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Saudi Journal of Kidney Diseases and Transplantation
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Table of Contents   
CASE REPORT  
Year : 2018  |  Volume : 29  |  Issue : 3  |  Page : 709-713
Genitourinary melioidosis in a Bangladeshi farmer with IgA nephropathy complicated by steroid-induced diabetes mellitus


Department of Nephrology, Bangladesh Institute of Research and Rehabilitation in Diabetes, Endocrine and Metabolic Disorders (BIRDEM) General Hospital, Dhaka, Bangladesh

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Date of Submission18-Mar-2017
Date of Decision19-Apr-2017
Date of Acceptance19-Apr-2017
Date of Web Publication28-Jun-2018
 

   Abstract 

Melioidosis is an emerging infectious disease in many countries including Bangladesh. Genitourinary infection due to Burkholderia pseudomallei is a well-recognized manifestation although less commonly reported in Asia than Australia. Here, we report case history of a 38-year-old Bangladeshi farmer, diagnosed with IgA nephropathy and on oral prednisolone, who presented with features of urinary tract infection. Diagnostic workup confirmed genitourinary infection due to B. pseudomallei and diabetes mellitus. He was treated with ceftazidime followed by the combination of co-trimoxazole and doxycycline. After two-year follow-up, he was free of symptoms with no recurrence of melioidosis. In the context of growing evidence of melioidosis endemicity in Bangladesh, physicians should be aware and include melioidosis as differential in appropriate clinical scenario. Melioidosis may cause urinary tract infections and should be suspected in high-risk groups like farmers and in the presence of risk factors such as diabetes mellitus and other immunosuppressive conditions.

How to cite this article:
Rahim MA, Samad T, Ananna MA, Ul Haque WM. Genitourinary melioidosis in a Bangladeshi farmer with IgA nephropathy complicated by steroid-induced diabetes mellitus. Saudi J Kidney Dis Transpl 2018;29:709-13

How to cite this URL:
Rahim MA, Samad T, Ananna MA, Ul Haque WM. Genitourinary melioidosis in a Bangladeshi farmer with IgA nephropathy complicated by steroid-induced diabetes mellitus. Saudi J Kidney Dis Transpl [serial online] 2018 [cited 2019 Jul 21];29:709-13. Available from: http://www.sjkdt.org/text.asp?2018/29/3/709/235205

   Introduction Top


Melioidosis is caused by Burkholderia pseu-domallei, a Gram-negative bacillus found in soil and surface water.[1] It is endemic in Southeast Asia and Northern Australia but is increa-singly being recognized elsewhere[1],[2],[3],[4],[5] with an estimated global incidence of 165,000 cases and 89,000 deaths/year.[3] It was first described in 1912 in Rangoon of Burma, a country neighboring Bangladesh.[6] Recently, melioidosis is being increasingly recognized in Bangladesh with published cases,[7],[8],[9],[10],[11],[12] seroepidemiological evidence,[13],[14] and identification of organism from soil.[13] Here, we describe a case of genitourinary melioidosis occurring in a Bangladeshi farmer, who had been suffering from IgA nephropathy and had developed prednisolone-induced diabetes mellitus.


   Case Report Top


A 38-year-old Bangladeshi farmer from Feni, a south-eastern coastal district of Bangladesh, presented with a three-day history of increased urinary frequency, dysuria, and fever along with pain and swelling of the scrotum. Two weeks previously, he had received a course of oral ciprofloxacin (500 mg Q 12-h for 5 days) over-the-counter for a presumptive lower urinary tract infection, resulting in symptomatic improvement. His current complaints developed six days after the completion of antibiotics. In this time, his systemic symptoms were more severe along with pain and swelling of the scrotum. Six weeks earlier, he had been evaluated for generalized edema and massive proteinuria (urinary total protein 7.28 g/24 h) in a tertiary care health facility. At that time, he was nondiabetic (fasting blood glucose and 2-h postglucose levels were 5.4 and 6.2 mmol/L respectively). Renal biopsy had confirmed IgA nephropathy, and he was started on oral prednisolone 60 mg/day (1 mg/kg/day). He had missed follow-up visits but had continued prednisolone. He never traveled outside Bangladesh.

