]. All forms of cholecystitis are most commonly the result of cholelithiasis, which is typically seen in patients who are obese, have hematologic disorders such as sickle cell disease, who require total parental nutrition, or have experienced periods of prolonged fasting [3].

Acalculous cholecystitis is proportionally more common in children than adults, occurring in about 50–70% of total pediatric cholecystitis cases as compared to 2–15% in adults [4]. Acute acalculous cholecystitis is typically observed in children with prolonged intensive care unit hospitalizations, burn patients, those with bone marrow transplantation or receiving chemotherapy, or in other chronically ill patients such as those with chronic granulomatous diseases or other forms of immunodeficiency [5, 6]. Acute acalculous cholecystitis in previously healthy children is less frequently observed and is most commonly associated with bacterial or viral infections [7]. While acute acalculous cholecystitis due to Salmonella species has been reported in adults and pediatric patients, it is uncommon in previously healthy individuals. Furthermore, there are no clear recommendations for management and treatment of acute acalculous cholecystitis secondary to salmonella typhi.

Case presentation

At our institution, a 13-month-old, healthy, full-term girl presented with a 36-h history of non-bilious, non-bloody emesis, diffuse abdominal pain, and lethargy. There was no recent travel or known exposure to dirty water, incorrectly prepared food, or animal feces. On arrival, she was tachycardic with low-grade temperatures to 100.5°F. Labs were notable for a normal hematocrit/hemoglobin and a white blood cell count of 14,970 (per μL), without a left shift. AST/ALT were 30/23 (U/L) respectively, total bilirubin was 0.6 (mg/dL), and alkaline phosphatase was 1047 (U/L). An abdominal ultrasound was obtained that showed only a few scattered lymph nodes and was non-diagnostic. A computed tomography scan was notable for gallbladder thickening and distention, non-specific free fluid, and small bowel thickening (Fig. 1).

Fig. 1
figure 1

CT image on admission demonstrating a distended gallbladder with wall-thickening and non-specific free fluid

Upon evaluation by the pediatric surgery, there was evidence of peritonitis on examination with diffuse abdominal tenderness, guarding, and rebound. Intravenous piperacillin-tazobactam was started, and she was taken for emergent diagnostic laparoscopy after resuscitation.

On laparoscopy, there was a large amount of white, non-malodorous pus in the abdomen and a normal appearing appendix. The examination of the abdomen with the laparoscope revealed a significantly distended gallbladder (Fig. 2) with omentum and duodenum adherent to the gallbladder. The aspiration of the gallbladder revealed frank pus. The laparoscopy was converted to an open procedure as exposure to the biliary tract was limited by inflammatory adhesions. There was no frank perforation in the gallbladder. While there was no perforation of the gallbladder, we hypothesize that the generalized peritonitis was secondary to transmural spread of the bacteria. A cholecystostomy tube and a Jackson-Pratt drain were placed.

Fig. 2
figure 2

Laparoscopic image of right upper quadrant demonstrating adherent omentum overlying the gallbladder

The culture of the pus obtained from the lumen of the gallbladder was positive for Salmonella typhi on postoperative day 1, and intravenous antibiotics were changed to ceftriaxone 50 mg/kg two times per day for 14 days. The patient progressed well and by POD # 5 was tolerating a regular diet. Repeat complete metabolic panel prior to discharge was normal and notable for decreased alkaline phosphatase (639 units/L), normal AST/ALT (27/13 units/L), and total bilirubin (< 0.3 mg/dL). No postoperative inflammatory markers were obtained after antibiotic treatment. Ceftriaxone was continued until discharge per infectious disease recommendations, and the patient was transitioned to oral amoxicillin/clavulanic acid until follow-up in clinic. Prior to her discharge, her drain was clamped. She was seen in clinic 2 weeks after in good spirits, with scant drain output when unclamped in clinic, tolerating a regular diet without abdominal pain or additional episodes of emesis. Eight weeks after placement of the cholecystostomy tube, a cholangiogram (Fig. 3) revealed normal biliary anatomy, and the cholecystostomy tube was removed. The patient has remained asymptomatic and is doing well over 9 months later.

