Ir J Med Sci DOI 10.1007/s11845-014-1143-y
CASE BASED REVIEW
Fever of unknown origin in a young man A. Gil-Diaz • R. Estevez-Dominguez • R. Daryanani-Daryanani • J. Gil-Reyes C. Monzon-Moreno
•
Received: 22 April 2014 / Accepted: 5 May 2014 Ó Royal Academy of Medicine in Ireland 2014
Abstract Introduction Sphingomonas paucimobilis bacteraemia is a rare infection typically related to nosocomial outbreaks. Material A 33-year-old immunocompetent man requested evaluation in the Internal Medicine Department for fever without source. His physical examination was normal. Laboratory analysis showed mild hypertransaminasemia. In both blood culture sets grew Gram-negative bacilli, being identified as S. paucimobilis. The patient was treated with oral levofloxacin with full recovery. Conclusion S.paucimobilis infections can occur in the community setting in a relatively non-immunocompromised patient. In a patient with bacteraemia, microbiological studies are crucial to ensure a successful outcome. Keywords Fever Sphingomonas Bacteraemia Community-acquired infection
Introduction Sphingomonas paucimobilis, previously called Pseudomonas paucimobilis, is a yellow-pigmented, aerobic, glucosenonfermenting gram-negative bacillus, and it was initially
A. Gil-Diaz (&) R. Estevez-Dominguez R. Daryanani-Daryanani J. Gil-Reyes Internal Medicine Service, Hospitales San Roque, Las Palmas de G.C, Calle Alcalde Francisco Herna´ndez Gonza´lez, 16, 18 planta, 35001, Las Palmas de Gran Canaria, Canary Islands, Spain e-mail: [email protected] C. Monzon-Moreno Microbiology Service, Hospitales San Roque, Las Palmas de G.C, Las Palmas de Gran Canaria, Canary Islands, Spain
reported as a human pathogen in 1979. It is widely distributed in the natural environment especially in water and soil, and has been implicated in nosocomial outbreaks associated with contaminated water and contaminated ventilator temperature probes [1, 2].
Case history A 33-year-old man with a medical history of morbid obesity (body mass index: 40) and altered basal glycaemia was evaluated in an Internal Medicine consultation room. He referred 1 week evolution of fever up to 40 degrees Celsius accompanied by headache during the fever episodes, disappearing for a few hours with usual antipyretics. He presented no other symptoms and denied any toxic habits. He had not recently traveled outside Spain and had not been exposed to animals. There were no significant findings on physical examination. Initial blood tests highlighted hypertransaminasemia with serum aspartate transaminase levels of 114 U/L (0–32), alanine transaminase of 156 U/L (0–31), gamma glutamyl transferase levels of 180 U/L (5–39), alcaline fosfatase of 131 U/L (35–104), lactic dehydrogenase levels of 691 U/L (240–480), and normal bilirubin levels. The CBC showed lymphocytosis (42 %) and light thrombocytopenia of 122,000/mm3. Image tests showed a normal chest X-Ray, abdominal ultrasound with cholelitiasis, and an echocardiography without signs of endocarditis. Blood cultures were obtained the first day of consult and a first serology determination for Epstein–Barr virus, cytomegalovirus, HIV, and Q fever was also obtained. We started empiric treatment with doxycycline. The first serology determination was negative and blood cultures were positive containing quinolone-sensitive gram-negative bacilli. The isolates were positive for
123
Ir J Med Sci
catalase and oxidase, grew on the blood agar and chocolate agar after 24 h of incubation at 35 °C but no growth was found on MacConkey agar. The isolate was identified as Sphingomonas paucimobilis using API 20 NE (Bio-Me`rieux, Marcy-IE´toile, France) and Wider (Soria Melguizo, Spain) systems. The antibiogram showed resistance to third- and fourth-generation cephalosporins, and susceptibility to quinolones, carbapenems, cotrimoxazole, and aminoglycosides. The patient was treated with levofloxacin 500 mg once a day for 1 week. He remained afebrile after levofloxacin initiation and hepatic enzyme levels became normal. A second serology determination obtained 3 weeks after the first analysis was also negative.
