CEN Case Reports (2020) 9:313–317

https://doi.org/10.1007/s13730-020-00479-5

CASE REPORT

Coexistence of emphysematous cystitis and bilateral emphysematous

pyelonephritis: a case report and review of the literature
Venice Chávez‑Valencia1 · Citlalli Orizaga‑de‑La‑Cruz1 · Omar Aguilar‑Bixano1 ·
Francisco Alejandro Lagunas‑Rangel2

Received: 30 November 2019 / Accepted: 12 April 2020 / Published online: 23 April 2020
© Japanese Society of Nephrology 2020

Abstract
Emphysematous pyelonephritis (EPN) is a necrotizing infection characterized by the production of gas in the renal paren-
chyma, collecting system or perirenal tissue. Meanwhile, emphysematous cystitis (EC) is a clinical entity characterized by
the presence of gas inside and around the bladder wall. Interestingly, although both diseases are common in patients with
diabetes mellitus, these are rarely combined. We report a rare case of a 56-year-old diabetic male suffering from fever, head-
ache and vomiting and in which a diagnosis of septic shock was established due to coexistence of EC and bilateral EPN. The
emphysematous diseases improved with a conservative treatment approach using antibiotic therapy and glycemic control,
we highlight that the nephrectomy was not necessary in our patient despite the fact that he presented risk factors that predict
the failure of conservative treatment.

Keywords  Emphysematous pyelonephritis · Emphysematous cystitis · Computed tomography · Type 2 diabetes mellitus ·
Septic shock

Introduction Emphysematous pyelonephritis has been defined as a

Diabetes mellitus is a chronic disease whose treatment has
traditionally focused on glycemic control, but accumulating
evidence suggests that the clinical management of patients
requires a more comprehensive approach to minimize asso-
ciated morbidity and mortality [1]. In this way, patients with
diabetes are more prone to get urinary tract infection (UTI),
because high levels of urine sugar provide a pathogen-
friendly growth environment, where emphysematous pye-
lonephritis (EPN), emphysematous cystitis (EC), renal and
perinephric abscesses, urosepsis, and bacteremia are some
common complications, although these are rarely combined.
necrotizing infection of the renal parenchyma and its sur-
roundings that results in the presence of gas in the renal
parenchyma, collecting system or perinephrotic tissues [2].
Meanwhile, emphysematous cystitis is a clinical entity, char-
acterized by the presence of gas inside and around the blad-
der wall produced by bacterial or fungal fermentation [3].
Here, we present a rare case of EC and bilateral EPN
coexistence in a 56-year-old male who was successfully
managed with medical treatment.
Case report

A 56-year-old male patient presented to the department of

* Venice Chávez‑Valencia
drvenicechv@yahoo.com.mx
1
Department of Nephrology, Hospital General Regional
Hospital No 1, Instituto Mexicano del Seguro Social,
Bosques de los Olivos No. 101. Av. La Goleta Mpo Charo,
CP: 61301 Morelia, Michoacán, Mexico
2
Department of Genetics and Molecular Biology, Centro
de Investigación y de Estudios Avanzados del Instituto
Politécnico Nacional (CINVESTAV), Mexico, Mexico
nephrology after 6 days suffering weakness, fever, headache
and vomiting. The patient, who weighed 58 kg and had a
height of 1.63 m (body mass index of 21.8), was previously
diagnosed with type 2 diabetes mellitus (DM2) 18 years
ago, maintaining a treatment with insulin glargine 10 IU/
day and with regular glucose control. He also had a history
of smoking for 40 years with 6 cigarettes per day, currently
suspended.

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314 CEN Case Reports (2020) 9:313–317

