British Journal of Neurosurgery (199 1) 5, 5 15-5 17
SHORT REPORT
Cysto-peritoneal shunt infection with Trichosporon beigelii
Br J Neurosurg Downloaded from informahealthcare.com by University of Auckland on 12/04/14 For personal use only.
RICHARD D. ASHPOLE,' KIM JACOBSON: ANDREW T. KING,' & ALEC E. HOLMES'
'
Department of Neurological Surgery and Department of Clinical Microbiology and Public Health Laboratory, Addenbrooke's Hospital, Hill 3 Road, Cambridge CB2 2QQ UK.
Abstract Infection is a frequent problem affecting cysto-peritoneal shunts, the usual causative organisms being Staphylococcus epidennidis and Staphylococcus aureus. Fungi are rarely isolated from such infections. We present the first report, as far as the authors are aware, of a case of Trichosporon beigelii shunt infection that responded to shunt removal and antifungal therapy. Key words: Shunt infection, fungus, Trichosporon beigelii.
Introduction A frequent problem associated with VP shunts is infection. Reported infection rates vary widely from 4 to 27% in different series and seem to vary with age, surgical technique and antimicrobial pro phyla xi^.'-^ The pathogens most often encountered are skin commensals with Staphylococcus epidermidis (45%), S. aureus (25%), and corynebacteria (5%) being the commonest.' Fungi have rarely been described as causes of shunt infection but Cyptococcus neoformans and Candida species are the most frequent isolates.'V5 We present the first report, as far as the authors are aware, of a case of Trichosporon beigelii shunt infection. Case report
A male patient was born at term after an ultrasound at 28 weeks gestation had revealed an intracranial arachnoid cyst. CT scanning at
birth confirmed a large intrahemispheric arachnoid cyst with agenesis of the corpus callosum and associated mass effect. A cysto-peritoneal shunt (medium pressure, Forth system) was inserted when he was 6 weeks old. The upper and lower ends were subsequently revised for leakage after 3 months and omental block after 7 months. His recovery was complicated by persisting malaise although no temperature or neurological signs were elicited. He presented again aged 11 months at another centre when examination of a CSF specimen revealed scanty pseudohyphae on Gram stain. The cell count was slightly high (26 x 1O6/litre polymorphs, 10 x 106/litre lymphocytes and 2 x 106/litre red cells) but the protein was normal at 0.1 g/l and the glucose was slightly low at 2.9 mmol/l (simultaneous blood glucose 5.3 mmol/l). Antifungal treatment was commenced with amphotericin B (2.5 mg intravenously) four times daily, and he was transferred to Addenbrooke's Hospital.
515
Br J Neurosurg Downloaded from informahealthcare.com by University of Auckland on 12/04/14 For personal use only.
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Richard D. Ashpole et al.
On arrival he was alert with no neurological signs and was mildly pyrexial at 37.3"C. A peripheral white cell count was 19.7 x lO9/litre and his CRP was 120 mg/l (normal (6 mg/l). The cysto-peritoneal shunt was removed and external ventricular drainage instigated. A ventricular CSF collected at operation had 360 x 106/litre red cells, 6 X 106/litre polymorphs, 1 X 106/litre lymphocytes and the protein concentration was 0.1 g/l. No organisms were seen on microscopy. Culture of the shunt assembly and original CSF grew a yeast subsequently identified as Ttl'chosporon beigelii. Further CSF samples were negative by Gram stain and culture, and blood cultures were also sterile. Amphotericin B (2.5 mg IV) once daily and 55ucytosine (500 mg IV) four times daily were continued for a total of 23 days and stopped when the CSF had been acellular and culture negative for 10 days. A new cysto-peritoneal shunt was then inserted and he made an uneventful recovery. Subsequent review of the patient's notes revealed that T. beigelii had been isolated from an acellular CSF after the second revision operation, 3 months prior to admission but at that time it was considered to be a skin contaminant and no treatment had been given. Conclusions T. beigilii is a yeast-like organism belonging to the family Cryptococcuceue which are widely distributed in the environment in skin, dust, and soil. It is a well-known cause of white piedra, an innocuous hair shaft infection common in tropical and temperate countries. Systemic infections with this organism have been reported in immunosuppressed patients: especially those with leukaemia, and one case of meningitis has been reported, again in a patient with acute lymphoblastic leukaemia.' Other reports have involved patients with prosthetic valves and intravenous drug misusers. In fungal meningitis, fungal elements are rarely observed on direct microscopy, the normal method of diagnosis being by culture or
