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Saudi Journal of Kidney Diseases and Transplantation
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Year : 2018  |  Volume : 29  |  Issue : 6  |  Page : 1484-1487
Encapsulating peritoneal sclerosis associated with Burkholderia pseudomallei peritonitis

Department of Internal Medicine, Kulliyyah of Medicine, International Islamic University Malaysia, Kuantan, Pahang, Malaysia

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Date of Submission03-Jan-2018
Date of Decision14-Feb-2018
Date of Acceptance17-Feb-2018
Date of Web Publication27-Dec-2018


Burkholderia pseudomallei is a known motile organism in soil. Its infection is usually described in immunocompromised patients. It inflicts serious infection with high mortality and morbidity rate. We report a rare case of an end-stage renal disease patient on regular continuous ambulatory peritoneal dialysis (PD) who developed melioidosis PD peritonitis. Within a short period of time, she developed encapsulating peritoneal sclerosis evidenced by the intraoperative findings of intraabdominal cocooning. Choice and duration of antibiotic are important for proper eradication of the organism. Early diagnosis and treatment of both conditions also may improve the prognoses.

How to cite this article:
Aulia Z, Wan Ali WS, Shahar MA. Encapsulating peritoneal sclerosis associated with Burkholderia pseudomallei peritonitis. Saudi J Kidney Dis Transpl 2018;29:1484-7

How to cite this URL:
Aulia Z, Wan Ali WS, Shahar MA. Encapsulating peritoneal sclerosis associated with Burkholderia pseudomallei peritonitis. Saudi J Kidney Dis Transpl [serial online] 2018 [cited 2022 Dec 2];29:1484-7. Available from: https://www.sjkdt.org/text.asp?2018/29/6/1484/248318

   Introduction Top

Melioidosis is caused by Burkholderia pseudomallei, a motile, aerobic gram-negative bacilli. The known portals of entry into human hosts are skin, pulmonary inhalation, and alimentary tract.[1] Melioidosis usually infects patients who have low immunity.[2] Although a wide range of clinical manifestations from this organism was known, peritoneal dialysis (PD) peritonitis secondary to melioidosis has rarely been reported. PD-related peritonitis due to melioidosis is uncommon but highly fatal.[3] The correct choice of antibiotic is important to reduce mortality.[4],[5] Even though encapsulating peritoneal sclerosis (EPS) is a known sequelae of peritonitis,[6]B. pseudomallei peritonitis associated encapsulating peritoneal sclerosis (EPS) has never been reported.

   Case Report Top

Miss NA, a 31-year-old female with end-stage renal failure, has started continuous ambulatory PD (CAPD) since 2011. She tolerated her CAPD very well without a single episode of peritonitis until September 2016 where she presented with abdominal pain, diarrhea, fever, and cloudy PD fluid for a week. There was no history of any travel nor any contact with soil before.

On initial assessment, she has spiking temperature and high blood pressure. The abdomen was tender on palpation, but the exit site of Tenckhoff catheter did not show any evidence of infection. Blood investigations revealed that she has inadequate dialysis as the urea was 28 mmol/L with creatinine of 1887 umol/L at presentation. Septic parameters were elevated with CRP of 327 mg/L and PD fluid leukocyte cell count of 2250 cells/mm3 (predominantly neutrophils, 92%).

She was started empirically on intraperitoneal (IP) antibiotics (ceftazidime 1 g loading and 250 mg/PD bag and cloxacillin 1 g loading and 250 mg/PD bag) and rapid PD cycle was done to clear up the PD fluid effluents. However, there was no improvement after five days of IP antibiotics. She has spiking temperature, and her PD leukocyte cell counts jumped up to 5634 cells/mm3. PD fluid culture and sensitivity (C and S) came back as positive for B. pseudomallei.