Fig. 3
figure 3

Eight weeks post-op cholangiogram through cholecystostomy demonstrating patent cystic duct entering common bile duct with appropriate, uninterrupted drainage to duodenum

Discussion

Acute acalculous cholecystitis is an infrequent yet serious medical and surgical condition. Salmonella typhi has previously been reported as a cause of acalculous cholecystitis in adults, but is exceptionally rare in the pediatric population, with an aggregate of only 27 cases described in a total of 6 case reports in the literature (Table 1) [8,9,10,11,12,13]. Salmonella is typically transmitted to humans through contaminated food or water. Although there was no known exposure for this patient, the family had been displaced by a category four hurricane, Hurricane Ida, which increased the risk of exposure.

Table 1 Reported cases of acute acalculous cholecystitis due to Salmonella typhi in pediatric patients

Interestingly, in the pre-operative ultrasound, there were no significant findings as noted by our radiologists; however, operatively, we observed significant gallbladder distention. In review of this discrepancy, the radiology team believes that technician techniques may have inhibited visualization of the free fluid. The review of such discrepancies in the literatures shows there is limited data from a single-institution study that demonstrates ultrasound is not as sensitive in diagnosing pediatric cholecystitis as previously thought. In this study by Blackwood et al., 95.3% of patients who underwent a preoperative ultrasound had it read as cholelithiasis while the pathology results showed 92% having either acute or chronic cholecystitis findings [2]. The exact sensitivity and specificity of ultrasound in the pediatric population is an area that will require further investigation.

While treatment for acute acalculous cholecystitis in adults can range from antibiotics and tube cholecystostomy or cholecystectomy, in the setting of suspected Salmonella cholecystitis, non-operative treatment with antibiotics has been successful [9, 13, 14]. Oftentimes, unless blood cultures are positive for Salmonella typhi, the exact etiology of the cholecystitis is unknown until pathology returns. In our case presentation, we believe that clearance of the infection will result in long-term resolution of this patient’s cholecystitis. As the disease process is not one of obstruction, but instead infection, we hypothesize the patient will not suffer from recurrent biliary colic or chronic cholecystitis. However, a long-term follow-up will be required to determine if the gallbladder remains healthy or if cholecystectomy will be required in the future. At this time, the patient is 9 months out from treatment of her acalculous cholecystitis without recurrence or issue.

Conclusion

The incidence of pediatric acalculous cholecystitis secondary to Salmonella typhi is an uncommon finding, with only 6 case series/reports in the literature. Cholecystectomy is oftentimes the treatment as these patients have cholecystitis on imaging and physical examination. However, in this case report and a few others, antibiotic treatment with drainage via a cholecystostomy tube is seemingly an acceptable treatment as this is not an obstructive disease process. Nevertheless, further long-term follow-up of our patient is warranted. Furthermore, investigation into the pathophysiology of this disease process and diagnostic methods used to formulate a diagnosis are required prior to pursuing antibiotic therapy and placement of a cholecystostomy tube as standard treatment.

Availability of data and materials

Available upon request.

References

  1. Lu YA, Chiu CH, Kong MS, Wang HI, Chao HC, Chen CC. Risk factors for poor outcomes of children with acute acalculous cholecystitis. Pediatr Neonatol. 2017;58(6):497–503.

    Article  Google Scholar 

  2. Blackwood BP, Grabowski J. Chronic cholecystitis in the pediatric population: an underappreciated disease process. Gastroenterol Hepatol Bed Bench. 2017;10(2):125–30.

    Google Scholar 

  3. Rescorla FJ, Grosfeld JL. Cholecystitis and cholelithiasis in children. Semin Pediatr Surg. 1992;1(2):98–106.

    CAS  PubMed  Google Scholar 

  4. Yi DY, Chang EJ, Kim JY, Lee EH, Yang HR. Age, predisposing diseases, and ultrasonographic findings in determining clinical outcome of acute acalculous inflammatory gallbladder diseases in children. J Korean Med Sci. 2016;31(10):1617–23.