by serology.8 Cisternal puncture may be more fruitful due to the predilection of fungi for the basal meninges although large volumes of CSF may be necessary for successful isolation. T. beigelii is identified by its morphological appearances on Gram stain and culture. It is a yeast-like organism with hyphae and pseudohyphae, blastoconidia and arthroconidia. It does not produce asexual endospores or sexual spores. It grows well aerobically on blood agar and Sabouraud's agar at 37"C, producing smooth, creamy colonies which become wrinkled after approximately 1 week. Further tests, useful in identification, include urease production, assimilation of many carbohydrates, and nitrate reduction. It is not fermentative. It shares a common capsular polysaccharide antigen with Cryptococc~sneoformans and therefore cross-reacts ser~logically.~ Too few cases have been reported to make firm recommendations about antifungal therapy; single and combination treatment have both been successful. Susceptibility testing gives variable results and in vitro susceptibility does not necessarily reflect the in oivo response. Overall mortality of T. beigelii meningitis in previous studies has approached 74%1° and successful treatment has followed with return of neutrophils and thus immunocompetence, or removal of an infected focus such as a prosthetic heart valve, an eye, or removal of an infected shunt as in this case. This report adds another unusual pathogen to the increasing list of agents that may infect the CSF and is of interest primarily because the patient was immunocompetent and treatment was successful. In this case it is impossible to separate the roles played by antifungal therapy and removal of the prothesis. Similar cases will probably occur and vigilance will be required to recognize and diagnose them correctly.
Acknowledgements The authors would like to thank Dr M. Farrington for his critical review of the manuscript; and the Department of Clinical Micro-
Cysto-pen'toneal shunt infection biology and Public Health at Ipswich for providing us with their initial results.
Address for correspondence: K. Jacobson, Department of Clinical Microbiology and Public Health Laboratory, Addenbrooke's Hospital, Hill's Road, Cambridge CB2 2QQ, UK. References 1 Shapiro S, Boaz J, Kleiman M et al. Origin of
organisms infecting ventricular shunts. Neurosurgery Br J Neurosurg Downloaded from informahealthcare.com by University of Auckland on 12/04/14 For personal use only.
1988; 22868-71.
2 Schoenbaum SC, Gardner P, Dhillito J. Infections of cerebrospinal fluid shunts: Epidemiology, clinical manifestations and therapy. J Infect Dis 1975; 131543-52. 3 Stromblad L, Schalen C, Steen A et al. Bacterial contamination in cerebrospinal fluid shunt surgery. Scand J Infect Dis 1987; 19211-4.
5 17
Y, Keren G et al. Cerebrospinal shunt infections in children. Paediatr Infect Dis J 1987; 6:921-4. 5 Yadev S, Perfect J, Friedman A. Successful treatment of cryptococcal ventriculoatrial shunt infection with systemic therapy alone. Neurosurgery 1988; 23:372-3. 6 Annaise EJ, Bodey GP, Rinaldi MG. Emerging fungal pathogens. Eur J Clin Microbiol Infect Dis 1989;
4 Meirovitch MD, Kitai-Cohen
8:323-30. 7 Surmount I, Vergauwen B, Marcelis L et al. First
report of chronic meningitis caused by Trichosporon beigelii. Eur J Clin flicobiol Infect Dis 1989; 8:323-30. 8 Salaki JS, Louria DB, Chmel H. Fungal and yeast infections of the central nervous system. Medicine (Baltimore) 1984; 63:108-32. 9 McManus EJ, Bozalech MJ, Jones JM. Roles of latex agglutination test for cryptococcal antigen in diagnosing disseminated infections with Trichosporon beigelii. J Infect Dis 1985; 151:1167-9. 10 Hay J, Kuo-Ching H, Bolston K et al. Trichosporon beigilii infection, a review. Rev Infect Dis 1986; 8:959-67.
