Running Head: PURPLE URINE BAG SYNDROME 1

Purple Urine Bag Syndrome: A Case Report

Kevin Osvic Rengel, Noel Gomez, and Charles Eryll Sy

Department of Internal Medicine

Jose B. Lingad Memorial Regional Hospital

ABSTRACT

BACKGROUND: Purple Urine Bag Syndrome (PUBS) is a purple discoloration of the urine

over a period of hours or days following urinary catheterization. It is a rare condition with

unknown prevalence in the Philippines. The main risk factors identified are female gender,

increased dietary tryptophan, alkaline urine, constipation, chronic catheterization, high

urinary bacterial load, renal failure, and the use of a polyvinylchloride (PVC) plastic

catheter.3

CASE SUMMARY: A 61 year old woman diagnosed with end-stage diabetic nephropathy

for a year. She presented with 1-week history of altered sensorium accompanied by fever,

jaundice, malaise, anorexia, insomnia, vomiting, and abdominal pain. She was noted to be

disoriented, febrile, jaundiced with icteric sclerae and edematous. She was initially treated as

a case of Ascending Cholangitis, Acute Liver Injury and Uremia secondary to diabetic

nephropathy. Urine catheter and urine bag was also inserted upon admission of the patient to

monitor urine output. On the 7th hospital day, the urine bag was noted to be purple in color

(Figure 1). Urine culture revealed ESBL positive E. coli with 50,000 colonies/ml of urine.

Blood cultures were also positive for the same pathogen. She was then treated as a case of

Septic Encephalopathy secondary to Urosepsis. Urinary catheter and urine bag was removed,

while antibiotics were shifted to meropenem in line with the sensitivity pattern of culture

results. After the 3rd day of antibiotic therapy, repeat blood and urine cultures turned negative.

The patient had improved neurological and clinical symptoms and was subsequently

discharged after 2 weeks of antibiotic treatment.

CONCLUSION: The case presented a patient with the identified risk factors in developing

PUBS such as female gender, high urinary bacterial load, renal failure and diabetes, as

described in previous studies. This phenomenon proved useful in recognition of an

underlying urinary tract infection among catheterized patients. Awareness of such syndrome

should be done due to the high morbidity and mortality if left untreated. Immediate removal

of urine catheter and prompt administration of appropriate antibiotics were necessary to

manage this case.

Keywords: Purple Urine Bag Syndrome, PUBS, E. coli

Purple Urine Bag Syndrome: A Case Report

Purple Urine Bag Syndrome (PUBS), as its name implies, is a purple discoloration of the

urine over a period of hours or days following urinary catheterization. The syndrome was

first formally reported in The Lancet in 1978 by Barlow & Dickson1. PUBS was observed by

Dealler et al to be present in about 9.8% of institutionalized patients with long-term

indwelling catheter use2. It is a rare condition with unknown prevalence in the Philippines.

The main risk factors identified are female gender, increased dietary tryptophan, alkaline

urine, constipation, chronic catheterization, high urinary bacterial load, renal failure, and the

use of a polyvinylchloride (PVC) plastic catheter.3

CASE REPORT

This is a case of a 61 year old woman diagnosed with end-stage diabetic nephropathy for a

year. She was previously advised to undergo hemodialysis but refused due to financial

constraints. She presented with 1-week history of altered sensorium accompanied by fever,

jaundice, malaise, anorexia, insomnia, vomiting, and abdominal pain. She is diabetic for 15

years, maintained on insulin, and had a history of coronary artery disease maintained on

clopidogrel, rosuvastatin, metoprolol, enalapril, and isosorbide mononitrate.

She was admitted in the medical ward. She was noted to be disoriented, febrile, jaundiced

with icteric sclerae and edematous. Laboratory work ups revealed elevated BUN, creatinine,

serum liver enzymes, alkaline phosphatase, total bilirubin and direct bilirubin. Hepatitis

profile revealed gray zone reactive anti-HAV. Her whole abdominal ultrasound revealed

hepatomegaly, minimal intraperitoneal fluid and unremarkable gallbladder, pancreas, spleen,

and kidneys.
PURPLE URINE BAG SYNDROME 5

She was initially treated as a case of Ascending Cholangitis, Acute Liver Injury and Uremia

secondary to diabetic nephropathy. Piperacillin-tazobactam, metronidazole and lactulose

were started, and emergency hemodialysis was also initiated. Urine catheter and urine bag

was also inserted upon admission of the patient to monitor urine output. However, no

improvement on the status of the patient was seen.

On the 7th hospital day, the urine bag was noted to be purple in color (Figure 1). Urine culture

revealed ESBL positive E. coli with 50,000 colonies/ml of urine. Blood cultures were also

positive for the same pathogen. The case was referred to an Infectious Disease specialist, and

then treated as a case of Septic Encephalopathy secondary to Urosepsis. Urinary catheter and

urine bag was removed, and antibiotics were shifted to meropenem in line with the sensitivity

pattern of culture results. After the 3 rd day of antibiotic therapy, repeat blood and urine

culture turned negative. The patient had improved neurological and clinical symptoms and

was subsequently discharged after 2 weeks of antibiotic treatment.

