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Renal Failure, 2013; 35(2): 302304 Copyright © Informa Healthcare USA, Inc. ISSN 0886-022X print/1525-6049 online DOI: 10.3109/0886022X.2012.743915 CASE REPORT Renal Cyst Infection Caused by Brucella abortus Serkan Öncü 1 , Özlem Saylak 1 , Kutsi Köseoğlu 2 , Barçın Öztürk 1 , AyşeEşiyok 1 , Esra Çörekli 1 and Serhan Sakarya 1 1 Department of Infectious Diseases and Clinical Microbiology, Adnan Menderes University Medical Faculty, Aydin, Turkey; 2 Department of Radiology, Adnan Menderes University Medical Faculty, Aydin, Turkey Abstract A 49-year-old man with a medical history of polycystic kidney disease was presented to the emergency department with fever and left flank pain. Abdominal examination revealed an enlarged and painful left kidney. The C-reactive protein level was significantly high and the magnetic resonance imaging revealed areas of abnormal intensity and fluidfluid levels in renal cysts. Brucella abortus was yielded from both blood and cyst fluid culture. Standard therapy (rifampicin plus doxycycline) of brucellosis was started, but the clinical and laboratory signs subsided after the addition of ciprofloxacin. There was no need for aspiration of infected cyst fluid. Hereby, according to the medical database search, we report that the first renal cyst infection caused by B. abortus was successfully treated with triple antibiotic therapy. Keywords: renal cyst, infection, Brucella, ciprofloxacin INTRODUCTION Polycystic kidney disease (PKD) is one of the most common inherited renal disorders. 1 The most remark- able characteristic of PKD is the occurrence of numer- ous renal cysts, resulting in enlargement of the kidneys. It affects one in 5001000 individuals and accounts for 410% of end-stage renal disease. 2 Patients with PKD sometimes develop complications (such as infection and bleeding). Among these complications, cyst infec- tions are relatively infrequent but are associated with serious outcomes. 3 Fever and abdominal pain are the main symptoms of cyst infection, but radiological and laboratory assessment is also required for the definite diagnosis. Pathogens are typically bowel flora such as Escherichia coli and retrograde infection of renal cysts via the ureter from the bladder is the main route of infec- tion. 2 However, unusual pathogens reported as the etio- logical agent in cyst infection suggest alternate route in infection. 3,4 Debate is also present for the treatment of cyst infection. Antibiotics with well penetrating capabil- ity have brought new treatment modality into such infections. Here we describe a case of renal cyst infec- tion caused by Brucella abortus, which was successfully treated with antibiotics. CASE REPORT A 49-year-old man with a medical history of PKD was presented to the emergency department with 10 days of fever and left flank pain. Except for a family history of PKD, there was no other remarkable information in his medical history. On admission, his vital signs were as follows: temperature 39 C, blood pressure 140/ 80 mm Hg, and pulse rate 75 beats/min. Abdominal examination revealed hepatomegaly and enlarged kid- neys. The flank pain was exacerbated by the palpation of left kidney. The results of the remainder of the physical examination were unremarkable. Laboratory studies showed hemoglobin 12.8 g/dL, white blood cells (WBC) 6270/μL, platelet count 254,000/μL, blood urea nitrogen 52 mg/dL, creatinine 1.27 mg/dL, aspartate aminotransferase (AST) 23 IU/L, alanine aminotransfer- ase (ALT) 29 IU/L, alkaline phosphatase (ALP) 46 IU/L, gamma-glutamyl transferase (GGT) 48 IU/L, lactate dehydrogenase (LDH) 219 IU/L, total protein 6.3 mg/ dL, albumin 3.1 mg/dL, C-reactive protein (CRP) 208 mg/L, and erythrocyte sedimentation rate (ESR) 68 mm/h. The urine sediment was normal. According to medical history, clinical presentation, and laboratory results cyst infection was the initial diagnosis. High-CRP Address correspondence to Serkan Öncü, Department of Infectious Diseases and Clinical Microbiology, Adnan Menderes University Medical Faculty, Aydin 09100, Turkey. Tel.: þ90-530-7776188; Fax: þ90-256-2144086; E-mail: [email protected] Received 25 September 2012; Revised 15 October 2012; Accepted 16 October 2012 302 Ren Fail Downloaded from informahealthcare.com by University of Bath on 11/09/14 For personal use only.