Admission assessment at our center revealed that the patient was febrile (101°F) but hemo-dynamically stable. He was pale with oral candidiasis and minimal bipedal edema. His scrotum was swollen and tender. Inguinal lymph nodes were not palpable. Physical examination was otherwise unremarkable. Bedside urine examination revealed proteinuria ++ and glycosuria +++. Random capillary blood glucose was 24.6 mmol/L.

Laboratory investigations revealed neutrophil leukocytosis (total leukocytes 22,900/mm[3] and neutrophils 89%), high erythrocyte sedimentation rate (50 mm in the 1st h), glycated hemoglobin (7.2%), serum creatinine (2.4 mg/dL), and low serum sodium (131 mmol/L). Urinary total protein was 5.5 g/24 h. There were numerous pus cells and red cells in urine. Ultrasonogram showed an enlarged and non-homogeneous tail of the right epididymis and adjacent testes [Figure 1]a, mildly thickened scrotal walls bilaterally, a small loculated collection in the left tunica vaginalis [Figure 1]b, mild ascites, and bilateral small pleural effusions. Human immunodeficiency virus serology (anti-HIV 1+2) was negative. Urine culture grew B. pseudomallei [Figure 2] (semiquantitative colony count >1 χ 105 colony-forming unit/mL; sensitive to augmentin, cefixime, ceftazidime, ceftriaxone, ciprofloxacin, co-trimoxazole, imipenem, mecillinam, and piperacillin-tazobactam combination and resistant to amikacin, colistin, gentamicin, netilmicin, and nitrofurantoin), but blood culture was negative. Based on the clinical and laboratory findings, he was diagnosed as a case of urinary tract infection and epididymo-orchitis due to B. pseudomallei.
Figure 1: Ultrasonography showing (a) enlarged and nonhomogeneous tail of the right epididymis and adjacent testes suggestive of acute epididymo-orchitis and small left epididymal cyst and (b) mild loculated collection in left tunica vaginal sac.

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Figure 2: Growth of Burkholderia pseudomallei from urine sample on MacConkey agar.

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He was initially started on intravenous ceftriaxone (1 g Q 24-h), but after receiving the urine culture report, antibiotic was changed to intravenous ceftazidime (2 g Q 8-h) which was continued for two weeks, followed by the combination of co-trimoxazole (960 mg Q 12 h) and doxycycline (100 mg Q 12 h) for another 20 weeks. He was started with insulin for his diabetes. His immunosuppressive therapy was adjusted according to international guidelines.[15] Other supportive therapy included fluid and electrolyte balance, paracetamol, oral antifungals, calcium, and Vitamin D. Ramipril was added later on, after normalization of his serum creatinine level, and he was followed up periodically with his predni-solone dose gradually tapered off over the next 12 months. At his last follow-up visit, two years following the diagnosis of IgA nephropathy, he had significant improvement. There were no edema and his serum creatinine was in normal range, and urinary total protein was 0.39 g/24 h. He was on ramipril 5 mg Q 24 h, and without any antidiabetic treatment, his fasting and 2-h postbreakfast blood glucose values were 5.6 and 7.3 mmol/L, respectively. There was no evidence of recurrence of melioidosis.


   Discussion Top


Melioidosis is now well established as an endemic disease in Bangladesh but is almost certainly underdiagnosed. A recent modeling study estimated that it may account for nearly 17,000 cases and 9,500 deaths in Bangladesh each year.[3] Our patient, a male with urinary tract infection and epididymo-orchitis due to B. pseudomallei, adds further evidence of endemicity of melioidosis in Bangladesh.

Melioidosis can involve every body systems and organs including the genitourinary tract. Genitourinary involvement was reported in 3.2% and 14% of all melioidosis cases in two different series from Malaysia[4] and Australia,[16] respectively. Genitourinary infection due to B. pseudomallei has also been reported from Bangladesh[11] as well as from West Bengal[17] and Karnataka[18] in India. Like other forms of the disease, genitourinary melioidosis occurs more frequently in males with complications such as prostatitis, prostatic abscess, renal abscess, epididymo-orchitis, and septicemia.[11],[16],[17],[18],[19]