Upon admission, the patient had a fever of 38.9  °C, Arterial gasometry revealed metabolic acidosis, hypoalbu-
tachypnea, tachycardia of 116 beats/min and hypotension minemia, serum sodium of 122 mmol/L, serum chloride
with a systolic blood pressure less than 90 mmHg, showing of 91 mmol/L and serum potassium of 5.6 mmol/L. Neu-
sepsis data that were confirmed with paraclinical studies. trophil–lymphocyte ratio at admission was 9.71 and lactic
acid was not measured, other laboratory results are shown
in Table 1.
Table 1  Results of laboratory tests The renal ultrasound showed evidence of air in the paren-
chyma, where the left kidney measured 148 × 79 × 66 mm
Laboratory tests Patient results upon Patient
admission results upon and the right kidney 128 × 73 × 57 mm, suggestive of EPN.
discharge Computed tomography (CT) of the abdomen and pel-
vis showed bilateral EPN together with the presence of EC
Total leukocytes (­ 103/µL) 24.08 8.8
(Fig. 1). EPN was classified within the class 4 according to
Total neutrophils ­(103/µL) 20.8 4.58
Huang et al. [2], which is considered the most serious.
Total lymphocytes (­ 103/ 2.14 2.26
µL) Patient blood and urine cultures showed growth of ESBL-
Band cells (%) 85 3 positive Escherichia coli, with sensitivity to ertapenem,
Hemoglobin (g/dL) 10.3 12 imipenem, meropenem, amikacin, tigecycline and trimetho-
Platelets ­(103/µL) 378 375 prim/sulfamethoxazole, but resistant to ampicillin, cefazolin,
Glucose (mg/dL) 534 150 cefepime and ceftriaxone.
Blood urea nitrogen (mg/ 76.77 49.9 The patient received treatment by central venous cath-
dL) eter with intravenous fluids, intravenous antibiotics (initially
Creatinine (mg/dL) 4.8 2.6 empirical ceftriaxone and on the third day after admission it
General urine test pH 5, protein traces, pH 6, glu- was changed to meropenem according to antibiotic sensitiv-
glucose 3+, leukocytes cose 1+, ity tests), insulin infusion and inotropic support with amines
30–35/field leukocytes as well as bladder catheterization.
5–8/field

Fig. 1  Abdominal computed tomography without contrast. a The and air densities (arrows) within the renal parenchyma. c The sagittal
axial section showed air bladder and wall (arrow). b The coronal sec- section showed air in the bladder, right and left kidney (arrows)
tion showed enlarged bilateral kidney with parenchymal destruction

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On the third day of hospitalization, our patient required
renal replacement therapy due to acute renal injury with
oliguria and refractory acidosis, so he received three ses-
sions of hemodialysis (HD).
Urology service suggested conservative treatment,
no percutaneous catheter drainage or nephrectomy. The
patient’s clinical conditions improved markedly on the
fifth day of treatment, and 15 days after admission, the
patient was afebrile, with total leukocytes of 8.8 × 103/μL,
albumin 2.5 g/dL, serum creatinine 2.6 mg/dL, serum urea
164.8 mg/dL, serum sodium 134 mmol/L, serum chloride
100 mmol/L, serum potassium of 4.4 mmol/L and a neu-
trophil–lymphocyte ratio of 0.98. Other laboratory results
are shown in Table 1.
The glomerular filtration rate was estimated one week
after the last HD session with the modification of diet
in renal disease (MDRD) equation obtaining 26  mL/
min/1.73m2.
The patient was discharged on day 17 after admission
where the abdominal examination with control CT showed
a complete resolution of renal and bladder emphysema, as
well as an undeveloped control urine culture report.
Discussion
Solitary kidney with EPN and EC are, each separately,
common in patients with DM2 or immunocompromised
patients (patients with renal graft, who use steroids, with
polycystic kidneys, neurogenic bladder, alcoholism, ana-
tomical abnormalities, renal failure and immune suppres-
sion), with a female-to-male ratio of 4–6:1 [4, 5], and aver-
age age of 57 years [6]. However, bilateral EPN combined
with EC in the same patient is a strange finding that has
only been reported rarely.
Overall, EPN occurs in 90% of patients with poor dia-
betes mellitus control [2–4, 6, 7], whose most common
pathogens involved are Escherichia coli, Klebsiella pneu-
moniae, Proteus mirabilis, Clostridium septicum, Candida
albicans, Pseudomonas, among others [2–4], and its clini-
cal manifestation is similar to high urinary tract infection.
Escherichia coli is the most common microorganism in
EC and its clinical manifestation is mainly low irritative
symptoms. CT scan is the best diagnostic method since it
also allows to know the anatomical extension [3].
A meta-analysis reports that in 52% of cases of EPN the
left kidney is affected, while 37.7% affects the right side
[5], being bilateral only in 5–10.2% of cases [5, 7]. The
diagnosis of EPN is classically made by demonstrating
the presence of gas in the kidney or in the perirenal tissue
by simple X-ray of the abdomen, renal ultrasound or CT
scan [6, 7].
Based on the radiological results in the CT scan, Huang
et al. [2] created a classification system that facilitates
treatment:
Class 1: gas only in the collecting system (called
emphysematous pyelitis).
Class 2: gas in the renal parenchyma without extension
to the extrarenal space.
Class: 3A: extension of the gas or abscess to the per-
inephric space.
Class 3B: extension of the gas or abscess to the para-
renal space.
Class 4: bilateral EPN or solitary kidney with EPN.
The differential diagnosis between emphysematous
pyelitis (gas in the collecting system) and EPN (gas in the
renal parenchyma) is very important, because EPN tends
to be more severe, with a mortality of 18–33% in bilateral
EPN [1, 2, 5].
High mortality has been reported in EPN patients
who require emergency hemodialysis, present shock on
initial presentation, altered mental status, severe hypoal-
buminemia, inappropriate empirical antibiotic treatment
and polymicrobial infection [8], as well as poor results
were reported in the presence of thrombocytopenia [2].
However, Kolla et al. [9] reported improvement in eight
patients only with conservative management without
mortality.
The literature reports a mortality of 54% in patients with
secondary shock to EPN [4]. Our patient was diagnosed with
class 4 disease, had no thrombocytopenia, mental disorder or
polymicrobial infection, but he had leukocytosis, hypoalbu-
minemia and manifest shock, inappropriate empirical anti-
biotic was used the first 3 days and required hemodialysis,
which may be necessary in 62.5% of patients [9], therefore,
he presented four risk factors of mortality, two of them also
associated with the failure of conservative treatment. How-
ever, treatment according to international guidelines for the
management of sepsis (intravenous fluids, antibiotic and sup-
portive treatment) was successful, no requiring percutane-
ous catheter drainage or nephrectomy. In this regard, similar
to that reported by other authors [10–15], our patient was
recovered and hemodialysis was suspended. Nephrectomy
is considered the last option.
In the cases presented in Table 2, the average age of the
disease was 54.7 ± 11 years, most patients have diabetes
and the predominant bacteria is Escherichia coli. Inter-
estingly, six of the cases are in men, which contrasts with
that reported for independent EPN where there is a higher
prevalence in women. According to the literature, leukocy-
tosis occurs in 70–80% of cases [5] and hypoalbuminemia
(< 3 g/dL) was present in 50% [8]. In addition, two patients
required percutaneous drainage, whereas only one patient
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316 CEN Case Reports (2020) 9:313–317