The antibiotics were then changed to intravenous (IV) ceftazidime 1 g BD, and the Tenckhoff catheter was removed. She continued her dialysis through hemodialysis through temporary intrajugular vein catheter. Septic parameters improved, however, she developed left foot abscess that required incision and drainage. The blood and pus culture and sensitivity from the foot abscess also grew B. pseudomallei which was sensitive to ceftazidime. Abdominal ultrasound revealed no other abscess or collection elsewhere.

She was diagnosed with disseminated melioidosis and has completed IV ceftazidime 1 g BD for six weeks. Repeated blood C and S revealed no growth and she improved clinically and biochemically. She was discharged with oral augmentin 1250 mg bd that was planned to be completed for five months.

After one month of maintenance antibiotic, the patient came back with abdominal pain and diarrhea. There was guarding over the abdomen and she was in septic shock. In this current admission, there were signs of malnutrition with cachexia and weight loss. Urgent Exploratory laparotomy was done in view of the suspicion of perforated viscus and found severe adhesions with generalized cocooning of the abdomen. She succumbed to death a few days after the surgery.

   Discussion Top

The fact that this patient has end stage renal disease and was on CAPD which required at least four times exchanges of peritoneal fluid a day has put this patient at risk of acquiring meliodosis. B. pseudomallei is ubiquitous although commonly associated with soil contamination.[7] The infection of the peritoneum also caused insufficient dialysis in this patient evidenced by the parameters done on admission.

The patient initially received IP antibiotic which subsequently changed into IV administration due to worsening septic parameters. International Society for PD guidelines recommended IP administration over IV administration and study found that concentration of antibiotics is higher if administered via IP.[8],[9] Systematic review also favor IP administration of antibiotic in peritonitis.[10] There is no study yet to compare the efficacy of IP versus IV antibiotics for the treatment of PD peritonitis secondary to melioidosis at the moment. Response to antibiotics would also be theoretically different in view of different pathogenesis involved in B. pseudomallei infection.

The choice of ceftazidime in this patient is based on sensitivity and recommendation. White et al demonstrated that ceftazidime could reduce overall mortality by 50% in a randomized clinical trial.[11] The biggest dilemma facing all clinicians managing a patient with melioidosis is the optimal duration of antibiotic therapy. As for this patient, we prolonged the IV antibiotic for six weeks as recommended for initial therapy in disseminated melioidosis, and continued with oral augmentin for five months for eradication therapy.[12]

To date, there was no case report on B. pseudomallei peritonitis associated EPS. The known risk of EPS is the duration of CAPD. This patient has been on CAPD for almost five years and risk of developing EPS is around 0.7% as reported in Japanese Prospective Study and up to 8.8% according to a Scottish cohort study.[13],[14] Recurrent peritonitis, especially caused by Staphylococcus, Pseudomonas and fungal infection, are also known risk factors of EPS.[15] However, this patient never had peritonitis before. She also did not have any signs or symptoms suggestive of EPS before this. B. pseudomallei is postulated to induce more severe inflammation and fibrosis compared to other types of microorganism. The mortality rate for patients with EPS is great at 25%–55%, predominantly in the year after diagnosis.[14] This patient only takes a few months to develop EPS after the diagnosis of melioidosis peritonitis and succumbed. The short sequence of events also makes it difficult to ascertain whether the EPS was a direct consequence of melioidosis peritonitis, or already an ongoing process from before.

   Conclusion Top

End-stage renal disease with immunocom-promised state is a known risk factor in acquiring melioidosis. Disseminated melioidosis involving peritonitis and EPS will increase the risk of mortality. Choice and duration of antibiotic are important in treating acute infection and eradication is a must to prevent recurrent infection. Mode of antibiotic administration is important to deliver the adequate dose required. EPS-related melioidosis need to be suspected early as additional of immunosuppressive agent/antifibrotic agent maybe helpful in reducing the mortality rate. Adequate nutritional support, for example, parenteral nutrition in melioidosis associated peritonitis should be considered as diagnosis of EPS might be delayed as clinical manifestation is usually late. Imaging and biomarkers, for example, CA125, interleukin-6 maybe considered in asymptomatic patient with melioidosis peritonitis.[15] Due to the severity of B. pseudomallei infection, melioidosis peritonitis needs to be highly suspected especially in immunocompromised patients. Early recognition of EPS and appropriate management is also vital to reduce mortality and morbidity.