All forms of melioidosis, including genitourinary melioidosis, are common in patients with underlying conditions, particularly diabetes mellitus;[11],[19] occasionally, presentation with melioidosis may help to unmask undetected diabetes[19] as in our case. Other underlying conditions that have been associated with genitourinary melioidosis include alco-holism,[19] renal transplantation,[20] steroid thrapy,[16] and IgA nephropathy.[21] In many cases, there is a clear history of occupational or recreational exposure to the environment.[16],[19] Infection of the genitourinary tract with B. pseudomallei is thought to result through the hematogenous route[1] although local invasion through abraded skin by penetrating injury[18] or during coitus[22] is also possible. Being a farmer, our patient was at risk of occupational exposure to environmental B. pseudomallei. It is worth mentioning that melioidosis has been reported from Feni, his locality in Bangladesh, in recent years.[12]

The clinical presentation of melioidosis depends on site(s) and extent of involvement and severity of the disease. It often mimics tuberculosis both clinically and radiologically,[23] resulting in diagnostic delay, especially in areas where it is less well known. Fever and rigors are common in genitourinary cases.[11],[17],[18],[19] Dysuria, increased urinary frequency, and urinary retention may occur, and patients may present with septic shock.[17]

On laboratory investigation, neutrophil leukocytosis is common. Imaging has important role; a chest X-ray and abdominal ultrasonography or computed tomography should be undertaken to identify the site, extension, and nature of disease. It may help in localization of abscesses and guided aspiration.[8] B. pseudomallei grows readily on ordinary media from blood, urine, or pus (if present)[17] although special selective media increase the yield from sites with a normal flora,[24] and sometimes, extended incubations may be warranted. A high index of clinical suspicion, good liaison between the treating physicians and micro-biologists, and the familiarity of microbiologists with the culture and staining characteristics of B. pseudomallei are all of immense importance. In inexperienced hands, cultures may be discarded as contaminants.[17] Growth of Pseudomonas-like organisms which are resistant to aminoglycosides and polymyxin/ colistin and sensitive to co-amoxiclav is important clue for laboratory personnel.

Treatment consists of initial intravenous ceftazidime or carbapenems followed by extended period of oral antibiotics[25] to reduce the risk of relapse. Inappropriate treatment either in terms of inappropriate antibiotic or incorrect dose or duration may result in treatment failure or recurrence. The overall outcome of genitourinary melioidosis is variable and depends on the rapidity of initiation of appropriate treatment, the extent and severity of disease, and the presence of complications such as renal failure and septicemia.[19] However, with appropriate management, a complete cure is possible, as evidenced by our case.


   Conclusion Top


The increasing recognition of clinical cases along with seroepidemiological evidence and the isolation of B. pseudomallei from the environment confirm that melioidosis is endemic in Bangladesh as it is in neighboring countries such as India and Myanmar (formerly Burma). A high index of suspicion is necessary for identification of the condition. Melioidosis should be considered as a differential diagnosis in any one with fever, pneumonia, abscesses, or genitourinary infection, especially if there is underlying diabetes with a history of contact with soil and surface water and if not responding to the usual antibiotics prescribed for community-acquired infections.

Conflict of interest: None declared.

 
   References Top

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White NJ. Melioidosis. Lancet 2003;361:1715-22.  Back to cited text no. 1
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Wiersinga WJ, Currie BJ, Peacock SJ. Melioidosis. N Engl J Med 2012;367:1035-44.  Back to cited text no. 2
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Limmathurotsakul D, Golding N, Dance DA, et al. Predicted global distribution of Burkholderia pseudomallei and burden of melioidosis. Nat Microbiol 2016;1. pii: 15008.  Back to cited text no. 3
    
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Zueter A, Yean CY, Abumarzouq M, et al. The epidemiology and clinical spectrum of melioidosis in a teaching hospital in a NorthEastern state of Malaysia: A fifteen-year review. BMC Infect Dis 2016;16:333.  Back to cited text no. 4
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Currie BJ, Dance DA, Cheng AC. The global distribution of Burkholderia pseudomallei and melioidosis: An update. Trans R Soc Trop Med Hyg 2008;102 Suppl 1:S1-4.  Back to cited text no. 5
    
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Whitmore A, Krishnaswami CS. An account of the discovery of a hitherto undiscovered infective disease occurring among the population of Rangoon. Indian Med Gaz 1912;47:262-7.  Back to cited text no. 6
    
