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 Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 21  |  Issue : 2  |  Page : 85-88

First report of intracranial epidermoid cyst infection and brain abscess caused by Salmonella Paratyphi A


1 Department of Microbiology, Sree Chitra Tirunal Institute for Medical Sciences and Technology, Thiruvananthapuram, Kerala, India
2 Department of Neurosurgery, Sree Chitra Tirunal Institute for Medical Sciences and Technology, Thiruvananthapuram, Kerala, India
3 Department of Imaging Sciences and Interventional Radiology, Sree Chitra Tirunal Institute for Medical Sciences and Technology, Thiruvananthapuram, Kerala, India

Date of Submission08-Nov-2019
Date of Decision18-Dec-2019
Date of Acceptance20-Dec-2019
Date of Web Publication17-Jan-2020

Correspondence Address:
Dr. Kavita Raja
Professor and Head, Department of Microbiology, Sree Chitra Tirunal Institute for Medical Sciences and Technology (SCTIMST), Thiruvananthapuram - 695 011, Kerala
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jacm.jacm_31_19

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  Abstract 


We report the first case of brain abscess caused by Salmonella Paratyphi A in a young immunocompetent boy with an intracranial epidermoid cyst. A 10-year-old boy presented with gradual onset of headache, progressively increasing over one month duration. The patient was diagnosed with a benign intracranial epidermoid cyst involving the left anterior cranial fossa insinuating to left medial frontal lobe due the initial presentation, and repeat magnetic resonance Imaging taken after two weeks during to worsening of headache revealed pyogenic infection of the epidermoid cyst. The drained pus showed plenty of polymorphonuclear cells with thin Gram-negative bacilli on Gram staining and culture revealed the growth of S. Paratyphi A, which was confirmed by VITEK 2 compact system and serotyping. The isolate was susceptible to Ampicillin, Cotrimoxazole, Ciprofloxacin, Ceftriaxone and Tetracycline. The patient was successfully managed initially with empirical intravenous Meropenem for two weeks and then deescalated to oral Cefixime and Ciprofloxacin. There were no focal neurological deficits while the patient was discharged with advice on antibiotic continuation for two weeks.

Keywords: Abscess, brain, epidermoid cyst, Salmonella Paratyphi A


How to cite this article:
Ponnambath DK, Khimji PB, Kaviyil JE, Raja K, Vilanilam GC, Thomas B. First report of intracranial epidermoid cyst infection and brain abscess caused by Salmonella Paratyphi A. J Acad Clin Microbiol 2019;21:85-8

How to cite this URL:
Ponnambath DK, Khimji PB, Kaviyil JE, Raja K, Vilanilam GC, Thomas B. First report of intracranial epidermoid cyst infection and brain abscess caused by Salmonella Paratyphi A. J Acad Clin Microbiol [serial online] 2019 [cited 2020 Feb 21];21:85-8. Available from: http://www.jacmjournal.org/text.asp?2019/21/2/85/276122




  Introduction Top


 Salmonella More Details is a Gram-negative, facultatively anaerobic bacilli belonging to the family Enterobacteriaceae. They cause a wide spectrum of illness in humans and are broadly classified as those causing enteric fever and those causing non-typhoidal  Salmonellosis More Details (NTS). Extra-intestinal focal Salmonella infections are mostly caused by  Salmonella typhi Scientific Name Search n-typhoidal Salmonella serotypes such as Salmonella Enteritidis and Salmonella Typhimurium while very rarely due to Salmonella Paratyphi A.[1] We report the first case of S. Paratyphi A infection of an intracranial epidermoid cyst with multiple tiny satellite brain abscesses in a 10-year-old boy who was managed successfully with neurosurgical excision and systemic antibiotics.


  Case Report Top


A 10-year-old immunocompetent boy presented with gradual onset of holocranial (involving the entire skull/head) headache of one-month duration. The frequency of the headache was initially 2–3 episodes per week with each episode lasting for half an hour. The frequency and severity of the headache increased in two weeks duration to become continuous in nature. There was no associated history of fever, nausea, vomiting, loose stools, abdominal pain, altered consciousness, neck stiffness, seizures, weakness, sensory or autonomic nervous system disturbances or features of cranial nerve palsies. There was no history or features suggestive of cardiovascular, haematological or immunosuppressive disorders. The patient had an episode of fever, headache and vomiting one year ago and was clinically diagnosed with tuberculous meningitis (TBM) for which he had recently completed anti-tuberculous treatment.

