Case Review
Stenotrophomonas maltophilia meningitis Sadok Ben Amor1, Saleh Baeesa2, Sonia Sebai3
Abstract: Infections due to stenotrophomonas maltophilia are increasingly recognised. Meningitis related to this nosocomial pathogen is however rarely reported. The authors report a new case of meningitis in a debilitating 59year-old patient. The patient underwent multiple neurosurgical procedures and cerebrospinal fluid (CSF) diversion. He was submitted to prolonged broad-spectrum antibiotherapy. Culture of the CSF and the ventricular catheter tip isolated stenotrophomonas maltophilia. The infection responded well to trimethoprim-sulfamethoxazole. Reported neurosurgical patients with stenotrophomonas meningitis are reviewed. Keywords: Meningitis, infection, stenotrophomonas maltophilia and VP shunting
Introduction
Case Report
Stenotrophomonas maltophilia (SM) is a multidrug resistant aerobic gram negative bacillus.4 It is isolated in specimens from patients with nosocomial infections with increasing frequency.8,12,17 Infections that result are extensive with the respiratory tract, soft tissue and skin being most frequently involved.27 Meningitis due to this organism is rare.19,21 We report a new case of meningitis caused by stenotrophomonas maltophilia (SM) complicating a ventriculo-peritoneal shunting in a debilitated patient.
This 59 year-old male is a known hypertensive and diabetic patient for 20 years. Operated 6 months prior in another institution for a non-functioning pituitary macroadenoma via transphenoidal approach, complicated by an intracerebral haematoma with intraventricular bleeding. The haematoma was evacuated as an emergency through temporal craniotomy and an external ventricular drain (EVD) was inserted. The patient developed meningitis due to staphylococcus aureus treated by a combination of ceftriaxone-vancomycin. EVD was then replaced 15 days later with a programmable ventriculo-peritoneal (VP) shunt. Pressure was modified three times viewing the fluctuating level of consciousness. During his stay in Intensive Care Unit, he developed a nosocomial chest infection due to pseudomonas aeroginosa responding well to 15 days of the combination imipenem-amikacine.
1
Department of Neurosurgery Drug Information Unit King Abdulaziz Hospital & Oncology Center Jeddah Saudi Arabia
3
2
Department of Neurosurgery King Abdulaziz University Hospital Jeddah Saudi Arabia
Correspondence: Dr. Sadok Ben Amor Surgical Department King Abdulaziz Hospital & Oncology Center P O Box 31467 Jeddah 21497 Saudi Arabia Tel: (966 5) 467 8477 Fax: (966 2) 637 9442 Email:
[email protected]
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The patient was discharged home, but 1 month later developed some gait difficulties, urinary incontinence and confusional state; hence he was referred to our department for management. Brain computerised tomography (CT) scan showed an active communicating hydrocephalus with porencephalic cavity in the right temporal lobe. A medium pressure VP shunt was inserted with rapid improvement of his mental status and gait. He was covered by a prophylactic antibiotherapy (cefazolin) for 24 hours. On the 5th day post surgically, the patient developed a low-grade fever with mild regression of his mental status. He vomited once and his neck was mildly stiff. Sputum, urine and blood cultures were negative. It was decided to tap the shunt; CSF was clear, and analysis showed a WBC count
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of 103 ells/mm3, with 71% polymorphs and 32% lymphocytes. Protein levels of 448 mg/dL and a glucose level of 2 mmo/L (blood sugar 4.6 mol/L), with CSF culture as growing gram-negative rods identified as stenotrophomonas maltophilia (SM) sensitive to ceftazidim, cefepim, ciprofloxacin, gentamycin and trimethoprim-sulfamethoxazole (TMP-SMX). VP shunt was then removed and the patient started on TMP-SMZ: 160-800 mg IV Q 8h associated with ciprofloxacin: 400 mg IV Q 12h. Culture of the tip of the ventricular shunt also identified growth of SM. Fever subsided within 24 hours and follow-up CSF culture yielded negative results after 3 days. Treatment was continued for 2 weeks, and a new VP shunt was then inserted with rapid improvement of his neurological status.