Table 2  Previous reports of coexistence of EC and bilateral EPN in adults

Author [References] Age/gender Diabetes mellitus/ Bacteria Treatment Antibiotics used Result
comorbidities

Arun [10] 64/M Yes/ADPKD E. coli/Staph. aureus MM Piperacillin-tazobac- Recovered

tam/vancomycin
Behera [11] 39/F Yes NM MM + PCD Meropenem/amikacin Recovered
Behera [11] 69/M Yes NM MM Meropenem/teico- Death
planin
Lee [12] 54/M Yes E. coli MM + PCD + HD Cefuroxime Recovered
Yeh [13] 60/F NM/HD/obesity/ E. coli/Enterococcus MM + bilateral Imipenem-cilastati/ Recovered
hypertension nephrectomy vancomycin
Wang [14] 38/M Yes/HD E. coli MM Levofloxacin Recovered
Momin [15] 58/M Yes E. cloacae MM Meropenem/ertap- Recovered
enem
Current 56/M Yes ESBL-positive MM + HD Meropenem Recovered
E. coli

ADPKD autosomal-dominant polycystic kidney disease, F female, HD hemodialysis, M male, MM medical management, PCD percutaneous
drainage, NM no mentioned

needed bilateral nephrectomy, only one patient died, and the
others recovered their health.
Patients with diabetes mellitus have several factors that
can favor the development of bilateral EPN and EC in the
same patient, such as hyperglycemic conditions that favor
the growth of bacteria, a greater propensity to urinary tract
necrosis due to stress or infection, and the low immunity of
patients with chronic kidney disease, among others [14, 15].
Punatar et al. [16] described that there is 1.8 greater risk
of an unfavorable outcome in those patients with a neutro-
phil–lymphocyte ratio greater than 5 at admission, which
happened with our patient, who required three hemodialysis
sessions and then recovered renal function.
Conclusion
Patient described here was a 56-year-old man with uncon-
trolled DM2 and Escherichia coli was the causative patho-
gen, where we highlight the coexistence of EC and bilateral
EPN, which is rare to find. Furthermore, the medical treat-
ment in our patient was successful and required only tem-
porary renal replacement therapy. We emphasize that timely
diagnosis and specific antibiotic treatment avoided the need
for surgery and reduced the risk of mortality.
Funding  This research did not receive any specific grant from funding
agencies in the public, commercial, or not-for-profit sectors.
Compliance with ethical standards  tous pyelonephritis and emphysematous cystitis with autosomal-
Conflict of interest  The authors declare no conflict of interest.
Informed consent  Written informed consent was obtained from the
patient.
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