Is emphysematous pyelitis a precursor lesion of emphysematous pyelonephritis? A case report of bilateral emphysematous pyelitis caused by extended-spectrum beta-lactamase Escherichia coli and literature review

Mehboob Ahmed Rehan; Asma Rashid; Quincy Tran; Ahmed Mahmood; Usman Khan; Speirs Shawn; Douglas Whatmore

doi:10.4103/IJAM.IJAM_27_19

CASE REPORT

Year : 2020 | Volume : 6 | Issue : 1 | Page : 36-39

Is emphysematous pyelitis a precursor lesion of emphysematous pyelonephritis? A case report of bilateral emphysematous pyelitis caused by extended-spectrum beta-lactamase Escherichia coli and literature review

Mehboob Ahmed Rehan¹, Asma Rashid¹, Quincy Tran², Ahmed Mahmood¹, Usman Khan¹, Speirs Shawn¹, Douglas Whatmore¹
¹ Department of Medicine, Eastern Idaho Regional Medical Center, Idaho Falls, Idaho, USA
² Department of Emergency Medicine, School of Medicine, University of Maryland, Baltimore, MD, USA

Date of Submission	04-Jun-2019
Date of Acceptance	18-Nov-2019
Date of Web Publication	27-Mar-2020

Correspondence Address:
Dr. Mehboob Ahmed Rehan
3100 Channing Way, Idaho Falls, ID 83404
USA

Source of Support: None, Conflict of Interest: None

Check

DOI: 10.4103/IJAM.IJAM_27_19

Abstract

The presence of air in different parts of renal excretory system needs accurate assessment, and careful consideration as its progression to renal parenchyma can lead to fulminant course with substantial mortality. Bilateral emphysematous pyelonephritis is a well-known entity, but bilateral emphysematous pyelitis (EP) along with emphysematous cystitis is extremely rare. An internet search with the term “bilateral emphysematous pyelitis” and “bilateral emphysematous pyelitis and ureteritis with cystitis” only found three case reports in English. We present an interesting case of bilateral EP, ureteritis, and cystitis in a nondiabetic male patient without any history of immunodeficiency (HIV or chronic steroid use) but with a history of nonobstructing bilateral multiple renal calculi who was successfully managed medically. EP, if diagnosed early and managed properly, has an excellent prognosis with complete recovery following medical treatment.
The following core competencies are addressed in this article: Practice-Based Learning and Improvement, Patient care and Procedural skills, Medical Knowledge.

Keywords: Emphysematous pyelitis, Escherichia coli, urinary tract infection

How to cite this article:
Rehan MA, Rashid A, Tran Q, Mahmood A, Khan U, Shawn S, Whatmore D. Is emphysematous pyelitis a precursor lesion of emphysematous pyelonephritis? A case report of bilateral emphysematous pyelitis caused by extended-spectrum beta-lactamase Escherichia coli and literature review. Int J Acad Med 2020;6:36-9

How to cite this URL:
Rehan MA, Rashid A, Tran Q, Mahmood A, Khan U, Shawn S, Whatmore D. Is emphysematous pyelitis a precursor lesion of emphysematous pyelonephritis? A case report of bilateral emphysematous pyelitis caused by extended-spectrum beta-lactamase Escherichia coli and literature review. Int J Acad Med [serial online] 2020 [cited 2022 Jan 25];6:36-9. Available from: https://www.ijam-web.org/text.asp?2020/6/1/36/281455

Introduction

The first case of gas in urinary tract was described in 1671.^[1] In 1825, Brierre de Boismont explained the production of gas in urinary tract and thought it as a form of flatulence or gas secretion by mucosal cells of urinary tract.^[1] On August 20, 1898, the Journal of the American Medical Association published an article called “Pneumaturia” authored by two brilliant surgeons from Baltimore, Kelly, and MacCallum who were the first to review case reports and literature on pneumaturia. They described urinary tract infection (UTI), instrumentation, and vesicoenteric fistula as the most common causes^[1] and also explained that it is more common in patients who are females, diabetics, and those who have urinary tract obstruction.^[1] The most common offending pathogen found was Bacillus coli communis,^[1] now known as Escherichia ^{More Details} coli.