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Afroze SR, Rahman MR, Barai L, Hossain MD, Uddin KN. Successful treatment outcome of primary melioidosis pneumonia A case report from Bangladesh. BMC Res Notes 2016;9:100.  Back to cited text no. 7
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Saha DK, Ahsan AS, Faruq MO, Fatema K, Ahmed F, Saha M. Hepatic abscess as presenting feature of melioidosis: A case report. Birdem Med J 2016;6:43-5.  Back to cited text no. 8
    
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Fatema K, Ahsan AS, Barai L, et al. A case of fatal meliodosis presenting as septic arthritis and septicaemia. Bangladesh Crit Care J 2015; 3:74-6.  Back to cited text no. 9
    
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Rahim MA, Afroze SR, Barai L, Uddin KN. Melioidosis: Truly uncommon or uncommonly diagnosed in Bangladesh? A case report. Birdem Med J 2015;5 Suppl 1:49-51.  Back to cited text no. 10
    
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Barai L, Jilani MS, Haq JA. Melioidosis Case reports and review of cases reported among Bangladeshi population from 19882014. Ibrahim Med Coll J 2014;8:25-31.  Back to cited text no. 11
    
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Majumder MI, Haque MM, Ahmed MW, et al. Melioidosis in an adult male. Mymensingh Med J 2013;22:413-6.  Back to cited text no. 12
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Jilani MS, Robayet JA, Mohiuddin M, et al. Burkholderia pseudomallei: Its detection in soil and seroprevalence in Bangladesh. PLoS Negl Trop Dis 2016;10:e0004301.  Back to cited text no. 13
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Maude RR, Maude RJ, Ghose A, et al. Seroepidemiological surveillance of Burkholderia pseudomallei in Bangladesh. Trans R Soc Trop Med Hyg 2012;106:576-8.  Back to cited text no. 14
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Kidney Disease: Improving Global Outcomes (KDIGO) Glomerulonephritis Work Group. KDIGO clinical practice guideline for glomerulonephritis. Kidney Int Suppl 2012;2:139-274.  Back to cited text no. 15
    
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Currie BJ, Ward L, Cheng AC. The epidemiology and clinical spectrum of melioidosis: 540 cases from the 20 year Darwin prospective study. PLoS Negl Trop Dis 2010;4:e900.  Back to cited text no. 16
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Viswaroop BS, Balaji V, Mathai E, Kekre NS. Melioidosis presenting as genitourinary infection in two men with diabetes. J Postgrad Med 2007;53:108-10.  Back to cited text no. 17
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Naganathan K, Pillai SB, Kumar P, Hegde P. Whitmore's disease: An uncommon urological presentation. BMJ Case Rep 2014;2014. pii: bcr2013201978.  Back to cited text no. 18
    
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Chong Vh VH, Sharif F, Bickle I. Urogenital melioidosis: A review of clinical presentations, characteristic and outcomes. Med J Malaysia 2014;69:257-60.  Back to cited text no. 19
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Varughese S, Mohapatra A, Sahni R, Balaji V, Tamilarasi V. Renal allograft recipient with melioidosis of the urinary tract. Transpl Infect Dis 2011;13:95-6.  Back to cited text no. 20
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Northfield J, Whitty CJ, MacPhee IA. Burkholderia pseudomallei infection, or melioidosis, and nephrotic syndrome. Nephrol Dial Transplant 2002;17:137-9.  Back to cited text no. 21
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Webling DD. Genito-urinary infections with Pseudomonas pseudomallei in Australian aboriginals. Trans R Soc Trop Med Hyg 1980; 74:138-9.  Back to cited text no. 22
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Wuthiekanun V, Dance DA, Wattanagoon Y, et al. The use of selective media for the isolation of Pseudomonas pseudomallei in clinical practice. J Med Microbiol 1990;33: 121-6.  Back to cited text no. 24
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Dance D. Treatment and prophylaxis of melioidosis. Int J Antimicrob Agents 2014;43: 310-8.  Back to cited text no. 25
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Correspondence Address:
Dr. Muhammad Abdur Rahim
Department of Nephrology, Bangladesh Institute of Research and Rehabilitation in Diabetes, Endocrine and Metabolic Disorders General Hospital, Dhaka-1000
Bangladesh
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DOI: 10.4103/1319-2442.235205

PMID: 29970750

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