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Taiwan Journal of Ophthalmology 3 (2013) 45e48 Contents lists available at SciVerse ScienceDirect Taiwan Journal of Ophthalmology journal homepage: www.e-tjo.com Case report Endogenous Serratia marcescens endophthalmitis associated with Port-a-Cath in an oncology patient: A case report and review of the literature Yi-Chieh Poon, Hsi-Kung Kuo* Department of Ophthalmology, Kaohsiung Chang Gung Memorial Hospital and Chang Gung University College of Medicine, Kaohsiung, Taiwan a r t i c l e i n f o a b s t r a c t Article history: Received 3 February 2012 Received in revised form 27 June 2012 Accepted 9 July 2012 Available online 20 August 2012 A 47-year-old woman with gum cancer was diagnosed with acute endogenous endophthalmitis. Serratia marcescens was cultured from the vitreous and blood samples. A venous access port for chemotherapy was the only identifiable source of infection. After receiving systemic antibiotics therapy, the patient recovered from the bacteremia but lost her vision as a result of Serratia endophthalmitis. Copyright Ó 2012, The Ophthalmologic Society of Taiwan. Published by Elsevier Taiwan LLC. All rights reserved. Keywords: endogenous endophthalmitis Port-a-Cath Serratia marcescens 1. Introduction Endogenous bacterial endophthalmitis is a serious and rare complication of bacteremia that occurs with an incidence rate of 0.2%.1 It develops when pathogens reach the eye via the bloodstream, and cross the blood-ocular barrier to cause intraocular infection. Risk factors for developing endogenous bacterial endophthalmitis include diabetes mellitus, malignancy, intravenous drug use, and immunodeficient state. Common causative bacteria differ according to geographic regions. In the non-East Asian countries, the most common pathogen is Gram-positive bacteria, such as Staphylococcus aureus and the Streptococcus species; while in the East Asian countries, Klebsiella pneumoniae, a Gram-negative bacteria is most common.2 In this case report, we will present a case of endogenous bacterial endophthalmitis caused by an unusual intraocular pathogen. 2. Case report A 47-year-old woman presented to our emergency department with acute onset of right eye pain, blurred vision, and tearing for 1 day. She had just been discharged from our hospital after receiving chemotherapy, 1 week prior to her return to the emergency * Corresponding author. Department of Ophthalmology, Kaohsiung Chang Gung Memorial Hospital, Number 123, Ta-Pei Road, Niao-Sung District, Kaohsiung City 833, Taiwan. E-mail address: [email protected] (H.-K. Kuo). department due to eye pain. The patient had a low-grade fever but no other systemic discomforts. The patient’s medical history was significant for having type 2 diabetes mellitus for more than 10 years, and gum cancer which was diagnosed 2 years earlier. The patient had been receiving concurrent chemoradiotherapy for gum cancer ever since its diagnosis. Chemotherapy was administrated through an implantable venous access device (Port-a-Cath) that was implanted subcutaneously below her left clavicle (Fig. 1). Upon examination at the emergency department, the patient was only able to see hand motion at 30 cm away, and had an intraocular pressure (IOP) of 27 mmHg in her right eye. Slit-lamp examination showed that the cornea was edematous and was coated on the endothelial side with keratic precipitates, and that there were hypopyon and dense fibrinous exudates in the anterior chamber of the eye. Examination of the fundus was obscured by the intense inflammatory reaction in the anterior chamber and the vitreous cavity. B-scan ultrasonography of the right eye revealed dense and heterogenous opacities in the vitreous cavity (Fig. 2). Orbital computed tomography image showed prominent periocular fat stranding and diffuse scleral thickening (Fig. 3). The diagnosis of endogenous endophthalmitis was made based on the above findings, and the patient was given a combination of vancomycin, amikacin, and dexamethasone injected intravitreally into the eye. Vitreous sample obtained on that day yielded the growth of Serratia marcescens, while the blood samples drawn from the peripheral blood were negative for bacterial growth. The patient was promptly admitted into the hospital for intravenous antibiotic therapy, given through a peripheral venous 2211-5056/$ e see front matter Copyright Ó 2012, The Ophthalmologic Society of Taiwan. Published by Elsevier Taiwan LLC. All rights reserved. http://dx.doi.org/10.1016/j.tjo.2012.07.002 46 Y.-C. Poon, H.-K. Kuo / Taiwan Journal of Ophthalmology 3 (2013) 45e48 Fig. 3. Computed tomography of the orbit showed extensive periocular fat stranding and diffuse scleral thickening in the right eye. Fig. 1. Chest X-ray revealed that the patient had an implanted Port-a-Cath. catheter. A broad-spectrum antibiotic, cefepime, was initially used, but was later switched to ceftriaxone according to drug sensitivity results from the bacterial cultures. Ultrasonography of the liver, cardioechography, urinalysis, and chest X-ray for infection survey were performed, but all yielded negative results. During hospitalization, the patient’s ocular inflammation signs remained stationary, but she continued to have low-grade fever of 37.7e37.9 C. Then, during the third week of hospitalization, the patient suddenly had a high fever of 39 C that was accompanied by