Case Report

A 55-year-old Spanish-speaking male presented with right flank pain, nausea, fever, chills, and pneumaturia of 2 weeks' duration. The patient denied any fecaluria and hematuria. His vitals at presentation were pulse 114 bpm, respiratory rate 18/min, blood pressure 95/60 mmHg, and maximum temperature of 102.7 F. He had severe costovertebral angle tenderness o physical examination. The patient denied any history of diabetes mellitus, and his A1C was 5.2. Urinalysis showed a moderate amount of leukocytes, bacterial clumps, and urine white blood cell count of 86 cells/uL. It was positive for microscopic hematuria without glycosuria or proteinuria. Laboratory values were significant for serum leukocytes 19,000 cells/uL with right shift, platelets of 236 k/mm³, creatinine of 2.5 mg/dL, and random blood glucose level of 136 mg/dL, his laboratories showed sodium 132, potassium 4.6, chloride 108, bicarbonate 16, blood urea nitrogen 40, and hemoglobin 11.4. Computerized tomography (CT) of kidney, ureter, and bladder showed air in the left and right renal collecting system, bilateral ureters [Figure 1], and the urinary bladder [Figure 2], there were bilateral intrarenal abscesses, with one measuring 1.7 cm × 1.3 cm in the right anterior mid pole, and bilateral non-obstructing calculi. Important finding on CT scan was that there was no left renal parenchymal necrosis and no gas in renal parenchyma or perinephric tissue, which could have changed the course of treatment. The urologist was consulted who recommended against any surgical intervention. The patient was treated with intravenous (IV) antibiotic Rocephin and IV fluids. On the 3^rd day of his admission, urine culture grew extended-spectrum beta-lactamase (ESBL) E. coli sensitive only to meropenem and imipenem. The patient's antibiotic was subsequently changed, and imipenem was started. During the hospitalization, the patient stabilized clinically on medical treatment alone, his creatinine came to baseline, and he was discharged home without any complication on day 5.

Figure 1: Gas seen in the left renal pelvis and ureter, right-sided kidney calyces show two stones

Click here to view

Figure 2: Air in urinary bladder

Click here to view

Discussion

It is very important to differentiate between gas in the renal collecting system (emphysematous pyelitis [EP]) from gas in the renal parenchyma (emphysematous pyelonephritis [EPN]), as treatment options and clinical course is completely different.^[2],[3] CT scan is the modality of choice to differentiate between both.^{[2],[3],[4],[5],[6]} EP has mortality rate of 20% while EPN's mortality is reported in literature to be between 69% and 80%, and most cases require surgical intervention from partial to complete nephrectomy.^[3],[4] There is no distinguishing difference in the clinical presentation of patients with EP or EPN, both entities present almost identically with high-grade fever, nausea, vomiting, costovertebral angle tenderness, and dysuria but pneumaturia seems to be more commonly associated with EP.^[6] Wan et al. did a retrospective review of clinical prognosis and CT imaging studies of patients diagnosed with gas in the urinary tract, he divided EPN into two types, and labeled EP as type II EPN based on CT imaging.^[7] Huang and Tseng provided the most cited study to date on this topic. They divided EPN into four types based on radiological findings and distribution of air beyond renal collecting system.^[8] Uncontrolled diabetes and renal system obstruction seem to be the leading predisposing factors. E. coli and Klebsiella species are common pathogens responsible for gas production.^[7],[8],[9] The proposed mechanism of gas production in patients with diabetes is carbon dioxide production by glucose-fermenting bacteria, while in nondiabetic patients, fermentation of lactulose and proteins in association with obstruction has been proposed.^{[9],[10],[11]} Our patients case is unique because he was nondiabetic, had air in urinary bladder, both ureters, renal pelvises. He also had nonobstructing bilateral renal calculi, bilateral renal parenchymal abscesses and urine cultures positive for ESBL resistantE. col i. It is important to mention that ESBL Enterobacteriaceae has been classified by world health organization (WHO) as (critical) class 1 pathogen along with Acinetobacter baumannii