SHORT REPORT
Cysto-peritoneal shunt infection with Trichosporon beigelii
Br J Neurosurg Downloaded from informahealthcare.com by University of Auckland on 12/04/14 For personal use only.
RICHARD D. ASHPOLE,' KIM JACOBSON: ANDREW T. KING,' & ALEC E. HOLMES'
'
Department of Neurological Surgery and Department of Clinical Microbiology and Public Health Laboratory, Addenbrooke's Hospital, Hill 3 Road, Cambridge CB2 2QQ UK.
Abstract Infection is a frequent problem affecting cysto-peritoneal shunts, the usual causative organisms being Staphylococcus epidennidis and Staphylococcus aureus. Fungi are rarely isolated from such infections. We present the first report, as far as the authors are aware, of a case of Trichosporon beigelii shunt infection that responded to shunt removal and antifungal therapy. Key words: Shunt infection, fungus, Trichosporon beigelii.
Introduction A frequent problem associated with VP shunts is infection. Reported infection rates vary widely from 4 to 27% in different series and seem to vary with age, surgical technique and antimicrobial pro phyla xi^.'-^ The pathogens most often encountered are skin commensals with Staphylococcus epidermidis (45%), S. aureus (25%), and corynebacteria (5%) being the commonest.' Fungi have rarely been described as causes of shunt infection but Cyptococcus neoformans and Candida species are the most frequent isolates.'V5 We present the first report, as far as the authors are aware, of a case of Trichosporon beigelii shunt infection. Case report
A male patient was born at term after an ultrasound at 28 weeks gestation had revealed an intracranial arachnoid cyst. CT scanning at
birth confirmed a large intrahemispheric arachnoid cyst with agenesis of the corpus callosum and associated mass effect. A cysto-peritoneal shunt (medium pressure, Forth system) was inserted when he was 6 weeks old. The upper and lower ends were subsequently revised for leakage after 3 months and omental block after 7 months. His recovery was complicated by persisting malaise although no temperature or neurological signs were elicited. He presented again aged 11 months at another centre when examination of a CSF specimen revealed scanty pseudohyphae on Gram stain. The cell count was slightly high (26 x 1O6/litre polymorphs, 10 x 106/litre lymphocytes and 2 x 106/litre red cells) but the protein was normal at 0.1 g/l and the glucose was slightly low at 2.9 mmol/l (simultaneous blood glucose 5.3 mmol/l). Antifungal treatment was commenced with amphotericin B (2.5 mg intravenously) four times daily, and he was transferred to Addenbrooke's Hospital.
515
Br J Neurosurg Downloaded from informahealthcare.com by University of Auckland on 12/04/14 For personal use only.
516
Richard D. Ashpole et al.
On arrival he was alert with no neurological signs and was mildly pyrexial at 37.3"C. A peripheral white cell count was 19.7 x lO9/litre and his CRP was 120 mg/l (normal (6 mg/l). The cysto-peritoneal shunt was removed and external ventricular drainage instigated. A ventricular CSF collected at operation had 360 x 106/litre red cells, 6 X 106/litre polymorphs, 1 X 106/litre lymphocytes and the protein concentration was 0.1 g/l. No organisms were seen on microscopy. Culture of the shunt assembly and original CSF grew a yeast subsequently identified as Ttl'chosporon beigelii. Further CSF samples were negative by Gram stain and culture, and blood cultures were also sterile. Amphotericin B (2.5 mg IV) once daily and 55ucytosine (500 mg IV) four times daily were continued for a total of 23 days and stopped when the CSF had been acellular and culture negative for 10 days. A new cysto-peritoneal shunt was then inserted and he made an uneventful recovery. Subsequent review of the patient's notes revealed that T. beigelii had been isolated from an acellular CSF after the second revision operation, 3 months prior to admission but at that time it was considered to be a skin contaminant and no treatment had been given. Conclusions T. beigilii is a yeast-like organism belonging to the family Cryptococcuceue which are widely distributed in the environment in skin, dust, and soil. It is a well-known cause of white piedra, an innocuous hair shaft infection common in tropical and temperate countries. Systemic infections with this organism have been reported in immunosuppressed patients: especially those with leukaemia, and one case of meningitis has been reported, again in a patient with acute lymphoblastic leukaemia.' Other reports have involved patients with prosthetic valves and intravenous drug misusers. In fungal meningitis, fungal elements are rarely observed on direct microscopy, the normal method of diagnosis being by culture or