    Article  Google Scholar 

  5. Simões AS, Marinhas A, Coelho P, Ferreira S. Acalculous acute cholecystitis during the course of an enteroviral infection. BMJ Case Rep. 2019;12(4):e228306.

    Article  Google Scholar 

  6. Yamashita Y, Kimura T, Tanaka N, Yazaki M, Itagaki T, Joshita S, et al. Salmonella Enteritidis cholecystitis with chronic granulomatous disease. IDCases. 2018;12:49–52.

    Article  Google Scholar 

  7. Poddighe D, Tresoldi M, Licari A, Marseglia GL. Acalculous acute cholecystitis in previously healthy children: general overview and analysis of pediatric infectious cases. Int J Hepatol. 2015;2015:459608.

    Article  Google Scholar 

  8. Abdur-Rahman OL, Adeniran OJ, Nasir AA. Outcome of acalculous cholecystitis from typhoid in Nigerian children. J Natl Med Assoc. 2009;101(7):717–9.

    PubMed  Google Scholar 

  9. Ali R, Ahmed S, Qadir M, Atiq H, Hamid M. Salmonella cholecystitis: atypical presentation of a typical condition. J Coll Physicians Surg Pak. 2013;23(10):826–7.

    PubMed  Google Scholar 

  10. Chirdan LB, Iya D, Ramyil VM, Sule AZ, Uba AF, Ugwu BT. Acalculous cholecystitis in Nigerian children. Pediatr Surg Int. 2003;19(1-2):65–7.

    Article  CAS  Google Scholar 

  11. Herman HK, Hampshire KN, Khoshnam N, Khan AA, Jerris R, Abramowsky CR, et al. Suppurative granulomatous cholecystitis in a pediatric chronic carrier with Salmonella enterica serotype typhi: a case report and review of literature. Fetal Pediatr Pathol. 2016;35(2):129–32.

    Article  Google Scholar 

  12. Crane O, Muthucumaru M, Clarke M. Typhoid fever complicated by cholecystitis in a 10-year-old boy. J Paediatr Child Health. 2019;55(5):594–6.

    Article  Google Scholar 

  13. Yulevich A, Cohen Z, Maor E, Bryk T, Mares AJ. Acute acalculous cholecystitis caused by Salmonella typhi in a 6-year-old child. Eur J Pediatr Surg. 1992;2(5):301–3.

    Article  CAS  Google Scholar 

  14. Ruiz-Rebollo ML, Sánchez-Antolín G, García-Pajares F, Vallecillo-Sande MA, Fernández-Orcajo P, Velicia-Llames R, et al. Acalculous cholecystitis due to Salmonella enteritidis. World J Gastroenterol. 2008;14(41):6408–9.

    Article  Google Scholar 

Download references

Acknowledgements

None

Funding

No funding was secured for this study.

Author information

Authors and Affiliations

Authors

Contributions

Dr. Ghio, Dr. Brandt, Dr. Billiot, and Dr. Zagory performed the literature review, drafted the initial manuscript, then reviewed, and revised the manuscript. The authors approved the final manuscript as submitted and agree to be accountable for all aspects of the work.

Corresponding author

Correspondence to Michael Ghio.

Ethics declarations

Ethics approval and consent to participate

Ethics approval is not applicable, and written informed consent was obtained from the patient for publication of this case report and accompanying images.

Competing interests

The authors declare that they have no competing interests.

Additional information

Publisher’s Note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

Rights and permissions

Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/.

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Ghio, M., Billiot, A., Zagory, J.A. et al. Cholecystitis secondary to Salmonella typhi: a rare pathology with an unreported management option—a case report and literature review. Ann Pediatr Surg 18, 67 (2022). https://doi.org/10.1186/s43159-022-00199-8

Download citation

  • Received:

  • Accepted:

  • Published:

  • DOI: https://doi.org/10.1186/s43159-022-00199-8

Keywords