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Page 1: Renal Cyst Infection Caused by               Brucella abortus

Renal Failure, 2013; 35(2): 302–304Copyright © Informa Healthcare USA, Inc.ISSN 0886-022X print/1525-6049 onlineDOI: 10.3109/0886022X.2012.743915

CASE REPORT

Renal Cyst Infection Caused by Brucella abortus

Serkan Öncü1, Özlem Saylak1, Kutsi Köseoğlu2, Barçın Öztürk1, Ayşe Eşiyok1, Esra Çörekli1

and Serhan Sakarya1

1Department of Infectious Diseases and Clinical Microbiology, Adnan Menderes University Medical Faculty, Aydin, Turkey;2Department of Radiology, Adnan Menderes University Medical Faculty, Aydin, Turkey

Abstract

A 49-year-old man with a medical history of polycystic kidney disease was presented to the emergency department withfever and left flank pain. Abdominal examination revealed an enlarged and painful left kidney. The C-reactive protein levelwas significantly high and the magnetic resonance imaging revealed areas of abnormal intensity and fluid–fluid levels inrenal cysts. Brucella abortus was yielded from both blood and cyst fluid culture. Standard therapy (rifampicin plusdoxycycline) of brucellosis was started, but the clinical and laboratory signs subsided after the addition of ciprofloxacin.There was no need for aspiration of infected cyst fluid. Hereby, according to the medical database search, we report thatthe first renal cyst infection caused by B. abortus was successfully treated with triple antibiotic therapy.

Keywords: renal cyst, infection, Brucella, ciprofloxacin

INTRODUCTION

Polycystic kidney disease (PKD) is one of the mostcommon inherited renal disorders.1 The most remark-able characteristic of PKD is the occurrence of numer-ous renal cysts, resulting in enlargement of the kidneys.It affects one in 500–1000 individuals and accounts for4–10% of end-stage renal disease.2 Patients with PKDsometimes develop complications (such as infectionand bleeding). Among these complications, cyst infec-tions are relatively infrequent but are associated withserious outcomes.3 Fever and abdominal pain are themain symptoms of cyst infection, but radiological andlaboratory assessment is also required for the definitediagnosis. Pathogens are typically bowel flora such asEscherichia coli and retrograde infection of renal cysts viathe ureter from the bladder is the main route of infec-tion.2 However, unusual pathogens reported as the etio-logical agent in cyst infection suggest alternate route ininfection.3,4 Debate is also present for the treatment ofcyst infection. Antibiotics with well penetrating capabil-ity have brought new treatment modality into suchinfections. Here we describe a case of renal cyst infec-tion caused by Brucella abortus, which was successfullytreated with antibiotics.

CASE REPORT

A 49-year-old man with a medical history of PKD waspresented to the emergency department with 10 days offever and left flank pain. Except for a family history ofPKD, there was no other remarkable information in hismedical history. On admission, his vital signs were asfollows: temperature 39�C, blood pressure 140/80 mm Hg, and pulse rate 75 beats/min. Abdominalexamination revealed hepatomegaly and enlarged kid-neys. The flank pain was exacerbated by the palpationof left kidney. The results of the remainder of the physicalexamination were unremarkable. Laboratory studiesshowed hemoglobin 12.8 g/dL, white blood cells(WBC) 6270/μL, platelet count 254,000/μL, blood ureanitrogen 52 mg/dL, creatinine 1.27 mg/dL, aspartateaminotransferase (AST) 23 IU/L, alanine aminotransfer-ase (ALT) 29 IU/L, alkaline phosphatase (ALP) 46 IU/L,gamma-glutamyl transferase (GGT) 48 IU/L, lactatedehydrogenase (LDH) 219 IU/L, total protein 6.3 mg/dL, albumin 3.1 mg/dL, C-reactive protein (CRP)208 mg/L, and erythrocyte sedimentation rate (ESR)68 mm/h. The urine sediment was normal. Accordingto medical history, clinical presentation, and laboratoryresults cyst infection was the initial diagnosis. High-CRP

Address correspondence to Serkan Öncü, Department of Infectious Diseases and Clinical Microbiology, Adnan Menderes UniversityMedical Faculty, Aydin 09100, Turkey. Tel.: þ90-530-7776188; Fax: þ90-256-2144086; E-mail: [email protected]