Image 1. Image of the purple urinary bag

DISCUSSION

PUBS is currently thought to be a result of production of indigo and indirubin pigments from

deamination of dietary Tryptophan to indole, followed by hepatic conjugation into Indoxyl

sulfate, acted upon by bacterial enzymes producing indoxyl. The purple color staining the

urine bag results from the combination of those pigments.4

Tryptophan

intestine bacteria

Indole

liver detoxification

Indoxyl sulphate

urine Indoxyl
sulphatase/
phosphatase*
Indoxyl

Indigo and
Indirubin pigements

Figure 1. Pathogenesis of purple discoloration of urine

In this case, the presence of urinary tract infection may have greatly increased the availability

of bacterial sulphatases and phosphatases resulting to more indigo and indirubin prigment

produced. Patient also had an end stage renal disease requiring hemodialysis, resulting to

impaired clerance of indoxyl sulphate, thereby increasing substrates for bacterial sulphatases.

These two significant risk factors were identified that may have greatly predisposed the

patient into developing PUBS.

In a study done by Hsiu-wu et.al 6, wherein published articles of PUBS from 1980-2016 were

gathered, and 116 cases were analyzed. The mean age of the patients was 75.6 years old, and

PUBS was more commonly observed in females than in males (1.5:1 ratio). Only 11.8% of

cases presented with fever, and 8.6% of cases with shock. Majority of the cases (93.1%) had

alkaline urine. The majority of cases (69.8%) had constipation, and 58.3% lived in long-term
PURPLE URINE BAG SYNDROME 7

care units. Regarding chronic co-morbidity, 19.2% of cases had diabetes mellitus and 18.8%

were uremic patients. Overall mortality rate was 6.8%. And the top 5 most common bacterial

species were E. coli, Enterococcus spp., Proteus spp., M. morganii, and Klebsiella spp. With

regard to our patient, she also presented with constipation, with chronic co-morbidity of

chronic kidney disease and diabtes mellitus.

The case reported the presence of Escherichia coli isolated from urine and blood, which is,

according to the study of Hadano et al., one of the most common microorganisms noted to

cause PUBS.7 Treatment in patients with ESBL-producing bacteria pose a great challenge to

clinicians because of broad range of beta lactamase resistance which limit treatment options.

In the study by Ramphal et.al patients with ESBL-producing bacteria has a four times higher

mortality compared to a non ESBL producing pathogen.8

CONCLUSION

The case presented a patient with the identified risk factors in developing PUBS such as

female gender, high urinary bacterial load, renal failure and diabetes, as described in previous

studies. This phenomenon proved useful in recognition of an underlying urinary tract

infection among catheterized patients. Awareness of such syndrome should be promoted due

to the high morbidity and mortality if left untreated. Immediate removal of urine catheter and

prompt administration of appropriate antibiotics were necessary to manage this case.

REFERENCES

1. Barlow, G., & Dickson, J. (1978). Purple Urine Bags. The Lancet, 311(8057), 220-

221. doi:10.1016/s0140-6736(78)90667-0
PURPLE URINE BAG SYNDROME 8

2. Dealler, S. F., Belfield, P. W., Bedford, M., Whitley, A. J., & Mulley, G. P. (1989).

Purple Urine Bags. Journal of Urology, 142(3), 769-770. doi:10.1016/s0022-

5347(17)38882-1

3. Khan, F., Chaudhry, M. A., Qureshi, N., & Cowley, B. (2011). Purple Urine Bag

Syndrome: An Alarming Hue? A Brief Review of the Literature. International Journal

of Nephrology, 2011, 1-3. doi:10.4061/2011/419213

4. Dealler SF, Hawkey PM, Millar MR. Enzymatic degradation of urinary indoxyl

sulfate by providencia stuartii and Klebsiella pneumonia causes the purple urine bag

syndrome. J Clin Microbiol 1988;26:2152–6

5. Kayal, A., Dhanuka, S., Mukhopadhyay, B. C., Mandal, T. K., & Bansal, C. L.

(2017). Purple urine bag syndrome in benign prostatic hyperplasia patient. Renal

Replacement Therapy, 3(1). doi:10.1186/s41100-017-0134-7

6. Yang, H., & Su, Y. (2018). Trends in the epidemiology of purple urine bag syndrome:

A systematic review. Biomedical Reports. doi:10.3892/br.2018.1046

7. Hadano, Y., Shimizu, Takada, Inoue, & Sorano. (2012). An update on purple urine

bag syndrome. International Journal of General Medicine, 707.

doi:10.2147/ijgm.s35320

8. Ramphal, R., & P. G. (2006). Extended-Spectrum β-Lactamases and Clinical

Outcomes: Current Data. Clinical Infectious Diseases, 42(Supplement_4).

doi:10.1086/500663