Discussion Stenotrophomonas maltophilia is a ubiquitous organism propagating in a moist environment.13 Different hospital sources have been reported including ice-making machines, sinks, shower heads, disinfectant solutions, intravenous infusates, ventilator equipment and even hands of health care personnel.9,28,29 It is usually a commensal, a contaminant, or part of the endogenous flora of hospitalised patients.10 It is recognised as a nosocomial pathogen. Community acquired cases of SM have been reported in spite of its limited invasiveness and low level of pathogenicity.14 Reports indicate that the incidence of serious infection due to SM is increasing as a consequence of larger populations of patients with predisposing factors, such as aggressive therapies for malignancies, immunosupression, neutropenia, and antimicrobial pressure.8,15,23 Meningitis caused by SM is rare. To our knowledge, only 13 cases have been reported in the literature.3,7,12,16,18,19,20,21,24,25,26
Spontaneous meningitis due to SM has been initially recognised by Patrick, et al in an elderly patient with emphysema and chronic obstructive pulmonary disease.20 Cases of 2 infants from developing countries have also been published.3,24 It has been described in two premature newborns.12,24 The initial case died before any antibiotherapy and a recently reported case responded well to ciprofloxacin therapy.12 Recently, Spencer & Baron reported a case with atypical presentation (thunderclap headache) in a healthy 31-year-old woman.25 The only significant historical finding was chronic sinusitis. Seven patients experienced SM meningitis after neurosurgical procedures.7,16,18,19,21 All the patients were adults (range 28-65 years). They underwent one or multiple neurosurgical procedures. CSF drainage is reported in 5 of them
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including 3 with Ommaya reservoir insertion. Our patient has multiple neurosurgical procedures, one EVD and 2 VP shunts. Infection occurred within approximately 3 months after the first neurosurgical procedure (1-120 days). The infection manifested in the 5th day in our patient. This form of meningitis is often insidious in onset with protracted course. Fever was constant in all patients, including our own. Neurological symptoms include alteration of the level of consciousness, seizures and hemiparesis.18,19 Progressive modification of the clinical status suggesting a shunt dysfunction was present in our patient, and meningismus, although subtle, was present as in other reported cases. In our patient, CSF analysis was typical of bacterial meningitis, including pleocytosis with neutrophilic predominance, hyperproteinorachia and hypoglucorachia. However, this was not always the case in other reported patients (WBC 14-650 cells/mm3), glucose (42-86 mg/dl), Prt (16-521 mg/dl). As a point of interest, one of the cases reported with spontaneous meningitis had lymphocytes predominance in the CSF.25 One should know that elevation in CSF cell counts and protein concentrations often result from the surgery. This may complicate the interpretation of the CSF for infection. The multiplicity of neurosurgical procedures and the history of post VP shunt meningitis plus diabetes were established risk factors in our patient studies.13,27 Four of the reported meningitis patients received previous antibiotic therapy. Two patients received ceftazidim and one received penicillin.7,19 Our patient, as well as one of Papadakis’ patients, received a course of imipenem-cilastine. The origin of SM remains unknown. Either the environment or the patient carrying a strain at the time of admission, combined with antibiotic pressure, may have favoured the development of such infection. The respiratory tract has been suggested as a reservoir by Gilardi, et al.6 Sputum culture documented Klebsiella pneumonia colonization but no SM in our patient. SM is known to be resistant to many available antimicrobial agents: broad spectrum ß-lactam drugs, aminoglycosides and quinolones.1,5,11 Surprisingly, our stain was sensitive to all these antibiotics. TMP-SMX is still the treatment of choice given its sufficient penetration in the CSF and the favourable outcomes in reported cases.16,18,19,21 TMP-SMX is however bacteriostatic for most isolates and high doses may be needed.22 Considering the age of our patient, his borderline renal function, a combination with ciprofloxacin was preferred to high doses. Of particular concern, some authors reported a 58% rate of resistance
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to cotrimoxazole. Such a high rate is mainly reported by oncology teams with profound immunosuppressed patients on high antimicrobial pressure.15 Removal of the VP shunt was preferred in our case to achieve the highest likelihood of curing his meningitis. Antibiotherapy (systemic and intrathecal) with removal of the VP shunt were effective in the case of Nguyen & Muder.18 Removal of the EVD was also preferred in the case of Muder, et al.16 Removal of the device is probably not mandatory for cases with Ommaya reservoir. Some authors were able to successfully treat meningitis by preserving the appliance giving systemic and direct instillation of antibiotics.2 Papadakis, et al treated successfully their 2 patients with SM meningitis by systemic antibiotherapy without removing the Ommaya reservoir.19 All neurosurgical cases reported responded well to appropriate antibiotherapy and recovered from meningitis (2 deaths unrelated to the infection) in spite of their poor clinical condition.7,16,18,19,21,26 Two deaths occurred with the spontaneous meningitis.3,24 They are related either to delayed treatment or inappropriate antibiotherapy. Mortality rates from SM infections are high in some series (24%). It is difficult to discern the relationship between mortality and SM infections. The poor medical condition of the patients and the presence of other pathogens may explain these high numbers.15 A strict policy of antibiotherapy, especially the use of carbapenems is mandatory. To limit their use in only severe infections due to a gram-negative multiresistant strain may limit the increasing incidence of SM infections.15 Contact precautions should be instituted with SM resistant antibiotherapy, using either TMPSMX or ticarcillin.
Conclusion The incidence of serious infections due to SM is increasingly reported as a consequence of larger populations with predisposing factors. It is most likely that meningitis due to this organism will increase in specific populations with multiple neurosurgical procedures, CSF diversion devices and long-term broadspectrum antibiotherapy. The prognosis remains good with prompt administration of invitro-active antibiotherapy. Nevertheless, high resistance rates to cotrimoxazole and borderline activity of ticarcillinclavulanic acid reported recently needs to be considered seriously and stricter policies in antibiotherapy instituted.
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GENTLE REMINDER Typical CSF findings in meningitis
Viral meningitis
Other diagnostic criteria
Predominant WCC
Protein
Glucose
Lymphocyte
Modest ↑
→
• • • •
Viral isolation from tissue culture Complement fixation Haemagglutinin
]
On serum give retrospective diagnosis
Polymerase chain reaction for herpes simplex
Bacterial meningitis
Neutrophil
↑↑
serum glucose ratio >0.31 in 70% of patients
•
Gram stain identifies organism in 60-90% of patients
Tuberculous meningitis
Lymphocyte
↑↑↑
↓↓↓
•
Chest radiograph - primary complex, effusion, tuberculoma
• •
Stain for acid-fast bacilli
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MRI head ? tuberculoma
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