Received 25 September 2012; Revised 15 October 2012; Accepted 16 October 2012

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value was in favor of cyst infection. Abdominal magneticresonance imaging (MRI) was also ordered to differenti-ate the diagnosis. MRI showed thick-walled, heteroge-neous cysts slightly hyperintense on T1-weighted MRimages and with peripheral enhancement after gadoli-nium injection (Figure 1). A cyst in the left kidney witha fluid–fluid level was also suggestive of infected cyst onT2-weighted image (Figure 2). On the diagnosis of cystinfection, empirical antibiotic with intravenous (IV) cef-triaxone (2 g/24 h) was prescribed after obtaining urineand blood cultures. The urine culture showed no bacter-ial growth and the patient remained febrile with the anti-biotic treatment. On the sixth day of the treatment, bloodculture yielded B. abortus. Wright agglutination test wasalso detected positive for 1/320 titers. Requestioning thepatient history revealed the consumption of unpasteur-ized milk and cheese. The treatment regimen was chan-ged to doxycycline (100 mg/12 h) plus rifampicin(900 mg/day). Patient’s symptoms were continued and

the CRP values did not decline despite the 10 days oftreatment with modified antibiotic regimen. Anultrasound-guided needle aspiration fluid was obtainedfor etiological diagnosis of cyst infection, and B. abortuswas also isolated from the specimen. Upon this IV, cipro-floxacin (400 mg/12 h) was added to the treatment regi-men. After a couple of days with this triple antibiotictherapy, the symptoms subsided and theCRP levels startedto decrease. At the end of the second week of efficienttherapy, clinical and laboratory response was completelyachieved. At this point, the patient was discharged from thehospital and the treatment was continued with oral anti-biotics until the end of the sixth week of the therapy.

DISCUSSION

Cyst infection is a well-recognized complication of PKDand has been reported to be the second most commoncause of death in this group of patients.5 Many micro-organisms have been reported as the etiological agent butdigestive tract microorganisms especially E. coli accountsfor the majority.6 In this case, the cause of cyst infectionwas B. abortus. Brucella spp. is an intracellular Gram-negative bacterium and is the causative agent of brucel-losis. Brucellosis is a zoonotic infection transmitted tohumans by contact with fluids from infected animals(sheep, cattle, goats, or other animals) or derived foodproducts such as unpasteurized milk and cheese. It hashigh morbidity for humans and is an important cause ofpublic health problems in many developing countries.Almost every organ system involvement has beenreported with Brucella spp., but the renal cyst infectionwas somewhat novel. The available databases weresearched for the association of Brucella spp. and renalcyst infection. “Brucella” and “PKD” were the searchterms. There was no search result and it seems thatrenal cyst infection due to Brucella spp. would be thefirst report. Accordingly, this case reveals that infectingagents other than bowel flora may be the causative agentof renal cyst infection. The involvement of renal cyst maybe a part of the systemic illness caused by such micro-organisms, but the marked symptoms will possibly focusthe physician to cyst infection with a resulting inap-propriate antibiotic prescription. This points that, micro-organisms such as Brucella spp. should be held indifferential diagnosis of cyst infection in patient living inendemic areas. Ascending route through urinary tract isthe most accepted concept for the development of cystinfection. Consequently, microorganism isolated fromthe urinary tract is commonly recognized as the respon-sible agent. But, the present and some reported casesshow that infection paths other than urinary tract arepresent.4 After ingestion or inoculation, Brucella spp.invade the mucosa, where polymorphonuclear leuko-cytes and activated macrophages mediate immuneresponses to eradicate the bacteria. However, Brucellacan multiply and survive intracellularly.7 They are then

Figure 2. T2-weighted image shows fluid–fluid level (arrow) in thecyst that is located in the upper pole of the left kidney.

Figure 1. Postgadolinium T1-weighted image shows slightlyhyperintense cyst (arrow) with peripheral contrast enhancement.

© 2013 Informa Healthcare USA, Inc.