Discussion In our environment, the most frequent causes of fever of unknown origin in young immunocompetent patients are infectious mononucleosis and Q fever, the former with frequent oropharyngeal involvement and the latter with hepatitis, as in our case. This is the reason why we started empirical doxycycline treatment after 10 days of initial febrile syndrome. After isolation of quinolone-sensible gram-negative bacilli, we started levofloxacin with fever resolution. Other recognized rare causes of fever of unknown origin are hematologic cancer like lymphomas and tuberculosis, sometimes without respiratory involvement. Thus, when it is suspected, it is recommendable to dismiss primary HIV infection, which can also be a cause of fever. S. paucimobilis infections are rare and typically occur in immunocompromised patients, but recovery from infections is the rule, even in debilitated hosts. The presence of atypical lipopolysaccharide that constitutes bounds in the outer membrane of S. paucimobilis, with the accompanying deficiency of the endotoxin activity, has been proposed to explain its low virulence [3]. S. paucimobilis has been associated with a variety of infections [4], including bacteraemia, ventilator-associated pneumonia, intravascular catheter-related blood stream infections, meningitis, peritonitis, osteomyelitis, septic arthritis, postoperative endophthalmitis, pleural empyema, splenic abscesses, urinary tract infections, and biliary tract infections. Antibiotic susceptibility of the microorganism varies from study to study. S. paucimobilis bacteraemia occurs infrequently. It has been observed in patients fitted with indwelling devices or in those who are immunocompromised, especially with haematological malignancy [5–7], but it has been encountered with increasing frequency in other clinical
123
settings [8–10]. A recent review [8] showed that S. paucimobilis can cause community-acquired infections in half of the cases. The most frequent community-acquired infection found in this study was primary bacteraemia. Risk factors for S. paucimobilis primary bacteraemia in this review were community-acquired infection, diabetes mellitus, and alcoholism. The patient presented here belonged to these two first categories. This rare case demonstrates that S. paucimobilis bacteraemia can also occur in a relatively non-immunocompromised patient and that in a patient with bacteraemia microbiological studies and susceptibility testing are crucial to ensure a successful outcome. Acknowledgments
There are no fundings.
Conflict of interest
None declared.
References 1. Ryan MP, Adley CC (2010) Sphingomonas paucimobilis: a persistent Gram-negative nosocomial infectious organism. J Hosp Infect 75(3):153–157 2. Bulut C, Yetkin MA, Koruk ST, Erdinc¸ FS¸ , Karakoc¸ EA (2008) Sphingomonas paucimobilis: a rare nosocomial bacteremia agent. Mikrobiyol Bul 42:685–688 3. Kawasaki S, Moriguchi R, Sekiya K, Nakai T, Ono E, Kume K et al (1994) The cell envelope structure of the lipopolysaccharidelacking gram-negative bacterium Sphingomonas paucimobilis. J Bacteriol 176:284–290 4. Cheong HS, Wi YM, Moon SY et al (2008) Clinical features and treatment outcomes of infections caused by Sphingomonas paucimobilis. Infect Control Hosp Epidemiol 29:990–992 5. Kilic A, Senses Z, Kurekci AE, Aydogan H, Sener K, Kismet E et al (2007) Nosocomial outbreak of Sphingomonas paucimobilis bacteremia in a hemato/oncology unit. Jpn J Infect Dis 60:394–396 6. Al-Anazi KA, Jafar SA, Al-Jasser AM, Al-Shangeeti A, Chaudri NA, Al Jurf MD et al (2008) Septic shock caused by Sphingomonas paucimobilis bacteremia in a patient with hematopoietic stem cell transplantation. Transpl Infect Dis 10:142–144 7. Perola O, Nousiainen T, Suomalainen S, Aukee S, Ka¨rkka¨inen UM, Kauppinen J, Ojanen T, Katila ML (2002) Recurrent Sphingomonas paucimobilis bacteraemia associated with a multibacterial water-borne epidemic among neutropenic patients. J Hosp Infect 50:196–201 8. Toh HS, Tay HT, Kuar WK, Weng TC, Tang HJ, Tan CK (2011) Risk factors associated with Sphingomonas paucimobilis infection. J Microbiol Immunol Infect 44(4):289–295 9. Lin JN, Lai CH, Chen YH, Lin HL, Huang CK, Chen WF et al (2010) Sphingomonas paucimobilis bacteremia in humans: 16 case reports and a literature review. J Microbiol Immunol Infect 43(1):35–42 10. Kuo IC, Lu PL, Lin WR, Lin CY, Chang YW, Chen TC et al (2009) Sphingomonas paucimobilis bacteraemia and septic arthritis in a diabetic patient presenting with septic pulmonary emboli. J Med Microbiol 58:1259–1263
