Metastatic intracranial subdural empyema from renal cyst infection in autosomal dominant polycystic kidney disease

Hiroshi Nishi1, Yugo Shibagaki1, Shuji Hatakeyama2, Takafumi Ito3, Taiji Nagata4, Minoru Ohno5, Akihiro Tojo1, Masaomi Nangaku1 and Toshiro Fujita1

1 Department of Nephrology and Endocrinology, 2 Infectious Diseases, 3 General Internal Medicine, 4 Respiratory Medicine and 5 Cardiovascular Medicine, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan

Correspondence and offprint requests to: Yugo Shibagaki, Department of Nephrology and Endocrinology, Graduate School of Medicine, The University of Tokyo, 7-3-1 Hongo, Bunkyo-ku, Tokyo 113-8655, Japan. Email: eugo{at}wc4.so-net.ne.jp

Keywords: autosomal dominant polycystic kidney disease (ADPKD); cyst infection; Escherichia coli (E.coli); pulse-field gel electrophoresis (PFGE); subdural empyema



   Introduction
 Top
 Introduction
 Case
 Discussion
 References
 
Approximately 30–50% of patients with autosomal dominant polycystic kidney disease (ADPKD) develop renal infection [1]. Cyst infection and acute pyelonephritis are the most common infectious complications, although bacteraemia, perinephric and extrarenal abscess can also be seen. The problem is that eradication of cyst infections with conventional antibiotics is sometimes unsuccessful chiefly because of poor penetration of antibiotics into infected cysts. As a result, a significant portion of patients with ADPKD suffer from latent cyst infection, which may result in bacteraemia and/or metastatic extrarenal infection.

We describe here a case of ADPKD whose intracranial subdural empyema indicated a metastatic abscess from an uncontrolled cyst infection accompanying ADPKD.



   Case
 Top
 Introduction
 Case
 Discussion
 References
 
A 76-year-old woman was admitted for sudden onset of fever and altered mental status in June 2005. The patient had developed progressive renal dysfunction due to ADPKD since 1991. Although her renal function deteriorated to end-stage renal disease, she refused to start dialysis. She had repetitive episodes of renal cyst infection, for which she was hospitalized and treated with ciprofloxacin, only resulting in suboptimal improvement.

On admission, she was disoriented, with body temperature of 39.2°C, blood pressure of 164/100 mmHg, and heart rate of 160 due to paroxysmal supraventricular tachycardia. Physical examination revealed no meningeal signs. Her abdominal exam was normal except for mild right cost-vertebral angle (CVA) tenderness. Abnormal laboratory findings included white blood cell (WBC) count of 15 800 x 103/µl, haemoglobin of 9.6 g/dl, urea nitrogen of 57.2 mg/dl, creatinine of 5.14 mg/dl and bicarbonate of 17.2 mEq/l. Urinary analysis showed protein 3+ by dipstick; red blood cell of 51–100/high power field (hpf), WBC of 11–20/hpf, and granular casts 1+ by microscopy. The renal indices did not differ from those of previous tests and thus uraemic encephalopathy seemed unlikely. Blood culture was negative, but trace amounts of Escherichia coli were isolated in urine culture. Lumbar puncture revealed clear cerebrospinal fluid with a cell count of 12/mm3 and a negative culture test, indicating the absence of a demonstrable association with severe bacterial meningitis. Abdominal computed tomography (CT) revealed inflamed fatty tissues peripheral to the lower pole of the right kidney, suggesting the presence of an active cyst infection (Figure 1A). Head CT showed a left-sided crescent-shaped intracranial subdural fluid collection (Figure 2A), while no other abnormalities in the skull or sinuses were pointed out. She quickly lost the ability to articulate, open her eyes spontaneously or obey commands. Subsequently, CT scan revealed an enlargement of the intracranial fluid collection with mass effect. She underwent urgent surgery, and a brownish yellow fluid was drained from the subdural space. The fluid neither included haematoma nor bloody components. Colonies of Gram-negative rods phagocytosed by large amounts of leucocytes were identified by microscopy, and E.coli were isolated from the drained pus culture.



View larger version (92K):
[in this window]
[in a new window]
 
Fig. 1. (A) Plain abdominal CT on admission revealed irregular-shaped inflamed fatty tissues around the lower pole of polycystic right kidney as high density area (arrow). This image indicated an active inflammation of the right kidney. (B) Inflamed fatty tissue disappeared on the follow up CT on the 15th day after admission.

 


View larger version (48K):
[in this window]
[in a new window]
 
Fig. 2. (A) Plain cranial CT on admission revealed a left-sided marked subdural effusion with mass effect (arrow). (B) Subdural effusion was not noted on the follow up CT on the 60th day after admission. The defect of the skull was formed at burr hole craniotomy.

 
After the patient was treated with ceftriaxone and phenytoin, her mental status gradually improved, to an extent where she was able to articulate and obey commands. On a head CT series, the subdural fluid collection decreased in size (Figure 2B), and inflammatory change of perirenal fatty tissue diminished (Figure 1B) as did bacteuria and pyuria. Her renal function, however, deteriorated progressively, and she started haemodialysis on a regular basis.

To determine whether E.coli isolated from the drained pus was identical to that isolated from the urine on admission, we performed two different analyses of these strains. First, in vitro microbiology sensitivity test demonstrated that both strains showed precisely the same minimum inhibitory concentration (MIC) against 15 different antibiotics. Second, after digestion of chromosomal DNA of both strains with restriction endonuclease, Xba1, restriction products were analysed by pulse-field gel electrophoresis (PFGE). PFGE profiles were analysed using GelCompar II software (Applied Maths BVBA, Belgium) and interpreted according to the guideline published previously [2]. To compare restriction patterns, we calculated percentage similarities with the Jaccard coefficient by the unweighted pair-group method using arithmetic averages. As a result, PFGE revealed indistinguishable Xba1 restriction patterns of both strains (Figure 3). These data demonstrated that the E.coli strain isolated from the drained pus was most probably identical from that isolated from the urine on admission.