Laboratory analysis revealed normocytic normochromic anaemia (Hb – 9.5 g/dl) and normal total and differential leucocyte count (6700 cells/mm 3, polymorphs – 73% and lymphocytes – 25%) with elevated erythrocyte sedimentation rate (80 mm/h). The patient was diagnosed with a benign intracranial epidermoid cyst involving the left anterior cranial fossa insinuating to left medial frontal lobe [Figure 1]a, [Figure 1]b, [Figure 1]c during the initial presentation with intermittent headache. Magnetic resonance imaging taken after two weeks due to increase in severity and frequency of headache showed enlargement of the lesion with significant perilesional oedema on T2-weighted axial images and irregular marginal enhancement in contrast-enhanced T1-fat saturation images, suggestive of pyogenic infection of the epidermoid cyst [Figure 1]d, [Figure 1]e,[Figure 1]f.
Figure 1: (a-c) Epidermoid tumour of the left anterior cranial fossa, (a) T2-weighted axial image showing hyper-intense well-defined extra-axial mass lesion in the left side middle cranial fossa insinuating to left medial frontal lobe (arrow). (b) Diffusion weighted imaging showing diffusion hyper-intensity of the lesion. (c) Contrast-enhanced fat-saturated axial image showing faint peripheral enhancement (small arrow). (d-f): Infected epidermoid with enlarged lesion and significant perilesional oedema shown on T2-weighted axial image (d) (arrow head), diffusion-weighted imaging showing diffusion hyper-intensity of the lesion (e) and contrast-enhanced T1 fat-saturated image showing irregular marginal enhancement (f)

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The patient underwent left frontotemporal craniotomy (sub-frontal approach) with pus aspiration and excision of cyst. The excised tissue sent for histopathological examination confirmed the diagnosis of epidermoid cyst with dense inflammation. Gram staining of the aspirated pus revealed numerous polymorphonuclear leucocytes with occasional thin Gram-negative bacilli [Figure 2]. Culture on 5% sheep blood agar showed greyish, non-haemolytic colonies with pale non-lactose fermenting colonies on MacConkey agar. The biochemical characters of the culture isolate were as follows: catalase positive, oxidase negative, indole negative, citrate negative, urease negative, triple sugar iron – K +/A , lysine – not decarboxylated, arginine and ornithine – decarboxylated, mannitol fermenter and motile. The culture isolate was confirmed as S. Paratyphi A by VITEK GN ID card (98% probability; excellent identification) by VITEK 2 compact system (BioMerieux, Marcy-l'Etoile, France). Cultures of blood, urine and stool specimens were negative for Salmonella or other pathogens. The Widal test was negative for agglutinating antibodies against the somatic (O) and flagellar (H) antigens of Salmonella causing enteric fever. Antimicrobial susceptibility testing by Kirby–Bauer disc diffusion method and minimum inhibitory concentration determination by VITEK showed susceptibility to Ampicillin, Chloramphenicol, Cotrimoxazole, Ciprofloxacin, Ceftriaxone and Tetracycline (Clinical Laboratory Standards Institute; M100: 29th edition-2019). The patient was initially managed empirically on intravenous Meropenem 1 g Q8H for two weeks, and after confirmation of the isolate as S. Paratyphi A, the antibiotic was deescalated to oral Cefixime 200 mg BD and oral Ciprofloxacin 250 mg BD. The patient was discharged with advice to continue treatment for two weeks. There were no focal neurological deficits observed during discharge of the patient.
Figure 2: Gram-staining of aspirated pus from the infected epidermoid cyst showing polymorphonuclear cells and thin, Gram-negative bacilli (black arrows) (×1000)

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  Discussion Top


Central nervous system (CNS) infections due to Salmonella are very rare. Less than 100 cases of Salmonella intracranial infections have been reported globally between 1884 and 2016. These reported CNS infections range from meningitis, subdural empyema, extradural empyema, brain abscess and rarely as cerebral venous infarcts.[2],[3] Intracranial Salmonella infections have been predominantly reported from infants and young children. Meningitis, subdural and extradural empyemas were frequently observed in infants and children under three years of age, while brain abscess was observed frequently in older children (>3 years age) and young adults.[3] The exact reason for increased predilection of paediatric age group to Salmonella CNS infections is not clear. There are also occasional reports of multiple-site involvement due to Salmonella, e.g., epidural and brain abscess due to S. Enteritidis,[4] subdural and intracerebral abscess due to S. Typhimurium [5] and skin and brain abscess due to S. Typhi.[6]