CASE BASED REVIEW
Fever of unknown origin in a young man A. Gil-Diaz • R. Estevez-Dominguez • R. Daryanani-Daryanani • J. Gil-Reyes C. Monzon-Moreno
•
Received: 22 April 2014 / Accepted: 5 May 2014 Ó Royal Academy of Medicine in Ireland 2014
Abstract Introduction Sphingomonas paucimobilis bacteraemia is a rare infection typically related to nosocomial outbreaks. Material A 33-year-old immunocompetent man requested evaluation in the Internal Medicine Department for fever without source. His physical examination was normal. Laboratory analysis showed mild hypertransaminasemia. In both blood culture sets grew Gram-negative bacilli, being identified as S. paucimobilis. The patient was treated with oral levofloxacin with full recovery. Conclusion S.paucimobilis infections can occur in the community setting in a relatively non-immunocompromised patient. In a patient with bacteraemia, microbiological studies are crucial to ensure a successful outcome. Keywords Fever Sphingomonas Bacteraemia Community-acquired infection
Introduction Sphingomonas paucimobilis, previously called Pseudomonas paucimobilis, is a yellow-pigmented, aerobic, glucosenonfermenting gram-negative bacillus, and it was initially
A. Gil-Diaz (&) R. Estevez-Dominguez R. Daryanani-Daryanani J. Gil-Reyes Internal Medicine Service, Hospitales San Roque, Las Palmas de G.C, Calle Alcalde Francisco Herna´ndez Gonza´lez, 16, 18 planta, 35001, Las Palmas de Gran Canaria, Canary Islands, Spain e-mail: [email protected] C. Monzon-Moreno Microbiology Service, Hospitales San Roque, Las Palmas de G.C, Las Palmas de Gran Canaria, Canary Islands, Spain
reported as a human pathogen in 1979. It is widely distributed in the natural environment especially in water and soil, and has been implicated in nosocomial outbreaks associated with contaminated water and contaminated ventilator temperature probes [1, 2].
Case history A 33-year-old man with a medical history of morbid obesity (body mass index: 40) and altered basal glycaemia was evaluated in an Internal Medicine consultation room. He referred 1 week evolution of fever up to 40 degrees Celsius accompanied by headache during the fever episodes, disappearing for a few hours with usual antipyretics. He presented no other symptoms and denied any toxic habits. He had not recently traveled outside Spain and had not been exposed to animals. There were no significant findings on physical examination. Initial blood tests highlighted hypertransaminasemia with serum aspartate transaminase levels of 114 U/L (0–32), alanine transaminase of 156 U/L (0–31), gamma glutamyl transferase levels of 180 U/L (5–39), alcaline fosfatase of 131 U/L (35–104), lactic dehydrogenase levels of 691 U/L (240–480), and normal bilirubin levels. The CBC showed lymphocytosis (42 %) and light thrombocytopenia of 122,000/mm3. Image tests showed a normal chest X-Ray, abdominal ultrasound with cholelitiasis, and an echocardiography without signs of endocarditis. Blood cultures were obtained the first day of consult and a first serology determination for Epstein–Barr virus, cytomegalovirus, HIV, and Q fever was also obtained. We started empiric treatment with doxycycline. The first serology determination was negative and blood cultures were positive containing quinolone-sensitive gram-negative bacilli. The isolates were positive for
123
Ir J Med Sci
catalase and oxidase, grew on the blood agar and chocolate agar after 24 h of incubation at 35 °C but no growth was found on MacConkey agar. The isolate was identified as Sphingomonas paucimobilis using API 20 NE (Bio-Me`rieux, Marcy-IE´toile, France) and Wider (Soria Melguizo, Spain) systems. The antibiogram showed resistance to third- and fourth-generation cephalosporins, and susceptibility to quinolones, carbapenems, cotrimoxazole, and aminoglycosides. The patient was treated with levofloxacin 500 mg once a day for 1 week. He remained afebrile after levofloxacin initiation and hepatic enzyme levels became normal. A second serology determination obtained 3 weeks after the first analysis was also negative.