View larger version (54K):
[in this window]
[in a new window]
 
Fig. 3. Pulse-field gel electrophoresis (PFGE) patterns were shown with percentage similarities for E.coli isolates from drained pus and from urine on admission. The PFGE patterns of both isolate were identified as indistinguishable using the 0.4% tolerance. 1, drained pus from subdural empyema; 2, urine; M, lambda ladder markers; C, control E.coli strain ATCC11775.

 


   Discussion
 Top
 Introduction
 Case
 Discussion
 References
 
Intracranial subdural empyema represents a closed infection between the outermost layer of the meninges, the dura, and the arachnoid. This intracranial infection is most frequently a complication of sinusitis, otitis or neurosurgical procedures [3]. However, in this case, these origins of subdural empyema were denied by history, physical examination and imaging studies. In addition, isolation of E.coli from drained empyema has been very rarely reported [4,5], which strongly suggested that the empyema was a metastasis from a sub-diaphragmatic site. Meanwhile, renal cyst infection is quite common in ADPKD but includes diagnostic problems [6]. In the present case, the patient had repetitive episodes of renal cyst infection and presented on admission with right CVA tenderness with pyuria and inflamed fatty tissue around the right cystic kidney on abdominal CT which indicated latent renal cyst infection. The fact that two strains of E.coli isolated from urine and empyema showed precisely the same antibiotic sensitivity strongly supported that the empyema was a metastasic renal cyst infection.

Furthermore, we performed PFGE; a well-established molecular sub-typing method in discriminating among bacterial strains. This method has been generally used for surveillance of bacterial outbreak, but recently it has been applied to analyse whether a particular strain was responsible for separate infections in the same individual [7]. The results clarified that in our case one particular E.coli strain was responsible for both the urinary tract infection and the extrarenal infections.

Bacteraemia or extrarenal abscess metastasized from urinary tract infections are potential complications of ADPKD. To our knowledge, however, there have been very few case reports of renal cystic infection associated with extrarenal infection [8] and this is the first case report of an empyema associated with renal cyst infection.

Advanced age and the female sex, as in this case, have been identified as possible predisposing factors of renal infection in ADPKD [9]. Renal cyst infection is notorious for its resistance to antibiotics. In our case, ciprofloxacin was administered repeatedly for renal cyst infection prior to this admission, but apparently this failed to eradicate the pathogen.

In summary, we report a case of ADPKD complicated with intracranial subdural empyema caused by E.coli. The link between these remote infections was confirmed by clinical pharmacological and molecular sub-typing methods of the responsible microorganism and suggest that the empyema was a metastatic renal infection. This case calls for the need for adequate control of urinary tract infections in ADPKD patients to avoid fatal metastatic infection.

Conflict of interest statement. None declared.



   References
 Top
 Introduction
 Case
 Discussion
 References
 

  1. Gabow PA, Bennett WM. Renal manifestations: complication management and long-term outcome of autosomal dominant polycystic kidney disease. Semin Nephrol 1991; 11: 643–652[ISI][Medline]
  2. Tenover FC, Arbeit RD, Goering RV et al. Interpreting chromosomal DNA restriction patterns produced by pulsed-field gel electrophoresis: criteria for bacterial strain typing. J Clin Microbiol 1995; 33: 2233–2239[Free Full Text]
  3. Bernardini GL. Diagnosis and management of brain abscess and subdural empyema. Curr Neurol Neurosci Rep 2004; 4: 448–456[Medline]
  4. Bakker S, Kluytmans J, den Hollander JC, Lie ST. Subdural empyema caused by Escherichia coli: hematogenous dissemination to a preexisting chronic subdural hematoma. Clin Infect Dis 1995; 21: 458–459[ISI][Medline]
  5. Miedema CJ, Kimpen JL. Hematogenous dissemination of Escherichia coli to a preexistent subdural hematoma in a child. Clin Infect Dis 1996; 23: 662[ISI][Medline]
  6. Gibson P, Watson ML. Cyst infection in polycystic kidney disease: a clinical challenge. Nephrol Dial Transplant 1998; 13: 2455–2457[Free Full Text]
  7. Chu VH, Sexton DJ, Cabell CH et al. Repeat infective endocarditis: differentiating relapse from reinfection. Clin Infect Dis 2005; 41: 406–409[CrossRef][ISI][Medline]
  8. Fuse H, Ohkawa M, Asamoto T. Infected renal cystic mass associated with bacterial meningitis: a case report. Int J Urol 1996; 3: 301–303[Medline]
  9. Sklar AH, Caruana RJ, Lammers JE, Strauser GD. Renal infections in autosomal dominant polycystic kidney disease. Am J Kidney Dis 1987; 10: 81–88[ISI][Medline]
Received for publication: 16. 8.05
Accepted in revised form: 6. 9.05





This Article
Extract
Full Text (PDF)
All Versions of this Article:
20/12/2820    most recent
gfi183v1
Alert me when this article is cited
Alert me if a correction is posted
Services
Email this article to a friend
Similar articles in this journal
Similar articles in ISI Web of Science
Similar articles in PubMed
Alert me to new issues of the journal
Add to My Personal Archive
Download to citation manager
Disclaimer
Request Permissions
Google Scholar
Articles by Nishi, H.
Articles by Fujita, T.
PubMed
PubMed Citation
Articles by Nishi, H.
Articles by Fujita, T.