The most common serotypes of Salmonella causing CNS infections are non-typhoidal Salmonella, S. Enteritidis and S. Typhimurium in the Western literature, while in developing countries where enteric fever is endemic, typhoidal Salmonella, S. Typhi remains the most common serotype.[1],[7] Intracranial infections due to Salmonella occur due to haematogenous seeding of the CNS tissue during enteric fever or transient bacteraemia occurring during NTS (gastroenteritis). In most reported cases of S. Typhi intracranial infections, obvious history or laboratory evidence suggestive of enteric fever (positive Widal test or blood culture) was absent. In our patient, careful history-taking revealed presumptive evidence of enteric fever, which was treated 10 months before this presentation. There was no documentary evidence to confirm the causative role and the Widal test performed during the current admission was also negative probably due to the waning antibody levels and prompt initiation of antibiotics for the enteric fever. The limitations of blood culture for diagnosis of enteric fever includes its lower sensitivity in patients started on antibiotics in the early phase, illness duration of more than a week and less blood volume collected for culture. Since the current CNS lesion was a focal abscess, the patient was non-bacteraemic and the antibody response would have been poor leading to negative Widal test. The predisposing factors reported for focal CNS Salmonella infections in the literature include meningitis, subdural effusion, immunosuppression, human immunodeficiency virus infection, diabetes mellitus, steroid use, pre-existing CNS disease or malignancy (primary or metastatic tumours) and haemoglobinopathies such as sickle cell disease.[8] In our patient, benign intracranial epidermoid cyst, tubercular meningitis and steroid use for the treatment of TBM were the associated risk factors. Only 12 cases of Salmonella brain abscess have been reported in relation to brain tumours in the literature. Most of the cases (10/12) were caused by non-typhoidal Salmonella serotypes (9 – S. Enteritidis, 1 – S. Typhimurium). The most common tumour that was associated was glioblastoma multiforme and S. Enteritidis was the causative serotype in all these cases. Only two cases of typhoidal Salmonella (S. Typhi) have been reported in relation to CNS malignancy (craniopharyngioma and astrocytoma).[9] While there are reports of S. Paratyphi A causing focal infections in various organs, there are no reports of its causal association with brain abscess in the literature.[1] Other serotypes of Salmonella causing enteric fever (S. Paratyphi B– 3, S. Paratyphi C – 1) have also been reported in the literature to cause focal CNS infections.[2],[10] The mortality rate for Salmonella intracranial infections ranges approximately between 15%–18% and up to 25% in patients with associated CNS malignancy.[3],[9] Prompt neurosurgical resection or drainage along with at least four weeks of antibiotic therapy, preferably with a combination of third-generation cephalosporin and ciprofloxacin, remains the best therapeutic option.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Sudhaharan S, Padmaja K, Solanki R, Lakshmi V, Umabala P, Aparna B. Extra-intestinal salmonellosis in a tertiary care center in South India. J Infect Dev Ctries 2014;8:831-7.  Back to cited text no. 1
    
2.
Rodriguez RE, Valero V, Watanakunakorn C. Salmonella focal intracranial infections: Review of the world literature (1884-1984) and report of an unusual case. Rev Infect Dis 1986;8:31-41.  Back to cited text no. 2
    
3.
Mahapatra AK, Pawar SJ, Sharma RR. Intracranial Salmonella infections: Meningitis, subdural collections and brain abscess. A series of six surgically managed cases with follow-up results. Pediatr Neurosurg 2002;36:8-13.  Back to cited text no. 3
    
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Blázquez D, Muñoz M, Gil C, Ruibal JL, El Knaichi F, Aleo E. Brain abscess and epidural empyema caused by Salmonella enteritidis in a child: Successful treatment with ciprofloxacin: A case report. Cases J 2009;2:7131.  Back to cited text no. 4
    
5.
Chadwick D, Mitra T, Sitoh YY. Salmonella typhimurium brain abscess. Lancet 2004;363:947.  Back to cited text no. 5
    
6.
Siddiqui MR, Islam QT, Rahman YU, Shena TY. Multiple skin and brain abscess caused by Salmonella typhi first reported case in Bangladesh. Bangladesh J Med 2015;26:83-5.  Back to cited text no. 6
    
7.
Lakshmi V, Umabala P, Anuradha K, Padmaja K, Padmasree C, Rajesh A, et al. Microbiological spectrum of brain abscess at a tertiary care hospital in South India: 24-year data and review. Patholog Res Int 2011;2011:1-12.  Back to cited text no. 7
    
8.
Kuruvath S, Basu S, Elwitigala JP, Yaneza A, Namnyak SS, Aspoas AR. Salmonella enteritidis brain abscess in a sickle cell disease patient: Case report and review of the literature. Int J Infect Dis 2008;12:298-302.  Back to cited text no. 8
    
9.
Luciani L, Dubourg G, Graillon T, Honnorat E, Lepidi H, Drancourt M, et al. Salmonella enterica serovar enteritidis brain abscess mimicking meningitis after surgery for glioblastoma multiforme: A case report and review of the literature. J Med Case Rep 2016;10:192.  Back to cited text no. 9
    
10.
Soetomenggolo TS, Passat J, Pusponegoro HD, Ismael S. Diagnosis and management of brain abscesses in children. Paediatr Indones 2019;6:118-24.  Back to cited text no. 10
    


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