Discussion In our environment, the most frequent causes of fever of unknown origin in young immunocompetent patients are infectious mononucleosis and Q fever, the former with frequent oropharyngeal involvement and the latter with hepatitis, as in our case. This is the reason why we started empirical doxycycline treatment after 10 days of initial febrile syndrome. After isolation of quinolone-sensible gram-negative bacilli, we started levofloxacin with fever resolution. Other recognized rare causes of fever of unknown origin are hematologic cancer like lymphomas and tuberculosis, sometimes without respiratory involvement. Thus, when it is suspected, it is recommendable to dismiss primary HIV infection, which can also be a cause of fever. S. paucimobilis infections are rare and typically occur in immunocompromised patients, but recovery from infections is the rule, even in debilitated hosts. The presence of atypical lipopolysaccharide that constitutes bounds in the outer membrane of S. paucimobilis, with the accompanying deficiency of the endotoxin activity, has been proposed to explain its low virulence [3]. S. paucimobilis has been associated with a variety of infections [4], including bacteraemia, ventilator-associated pneumonia, intravascular catheter-related blood stream infections, meningitis, peritonitis, osteomyelitis, septic arthritis, postoperative endophthalmitis, pleural empyema, splenic abscesses, urinary tract infections, and biliary tract infections. Antibiotic susceptibility of the microorganism varies from study to study. S. paucimobilis bacteraemia occurs infrequently. It has been observed in patients fitted with indwelling devices or in those who are immunocompromised, especially with haematological malignancy [5–7], but it has been encountered with increasing frequency in other clinical
123
settings [8–10]. A recent review [8] showed that S. paucimobilis can cause community-acquired infections in half of the cases. The most frequent community-acquired infection found in this study was primary bacteraemia. Risk factors for S. paucimobilis primary bacteraemia in this review were community-acquired infection, diabetes mellitus, and alcoholism. The patient presented here belonged to these two first categories. This rare case demonstrates that S. paucimobilis bacteraemia can also occur in a relatively non-immunocompromised patient and that in a patient with bacteraemia microbiological studies and susceptibility testing are crucial to ensure a successful outcome. Acknowledgments
There are no fundings.
Conflict of interest
None declared.
References 1. Ryan MP, Adley CC (2010) Sphingomonas paucimobilis: a persistent Gram-negative nosocomial infectious organism. J Hosp Infect 75(3):153–157 2. Bulut C, Yetkin MA, Koruk ST, Erdinc¸ FS¸ , Karakoc¸ EA (2008) Sphingomonas paucimobilis: a rare nosocomial bacteremia agent. Mikrobiyol Bul 42:685–688 3. Kawasaki S, Moriguchi R, Sekiya K, Nakai T, Ono E, Kume K et al (1994) The cell envelope structure of the lipopolysaccharidelacking gram-negative bacterium Sphingomonas paucimobilis. J Bacteriol 176:284–290 4. Cheong HS, Wi YM, Moon SY et al (2008) Clinical features and treatment outcomes of infections caused by Sphingomonas paucimobilis. Infect Control Hosp Epidemiol 29:990–992 5. Kilic A, Senses Z, Kurekci AE, Aydogan H, Sener K, Kismet E et al (2007) Nosocomial outbreak of Sphingomonas paucimobilis bacteremia in a hemato/oncology unit. Jpn J Infect Dis 60:394–396 6. Al-Anazi KA, Jafar SA, Al-Jasser AM, Al-Shangeeti A, Chaudri NA, Al Jurf MD et al (2008) Septic shock caused by Sphingomonas paucimobilis bacteremia in a patient with hematopoietic stem cell transplantation. Transpl Infect Dis 10:142–144 7. Perola O, Nousiainen T, Suomalainen S, Aukee S, Ka¨rkka¨inen UM, Kauppinen J, Ojanen T, Katila ML (2002) Recurrent Sphingomonas paucimobilis bacteraemia associated with a multibacterial water-borne epidemic among neutropenic patients. J Hosp Infect 50:196–201 8. Toh HS, Tay HT, Kuar WK, Weng TC, Tang HJ, Tan CK (2011) Risk factors associated with Sphingomonas paucimobilis infection. J Microbiol Immunol Infect 44(4):289–295 9. Lin JN, Lai CH, Chen YH, Lin HL, Huang CK, Chen WF et al (2010) Sphingomonas paucimobilis bacteremia in humans: 16 case reports and a literature review. J Microbiol Immunol Infect 43(1):35–42 10. Kuo IC, Lu PL, Lin WR, Lin CY, Chang YW, Chen TC et al (2009) Sphingomonas paucimobilis bacteraemia and septic arthritis in a diabetic patient presenting with septic pulmonary emboli. J Med Microbiol 58:1259–1263
![[Fever of unknown origin in 74-year-old multimorbid man].](/assets/img/hold.png)