chills and local tenderness near the site of the implanted Port-aCath. Coincidentally, on the same day, the patient had just switched from the use of a peripheral venous catheter line to the use of the Port-a-Cath for the administration of antibiotic therapy. Because of high fever, blood cultures were obtained, and both samples from the Port-a-Cath and the peripheral catheter yielded Fig. 2. B-scan showed that there were dense vitreous opacities. S. marcescens. Despite the high suspicion for Port-a-Cath-related infection, the patient needed this venous access port for chemotherapy, thus the decision was made not to remove it for tip culture. Systemic antibiotic was switched to ceftazidime because of persistent Serratia bacteremia and that this was a catheter-related bloodstream infection. The patient received a total of 6 weeks of systemic antibiotic therapy, and was discharged from the hospital after subsequent blood cultures no longer yielded bacterial growth. Despite the improvements in her systemic conditions and ocular inflammation, the patient’s right eye vision eventually progressed to no light perception, and phthisis bulbi developed. 3. Discussion S. marcescens, an aerobic, Gram-negative bacilli, has become an increasingly common cause of hospital-acquired infections worldwide.3e5 It can cause a variety of diseases, including respiratory tract infections, urinary tract infections, bacteremia, wound infections, keratoconjunctivitis, and meningitis.6 Outbreaks of S. marcescens bacteremia in the nosocomial setting has been associated with high mortality rates of 25.4e32%,4,5 and have been linked to contaminated prefilled syringes and drug vials,7,8 multiple dose usage for single-use drug vials,9 and inappropriate aseptic techniques of the healthcare personnels.10 Endogenous endophthalmitis caused by S. marcescens is rare. Based on our literature search, fewer than 10 well-documented cases (not including cases written in languages other than in English) have been reported to date (Table 1).6,11e19 Endogenous Serratia endophthalmitis has been associated with intravenous drug therapy, general surgery, and the use of indwelling catheters.6 Additional risk factors include diabetes mellitus, malignancy, and immunocompromised state. S. marcescens can be highly destructive to the eye, owing to its ability to secrete proteases that cause the loss of proteoglycan ground substances which then lead to the dispersal of collagen fibrils, liquefactive necrosis, and subsequently, ocular perforation.6 Endogenous Serratia endophthalmitis often leads to very poor visual outcomes. In their review, Equi and Green6 found that nearly Y.-C. Poon, H.-K. Kuo / Taiwan Journal of Ophthalmology 3 (2013) 45e48 47 Table 1 Clinical characteristics of S. marcescens endogenous endophthalmitis in our case and previous reports. Author (reference) Age, y Sex Comorbidity or risk factors Source of positive bacterial isolates Intravitreal antibiotics Systemic antibiotics Final outcome Gammon et al.11 60 Female Aqueous, urine None Gentamicin/amikin Death Radda12 59 Male Vitreous None Amoxicillin/carbenicillin and gentamicin/ticarcillin/ cefotaxime Evisceration de Courten et al.13 10 days male Craniotomy for intercavernous aneurysm, indwelling urinary catheter Chronic lymphatic leukemia, diabetes mellitus, transurethral electroresection of prostate for prostate adenoma, hemodialysis, Colostomy for unperforated anus Vitreous, blood None Enucleation Alverez et al14 32 male HIV infection, intravenous drug user Vitreous Tobramycin, vancomycin Al Hazzaa et al.15 3 days Umbilical artery catheter Blood, catheter tip, aqueous Renal abscess pus culture Vitreous, aqueous humor, blood culture Vitreous, blood, catheter tip Gentamicin, cefazolin Vancomycin, amikin Ceftazidime, vancomycin Vancomycin, gentamicin Ceflatonin and ampicillin/ ampicillin and amikin Ceftazidime, amikin, and vancomycin/ceftriaxone and amikin Gentamicin Cefazolin/pefloxacin/ ceftizoxime Piperacillin, vancomycin, and gentamicin/cefotaxime Vancomycin and gentamicin/aztreonam No light perception Enucleation Blood, sputum, Ceftazidime, vancomycin None Amikin, vancomycin Cefuroxime and gentamicin Death Amikin/ciprofloxacin Cefepime/ceftriaxone/ ceftazidime Enucleation Phthisis bulbi Lin et al. 68 Not specified Female Marinella et al.17 48 Male Equi et al.6 54 Female Williams et al.18 56 Female Latorre19 Our case 20 days 47 Female Female 16 Diabetes mellitus, renal abscess Diabetes mellitus, hemodialysis, temporary central venous catheter Diabetes mellitus, hemodialysis, hypertension, temporary central venous catheter Bowel resection for Crohn’s disease, pneumonia Umbilical artery and vein catheters Diabetes mellitus, gum cancer with port-a-cath all of the previously reported cases of endogenous Serratia endophthalmitis resulted in worse than no light perception, including death, evisceration, and phthisis bulbi. Because of this organism’s ability to produce pigment, pigmented hypopyon, such as pink15 or dark6 hypopyon can sometimes also be observed in case of endogenous endophthalmitis. However, even though this is a distinctive feature of Serratia infection, it cannot be relied upon for diagnosis or identification of the organism, because only a minority of S. marcescens strains elaborates pigment.6 In our patient, no particular discoloration or pigmentation of hypopyon upon presentation was noted. Management