Two Cases of Meningitis Associated with Gram Negative Bacteremiae and Strongyloidiasis

A B S T R A C T

Background: Strongyloidiasis is a neglected tropical disease and is widely distributed. HTLV-1 co-infection is frequent, and this disease increases the risk of hyper infestation. Disseminated strongyloidiasis often causes severe enterobacteria infection.
Case Presentation: We report the cases of two patients presenting with meningial syndrome. The meningitis was associated with a Gram negative bacteremiae. The investigations performed proved an infection or a previous contact with Strongyloides stercoralis. The infectious outcome was favorable after antibiotic treatment and antiparasitic treatment but one of them was also co-infected with HTLV-1 and died after a lymphoma occurred.
Discussion: S. stercoralis infects the intestinal mucosa, and chronic infection of this pathogen induces inflammation of the intestinal mucosa. Enteric bacteria can gain systemic access and be responsible of organ involvement, including Central Nervous System. HTLV-1 infection increases the prevalence of strongyloidiasis, the rate of treatment failure, and the risk of hyperinfestation.
Conclusions: Strongyloidiasis should not be underdiagnosed, as a specific treatment with antiparasitic drugs is available. We should perform direct microscopy of the stool to detect S. stercoralis in patients who develop severe enterobacterial infection when patients are from an endemic area.

Keywords

Meningitis, strongyloides stercoralis, bacteremiae, HTLV-1

Background

Strongyloidiasis is a disease caused by helminths, mainly by Strongyloides stercoralis. It is a neglected tropical disease and is widely distributed [1, 2]. It can cause intermittent symptoms that mainly affect the intestine, the lungs or the skin [3]. Disseminated strongyloidiasis often causes severe enterobacteria infection especially in immunocompromised hosts, such as those who are human T-lymphotropic virus 1 (HTLV-1) or human immunodeficiency virus (HIV) carriers and those who have been administered systematic corticosteroids [4]. An increasing number of serious infections has been reported in the literature. We herein report on two new cases of enterobacteria infection with miningitis and strongyloidiasis.

Cases Presentations

The first patient is a 51-year-old man presented a meningitis due to Escherichiae coli in January 2015 treated with Cefotaxim. He came from Congo and his medical history consisted only in a chronic B hepatitis. HTLV-1 serology was positive. A disseminated strongyloidiasis was also diagnosed, with a research positive at the parasitologic examination of stool. A first treatment with Ivermectine was initiated. In June, a second meningitis occurred, and cerebrospinal fluid analysis was positive to Klebsiella pneumoniae. A new parasitologic analysis of stool was positive for strongyloidiasis and the patient underwent a gastric endoscopy, and the liquid were found larvae of S. stercoralis. A treatment with Ivermectine was conducted again for three days, and the patient was treated with three weeks with Cefotaxim for the meningitis. Cerebral Magnetic Resonance Imaging showed unspecific hyperintensities of white substance. A treatment with Ivermectine three days a month was decided, and the B hepatitis has been treated with Entecavir. In March 2018, the patient was admitted in intensive unit care for HTLV-1 acutisation (ATCL, adult T-cell leukemia/lymphoma). Clinical outcome was rapidly unfavorable and the patient died.

The second patient is a 30-year-old man who was admitted on May 2019 for fever and headaches. He came from Cameroun the previous week. Cerebrospinal fluid was optically normal, with 232 cells/mm3, 45% PMN and 55% of lymphocytes, without hypoglyrachy, and protein at 0.93 g/L. The direct examination as the culture were negative without any antibiotherapy. Two blood cultures were positives to Serratia marcescens. Serologies were negative for HIV, and HTLV-1/2 and positive for S. stercoralis. The parasitologic examination of stool were negative. Cerebral CT scan did not find any abnormality. Colonoscopy performed found a Forrest III bulb ulcer and a Helicobacter pylori gastritis. The patient was treated with antibiotics and Ivermectine.

Discussion

S. stercoralis infect the intestinal mucosa. Nutman proposed that larvae in non-disseminated hyperinfection were increased in numbers but confined to the organs normally involved in the autoinfective cycle [5]. Systemic infection can be responsible for several diverse clinical manifestations such as reactive arthritis [6]. Larves are also known to give direct involvement of central nervous system (CNS) [7, 8]. Meningeal signs and altered mental status are the most common manifestations seen in patients with CNS strongyloidiasis [8, 9].

CNS culture is often negative but can also be positive for enteric organisms [5, 10, 11]. Recent hypothesis is evoked as that larvae may cause cerebral perfusion defects through capillary obstruction which is seen on Magnetic Resonance Imaging as diffuse infarcts [12]. HTLV-1 infection and strongyloidiasis are two diseases that often share a common geographic distribution. French Guiana is known to harbor high levels of endemicity for both of them [13]. HTLV-1 infection increases the prevalence of strongyloidiasis, the rate of treatment failure, and the risk of hyperinfestation [14, 15].