seudomonas aeruginosa.^[12] We could not find any reports of emphysematous urinary tract infections caused by ESBL pathogen. Most popular classifications of emphysema in urinary tract are based on radiological findings, and all of them include EP as a part of EPN. We could not find a single article which discusses the progression of emphysema from renal collecting system to renal parenchyma ultimately leading to necrosis. The reason for the presentation of this case report is to discuss the possibility of EPN beginning as EP. Our patient had bilateral multiple renal abscesses but no parenchymal necrosis or gas; if not treated at that time, we believe that it would have had progressed to EPN.^[13] If EPN and EP are completely separate clinical conditions than pathophysiology of both conditions should be different too, in both case scenarios, large scale prospective studies are needed with the aim to focus on the association and pathophysiology of both clinical entities. It is also important for medical community to recognize the importance of early detection of these conditions o early imaging (difficult to interpret on X-ray due to intestinal gas), especially CT scan as a diagnosis at earlier stages not only decreases morbidity and mortality but also decreases the need for surgical intervention including nephrectomy.

Conclusions

We propose that EP and EPN are different spectra of the same disease process and that EP might be an early stage of EPN which if not identified early can progress to EPN with detrimental consequences. More large scale studies are needed to better explain the severity and implications associated with these conditions, which will likely help reduce morbidity and mortality associated with emphysema in the urinary tract.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Ethical conduct of research

The authors declare that this scientific work complies with reporting quality, formatting, and reproducibility guidelines set forth by the EQUATOR Network. The authors also attest that this clinical investigation did not require Institutional Review Board/Ethics Committee Review. For this work, formal consent of the patient was obtained.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

1.	Kelly HA, MacCallum WG. Pneumaturia. J Am Med Assoc 1898;31:375-81.
2.	Bohlman ME, Sweren BS, Khazan R, Minkin SD, Goldman SM, Fishman EK. Emphysematous pyelitis and emphysematous pyelonephritis characterized by computerized tomography. South Med J 1991;84:1438-43.
3.	Kuo CY, Lin CY, Chen TC, Lin WR, Lu PL, Tsai JJ, et al. Clinical features and prognostic factors of emphysematous urinary tract infection. J Microbiol Immunol Infect 2009;42:393-400.
4.	Mokabberi R, Ravakhah K. Emphysematous urinary tract infections: Diagnosis, treatment and survival (case review series). Am J Med Sci 2007;333:111-6.
5.	Srinivasan S, Teh HS, Clarke MJ. Clinics in diagnostic imaging (138). Emphysematous pyelitis. Singapore Med J 2012;53:214-7.
6.	Tan C C K Ho GH, Bahadzor B, Praveen S, Goh EH, Syahril AS, Zulkifli MZ, et al. Bilateral emphysematous pyelitis: A rare encounter in urology. Clin Ter 2013;164:319-21.
7.	Wan YL, Lee TY, Bullard MJ, Tsai CC. Acute gas-producing bacterial renal infection: Correlation between imaging findings and clinical outcome. Radiology 1996;198:433-8.
8.	Huang JJ, Tseng CC. Emphysematous pyelonephritis: Clinicoradiological classification, management, prognosis, and pathogenesis. Arch Intern Med 2000;160:797-805.
9.	Kua CH, Abdul Aziz Y. Air in the kidney: Between emphysematous pyelitis and pyelonephritis. Biomed Imaging Interv J 2008;4:e24.
10.	Huang JJ, Chen KW, Ruaan MK. Mixed acid fermentation of glucose as a mechanism of emphysematous urinary tract infection. J Urol 1991;146:148-51.
11.	Anwar N, Chawla LS, Lew SQ. Emphysematous pyelitis presenting as an acute abdomen in an end-stage renal disease patient treated with peritoneal dialysis. Am J Kidney Dis 2002;40:E13.
12.	Asokan GV, Ramadhan T, Ahmed E, Sanad H. WHO Global Priority Pathogens List: A Bibliometric Analysis of Medline-PubMed for Knowledge Mobilization to Infection Prevention and Control Practices in Bahrain. Oman Medical Journal 2019;34:184.
13.	Grayson DE, Abbott RM, Levy AD, Sherman PM. Emphysematous infections of the abdomen and pelvis: A pictorial review. Radiographics 2002;22:543-61.