of this organism can be difficult because it is typically a multi-drug resistant organism. A 10-year retrospective study4 at a tertiary-care hospital showed that the resistance rate to third-generation cephalosporin was about 46%, extended-spectrum penicillin was 57%, and ciprofloxacin was 32%. Bacteria can be introduced into the blood resulting in bacteremia through various portals of entry, and may cause endogenous endophthalmitis when the bacteria are inoculated in the eye via hematogenous spread. Our patient had no history of penetrating injury or surgery to the eye, which suggests that her endophthalmitis was endogenous, and that the bacteria came from the bloodstream. Even though S. marcescens was cultured from our patient’s vitreous initially, her first set of blood culture, which was obtained from the peripheral vein instead of from her Port-a-Cath, had no bacterial growth. In a review comparing blood cultures obtained from intravascular catheters and peripheral vein, Falagas et al.20 showed that the blood drawn from peripheral vein had a higher false negative rate, and could result in undertreatment of true bacteremia. Furthermore, we speculate that based her initial subclinical systemic presentation, the bacterial load in her blood could have been relatively low, which may have resulted in a falsely negative bacterial yield. As a survey for infection source revealed no apparent source of infection other than her catheter, and also because subsequent blood culture obtained from the Port-a-Cath grew the same bacteria as the vitreous, makes the Port-a-Cath the mostly likely portal of entry for the bacteria. In addition to Blood Vitreous, blood Phthisis bulbi Phthisis bulbi Enucleation having an intravascular device, she also had type 2 diabetes mellitus and gum cancer, which together made her vulnerable to developing endogenous endophthalmitis. The Port-a-Cath is an implantable venous access device that is composed of a catheter that connects a portal to the large vein. It is commonly used for receiving chemotherapy or parenteral nutrition in cancer patients, but is also the leading cause of health-care related bloodstream infections21 in this group of patients. The definitive diagnosis of catheter-related blood stream infections is typically made by the removal of the catheter and obtaining a catheter tip culture. However, catheters cannot always be removed easily, especially when the catheters are long-term catheters, need to be surgically removed, or when insertion of a new catheter is difficult. The Port-a-Cath of our patient was not removed because she depended on it for chemotherapy, and because the patient was physically weak and could not withstand another surgical procedure. In a recently updated guideline by the Infectious Diseases Society of America22 in 2009, two methods for definitive diagnosis of catheter-related bloodstream infection (CRBSI) without the removal of catheters were also listed, which are the quantitative blood culture method and the differential time to positivity method. Two blood samples need to be obtained, one drawn from the catheter, and the other from the peripheral vein. For the quantitative blood culture method, a definitive diagnosis of CRBSI is made when the colony count of microbes grown from the blood obtained from the catheter is threefold greater than the colony count of the blood obtained from a peripheral vein. For the differential time to positivity method for diagnosing CRBSI, growth of microbes from the blood drawn from a catheter occurs >2 hours before microbial growth is detected in a blood sample obtained from a peripheral vein. The above catheter-sparing methods are high in sensitivity and specificity (sensitivity 93% and 89e90%, respectively; specificity 97e100% and 72e87%, respectively),21 and facilitate accurate diagnosis of CRBSI without the removal of catheters. The differential time to positivity method is a simple technique and can be done in most laboratories; however, the routine recording of time to positivity for microbial growth was not 48 Y.-C. Poon, H.-K. Kuo / Taiwan Journal of Ophthalmology 3 (2013) 45e48 implemented in our microbiology lab until recently. Therefore, this piece of information was not available to us, and a definite diagnosis of Port-a-Cath infection could not be made in our case. Nevertheless, a high degree of suspicion remains for the Port-aCath as the source of infection, as no other apparent source or portal of entry for infection could be found in our patient. In the case of our patient, it is possible that the bacteria had already been colonized within the port, and may have been washed into the bloodstream with each use of the Port-a-Cath, which consequently resulted in the infection of the eye and the bloodstream. Therefore, it is important to recognize Port-a-Cath as a potential portal of entry for bacteria in cancer patients who present with endogenous endophthalmitis, particularly when no other infection source could be identified. In conclusion, because endogenous endophthalmitis is a severe complication of bloodstream infection with devastating consequences, a detailed evaluation, including physical examination and recording of medical history is essential, so that any indwelling catheters or intravascular devices, particularly the Port-a-Cath in cancer patients are not missed. 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