Conclusion

Strongyloidiasis should not be underdiagnosed, as a specific treatment with antiparasitic drugs is available. We should perform direct microscopy of the stool to detect S. stercoralis in patients who develop severe enterobacterial infections.

Highlights

• We report two cases of meningitis associated with Gram negative bacteremiae and stongyloidiasis.
• One of our case was also associated with HTLV-1 acutisation (ATCL, adult T-cell leukemia/lymphoma).
• Human T-lymphotropic virus 1 (HTLV-1) infection and strongyloidiasis are two diseases that often associated.

Ethics Approval and Consent to Participate

Not applicable.

Consent for Publication

The patients consented for publication.

Availability of Data and Material

Not applicable.

Competing Interests

None.

Funding

None.

Author Contributions

KD drafted the manuscript. AB, JB, and PCP revised it critically for important intellectual content. All authors read and approved the final manuscript.

Article Info

Article Type
Case Report
Publication history
Received: Sun 05, Apr 2020
Accepted: Fri 17, Apr 2020
Published: Fri 24, Apr 2020
Copyright
© 2023 Kevin DIALLO. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. Hosting by Science Repository.
DOI: 10.31487/j.JCMCR.2020.01.04

Author Info

Corresponding Author
Kevin DIALLO
Department of Infective and Tropical Diseases, Intercommunal Hospital Center of Villeneuve-Saint-Georges, France

Figures & Tables

References

  1. Olsen A, van Lieshout L, Marti H, Polderman T, Polman K et al. (2009) Strongyloidiasis--the most neglected of the neglected tropical diseases? Trans R Soc Trop Med Hyg 103: 967-972. [Crossref]
  2. World Health Organisation. Intestinal Worms. Strongyloidiasis. Epidemiology.
  3. Jourdan PM, Lamberton PHL, Fenwick A, Addiss DG (2018) Soil-transmitted helminth infections. Lancet 391: 252-265. [Crossref]
  4. Hosoda T, Sakamoto M, Orikasa H, Kubomura A, Misaki T et al. (2020) Septic Meningitis and Liver Abscess due to Hypermucoviscous Klebsiella pneumoniae Complicated with Chronic Strongyloidiasis in a Human T-lymphotropic Virus 1 Carrier. Intern Med 59: 129-133. [Crossref]
  5. Keiser PB, Nutman TB (2004) Strongyloides stercoralis in the Immunocompromised Population. Clin Microbiol Rev 17: 208-217. [Crossref]
  6. Patey O, Bouhali R, Breuil J, Chapuis L, Courillon Mallet A et al. (1990) Arthritis associated with Strongyloides stercoralis. Scand J Infect Dis 22: 233-236. [Crossref]
  7. Oktar N, Ozer H, Demirtas E (2018) Central nervous system Strongyloides stercoralis. A case report. Turk Neurosurg. [Crossref]
  8. Rodriguez M, Flores P, Ahumada V, Vazquez Vazquez L, Alvarado de la Barrera C et al. (2012) Central Nervous System Strongyloidiasis and Cryptococcosis in an HIV-Infected Patient Starting Antiretroviral Therapy. Case Rep Med 2012: 575470. [Crossref]
  9. Morgello S, Soifer FM, Lin CS, Wolfe DE (1993) Central nervous system Strongyloides stercoralis in acquired immunodeficiency syndrome: a report of two cases and review of the literature. Acta Neuropathol 86: 285-288. [Crossref]
  10. Woll F, Gotuzzo E, Montes M (2013) Strongyloides stercoralis infection complicating the central nervous system. Handb Clin Neurol 114: 229-234. [Crossref]
  11. Greaves D, Coggle S, Pollard C, Aliyu SH, Moore EM (2013) Strongyloides stercoralis infection. BMJ 347: f4610. [Crossref]
  12. Marcos LA, Terashima A, Canales M, Gotuzzo E (2011) Update on strongyloidiasis in the immunocompromised host. Curr Infect Dis Rep 13: 35-46. [Crossref]
  13. Carme B, Motard A, Bau P, Day C, Aznar C et al. (2002) Intestinal parasitoses among Wayampi Indians from French Guiana. Parasite 9: 167-174. [Crossref]
  14. Gotuzzo E, Terashima A, Alvarez H, Tello R, Infante R et al. (1999) Strongyloides stercoralis hyperinfection associated with human T cell lymphotropic virus type-1 infection in Peru. Am J Trop Med Hyg 60: 146-149. [Crossref]
  15. Patey O, Gessain A, Breuil J, Courillon Mallet A, Daniel MT et al. (1992) Seven years of recurrent severe strongyloidiasis in an HTLV-I-infected man who developed adult T-cell leukaemia. AIDS 6: 575-579. [Crossref]