Renal Cyst Infection Caused by Brucella abortus 303

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transported intracellularly via the lymphatics to organsrich in reticuloendothelial cells, and from there travel toother organs and tissues, where they can cause infection.Accordingly, although the precise infectious route wasunidentified, hematogenous seeding of the cyst was themost probable way of infection. Hence, cyst fluid aspira-tion should be preferred for culture in patients withoutobvious urinary infection findings (such as dysuria, pol-lakiuria, and pyuria) and for those nonresponding toantibiotic therapy. The treatment of renal cyst infectionis often difficult and there is a high treatment failure ratesdespite prolonged appropriate antibiotic therapy.Percutaneous or surgical drainage of the infected cyst isoften an applied procedure in such cases to avoid a fataloutcome.8 But some antibiotics with their high penetrat-ing capability to renal cysts have raised a noninvasivetreatment option in such infections. Intracystic antibioticdiffusion and accumulation vary widely and probablydepend on passive diffusion as well as active transepithe-lial transport. Among the antibiotics, quinolones areusually favored because of their lipophilic propertiesthat lead to an increased diffusion in infected cysts andtheir bactericidal activity.9,10 But the problem with qui-nolones is the growing resistance to these antibiotics.The frequent and inappropriate prescription of quino-lones to community-based patients has raised the pro-blem to current situation.11 In areas faced with this fact,the empirical antibiotic preference for urinary tract infec-tion has changed from quinolones to β-lactams (such asbroad-spectrum cephalosporins). This was the reason forinitial ceftriaxone therapy in our case. Nevertheless,according to the available data, quinolones seem to bethe best option for cyst infection and precedence shouldbe given to these drugs after establishing the antibioticsusceptibility. This approach may also be feasible forthe treatment of cyst infections caused by non-Enterobacteriaceae microorganisms like Brucella spp.The recommended first-line therapy for brucellosisincludes streptomycin or doxycycline in combinationwith rifampicin.12 Although there are some cases serieslyreporting successful outcomes with quinolone includingregimen, it is not supported by randomized clinical stu-dies.13,14 Even so, the long duration of unresponsivenessto doxycycline plus rifampicin pushed us also to addciprofloxacin to the treatment according to the availableantibiotic properties and reported case series. However,the success achieved with the applied therapy should be

supported by randomized studies to recommend quino-lones in the first-line therapy for cyst infections.

Declaration of interest: The authors report no con-flicts of interest. The authors alone are responsible for thecontent and writing of the article.

REFERENCES

[1] Sklar AH, Caruana RJ, Lammers JE, Strauser GD. Renal infec-tions in autosomal dominant polycystic kidney disease. Am JKidney Dis. 1987;10:81–88.

[2] SalleeM, Rafat C, Zahar JR, et al. Cyst infections in patients withautosomal dominant polycystic kidney disease. Clin J Am SocNephrol. 2009;4:1183–1189.

[3] Tsuchiya Y, Ubara Y, Suwabe T, et al. The renal cyst infectioncaused by Salmonella enteritidis in a patient with autosomaldominant polycystic kidney disease: how did this pathogencome into the renal cysts? Clin Exp Nephrol. 2011;15:151–153.

[4] Yang CC, Chuang FR, Chen JB, Lee CT, Wu CH. A potentialsalvage therapy for refractory renal cyst infection in patients withautosomal dominant polycystic kidney disease.Clin Exp Nephrol.2012;16:183–184.

[5] Fick GM, Johnson AM, Hammond WS, Gabow PA. Causes ofdeath in autosomal dominant polycystic kidney disease. J AmSocNephrol. 1995;5:2048–2056.

[6] Migali G, Annet L, LonneuxM,Devuyst O. Renal cyst infectionin autosomal dominant polycystic kidney disease. Nephrol DialTransplant. 2008;23:404–405.

[7] Fugier E, Pappas G, Gorvel JP. Virulence factors in brucellosis:implications for aetiopathogenesis and treatment.Expert RevMolMed. 2007;9:1–10.

[8] Torres VE, Harris PC. Autosomal dominant polycystic kidneydisease: the last 3 years. Kidney Int. 2009;76:149–168.

[9] Elzinga LW, Golper TA, Rashad AL, Carr ME, Bennett WM.Ciprofloxacin activity in cyst fluid from polycystic kidneys.Antimicrob Agents Chemother. 1988;32:844–847.

[10] Hiyama L, Tang A, Miller LG. Levofloxacin penetration into arenal cyst in a patient with autosomal dominant polycystic kidneydisease. Am J Kidney Dis. 2006;47:e9–e13.

[11] Kurtaran B, Candevir A, Tasova Y, et al. Antibiotic resistance incommunity-acquired urinary tract infections: prevalence andrisk factors. Med Sci Monit. 2010;16:CR246–CR251.

[12] Bossi P, Tegnell A, Baka A, et al. Bichat guidelines for theclinicalmanagement of brucellosis and bioterrorism-related bru-cellosis. Euro Surveill. 2004;9:E15–E16.

[13] Falagas ME, Bliziotis IA. Quinolones for treatment of humanbrucellosis: critical review of the evidence from microbiologi-cal and clinical studies. Antimicrob Agents Chemother.2006;50:22–33.

[14] Solis Garcia del Pozo J, Solera J. Systematic review and meta-analysis of randomized clinical trials in the treatment of humanbrucellosis. PLoS One. 2012;7:e32090.

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