by serology.8 Cisternal puncture may be more fruitful due to the predilection of fungi for the basal meninges although large volumes of CSF may be necessary for successful isolation. T. beigelii is identified by its morphological appearances on Gram stain and culture. It is a yeast-like organism with hyphae and pseudohyphae, blastoconidia and arthroconidia. It does not produce asexual endospores or sexual spores. It grows well aerobically on blood agar and Sabouraud's agar at 37"C, producing smooth, creamy colonies which become wrinkled after approximately 1 week. Further tests, useful in identification, include urease production, assimilation of many carbohydrates, and nitrate reduction. It is not fermentative. It shares a common capsular polysaccharide antigen with Cryptococc~sneoformans and therefore cross-reacts ser~logically.~ Too few cases have been reported to make firm recommendations about antifungal therapy; single and combination treatment have both been successful. Susceptibility testing gives variable results and in vitro susceptibility does not necessarily reflect the in oivo response. Overall mortality of T. beigelii meningitis in previous studies has approached 74%1° and successful treatment has followed with return of neutrophils and thus immunocompetence, or removal of an infected focus such as a prosthetic heart valve, an eye, or removal of an infected shunt as in this case. This report adds another unusual pathogen to the increasing list of agents that may infect the CSF and is of interest primarily because the patient was immunocompetent and treatment was successful. In this case it is impossible to separate the roles played by antifungal therapy and removal of the prothesis. Similar cases will probably occur and vigilance will be required to recognize and diagnose them correctly.
Acknowledgements The authors would like to thank Dr M. Farrington for his critical review of the manuscript; and the Department of Clinical Micro-
Cysto-pen'toneal shunt infection biology and Public Health at Ipswich for providing us with their initial results.
Address for correspondence: K. Jacobson, Department of Clinical Microbiology and Public Health Laboratory, Addenbrooke's Hospital, Hill's Road, Cambridge CB2 2QQ, UK. References 1 Shapiro S, Boaz J, Kleiman M et al. Origin of
organisms infecting ventricular shunts. Neurosurgery Br J Neurosurg Downloaded from informahealthcare.com by University of Auckland on 12/04/14 For personal use only.
1988; 22868-71.
2 Schoenbaum SC, Gardner P, Dhillito J. Infections of cerebrospinal fluid shunts: Epidemiology, clinical manifestations and therapy. J Infect Dis 1975; 131543-52. 3 Stromblad L, Schalen C, Steen A et al. Bacterial contamination in cerebrospinal fluid shunt surgery. Scand J Infect Dis 1987; 19211-4.
5 17
Y, Keren G et al. Cerebrospinal shunt infections in children. Paediatr Infect Dis J 1987; 6:921-4. 5 Yadev S, Perfect J, Friedman A. Successful treatment of cryptococcal ventriculoatrial shunt infection with systemic therapy alone. Neurosurgery 1988; 23:372-3. 6 Annaise EJ, Bodey GP, Rinaldi MG. Emerging fungal pathogens. Eur J Clin Microbiol Infect Dis 1989;
4 Meirovitch MD, Kitai-Cohen
8:323-30. 7 Surmount I, Vergauwen B, Marcelis L et al. First
report of chronic meningitis caused by Trichosporon beigelii. Eur J Clin flicobiol Infect Dis 1989; 8:323-30. 8 Salaki JS, Louria DB, Chmel H. Fungal and yeast infections of the central nervous system. Medicine (Baltimore) 1984; 63:108-32. 9 McManus EJ, Bozalech MJ, Jones JM. Roles of latex agglutination test for cryptococcal antigen in diagnosing disseminated infections with Trichosporon beigelii. J Infect Dis 1985; 151:1167-9. 10 Hay J, Kuo-Ching H, Bolston K et al. Trichosporon beigilii infection, a review. Rev Infect Dis 1986; 8:959-67.