Conflict of interest: None declared.

   References Top

Barnes JL, Ketheesan N. Route of infection in melioidosis. Emerg Infect Dis 2005;11:638-9.  Back to cited text no. 1
Brett PJ, Woods DE. Pathogenesis of and immunity to melioidosis. Acta Trop 2000;74: 201-10.  Back to cited text no. 2
Kanjanabuch T, Lumlertgul N, Pearson LJ, et al. Peritoneal dialysis-related peritonitis due to melioidosis: A potentially devastating condition. Perit Dial Int 2017;37:183-90.  Back to cited text no. 3
Cheng AC, Fisher DA, Anstey NM, Stephens DP, Jacups SP, Currie BJ. Outcomes of patients with melioidosis treated with meropenem. Antimicrob Agents Chemother 2004; 48:1763-5.  Back to cited text no. 4
Raja NS. Cases of melioidosis in a university teaching hospital in Malaysia. J Microbiol Immunol Infect 2008;41:174-9.  Back to cited text no. 5
Yamamoto T, Nagasue K, Okuno S, Yamakawa T. The role of peritoneal lavage and the prognostic significance of mesothelial cell area in preventing encapsulating peritoneal sclerosis. Perit Dial Int 2010;30:343-52.  Back to cited text no. 6
Cheng AC, Currie BJ. Melioidosis: Epidemiology, pathophysiology, and management. Clin Microbiol Rev 2005;18:383-416.  Back to cited text no. 7
Li PK, Szeto CC, Piraino B, et al. Peritoneal dialysis-related infections recommendations: 2010 update. Perit Dial Int 2010;30:393-423.  Back to cited text no. 8
Vlaar PJ, van Hulst M, Benne CA, Janssen WM. Intraperitoneal compared with intravenous meropenem for peritoneal dialysis-related peritonitis. Perit Dial Int 2013;33:708-9.  Back to cited text no. 9
Ballinger AE, Palmer SC, Wiggins KJ, et al. Treatment for peritoneal dialysis-associated peritonitis. Cochrane Database Syst Rev 2014; 4:CD005284.  Back to cited text no. 10
White NJ, Dance DA, Chaowagul W, Wattanagoon Y, Wuthiekanun V, Pitakwatchara N. Halving of mortality of severe melioidosis by ceftazidime. Lancet 1989;2:697-701.  Back to cited text no. 11
How SH, Ng TH, Jamalludin AR, et al. Pahang melioidosis registry. Med J Malaysia 2009; 64:27-30.  Back to cited text no. 12
Kawanishi H, Kawaguchi Y, Fukui H, et al. Encapsulating peritoneal sclerosis in Japan: A prospective, controlled, multicenter study. Am J Kidney Dis 2004;44:729-37.  Back to cited text no. 13
Brown MC, Simpson K, Kerssens JJ, Mactier RA; Scottish Renal Registry. Encapsulating peritoneal sclerosis in the new millennium: A national cohort study. Clin J Am Soc Nephrol 2009;4:1222-9.  Back to cited text no. 14
Sampimon DE, Korte MR, Barreto DL, et al. Early diagnostic markers for encapsulating peritoneal sclerosis: A case-control study. Perit Dial Int 2010;30:163-9.  Back to cited text no. 15

Correspondence Address:
Dr. Wan Ahmad Syahril Rozli Wan Ali
Department of Internal Medicine, Kulliyyah of Medicine, International Islamic University Malaysia, Kuantan, Pahang
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/1319-2442.248318

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