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Scientific letter
Recurrent meningitis associated to Strongyloides hyperinfection
Meningitis recurrente asociada a hiperinfección por Strongyloides
Inês Pintado Maurya,
Corresponding author
ines.pintadomaury@gmail.com

Corresponding author.
, Diana Nevesa, Aida Pereiraa,b
a Centro Hospitalar Lisboa Norte, Hospital Santa Maria, Department of Infectious Diseases, Lisbon, Portugal
b Lisbon University, Medicine Faculty, Lisbon, Portugal
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Strongyloides stercoralis</span> is an intestinal nematode endemic in tropical areas with high public health impact&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#8211;3</span></a> In Europe&#44; sporadic cases of local infection have been reported in countries like Spain&#46;<a class="elsevierStyleCrossRefs" href="#bib0070"><span class="elsevierStyleSup">4&#44;5</span></a> Exposure to contaminated soils with larvae leads to its penetration into the human skin and venous system&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> It is known to cause a persistent autoinfection in the host&#44; regulated by the Th2 immune response&#44; mediated by interleukins and IgE&#44; which confers protection against hyperinfection&#46;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> Severe strongyloidiasis cases with autoinfection occur when immunity is impaired with consequent increase of larvae burden&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> Consequently the parasites cross the intestinal wall&#44; causing a hyperinfection syndrome &#40;HIS&#41; or disseminated strongyloidiasis with high mortality rate&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> Carriage of Gram negative bacteria through the intestine along with the larvae or through bowel ulcers is a frequent complication&#44; with consequent septic shock and meningitis&#46;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">7&#44;8</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Human T-cell leukemia virus type 1 &#40;HTLV-1&#41;&#44; a retrovirus with clusters of high endemicity around the world&#44;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">7&#8211;9</span></a> and strongyloidiasis have been associated&#46;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">7&#44;8</span></a> Infection of T cells lymphocytes leads to an altered humoral immunity response with decreased immunoglobulin secretion&#46;<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">6&#44;7</span></a> Other HTLV-1-associated diseases such as lymphoproliferative&#44; autoimmune disorders T-cell leukemia&#44; tropical spastic paraparesis and uveitis&#44; and opportunistic infections<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> have been described&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">It is important to recognize the risk of strongyloidiasis and HTLV-1 co-infection among the immigrant population<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;10</span></a> in order to prevent severe complications&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Clinical case</span><p id="par0020" class="elsevierStylePara elsevierViewall">A 53-year-old Brazilian man&#44; living in Portugal for 15 years&#44; with previous history of bacterial meningitis &#40;six months before&#41; without microbiological identification&#44; treated with ampicillin&#44; ceftriaxone&#44; vancomycin and corticosteroids&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">The patient was admitted with the hypothesis of a bacterial meningitis &#40;CSF<span class="elsevierStyleHsp" style=""></span>&#62;<span class="elsevierStyleHsp" style=""></span>1000 polymorphonuclears&#44; 413<span class="elsevierStyleHsp" style=""></span>mg&#47;dL proteins and hypoglycorrhachia 4<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#41;&#46; Antibiotherapy with ampicillin plus ceftriaxone and dexamethasone &#40;10<span class="elsevierStyleHsp" style=""></span>mg every 6<span class="elsevierStyleHsp" style=""></span>h&#41; were started with initial clinical improvement&#46; Gram stain and cultures were negative&#46; After 14 days of admission fever&#44; headache and neck stiffness were noticed with high CSF polymorphonuclears count and hypoglycorrhachia&#46; Assuming a nosocomial meningitis&#44; meropenem and vancomycin were initiated&#46; After <span class="elsevierStyleItalic">Enterococcus faecium</span> isolation in the CSF&#44; meropenem was suspended&#46; Considering the anemia and recurrent meningitis&#44; further investigation was done&#58; negative HIV screening&#44; CD4 T cell count 324<span class="elsevierStyleHsp" style=""></span>cells&#47;mm<span class="elsevierStyleSup">3</span> &#40;38&#37;&#41; with lymphocytes 840<span class="elsevierStyleHsp" style=""></span>cells&#47;mm<span class="elsevierStyleSup">3</span>&#44; positive HTLV-1 serology&#44; negative neoplastic screening&#46; Myelogram showed increased eosinophils &#40;14&#37;&#41; with peripheral eosinophils 270<span class="elsevierStyleHsp" style=""></span>cells&#47;mm<span class="elsevierStyleSup">3</span>&#46; Small bowel enteroscopy showed jejunum and duodenum segmentary changes with scalloping areas &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41;&#46; The biopsy revealed an infectious enteritis with several eggs and larvae suggestive of <span class="elsevierStyleItalic">S&#46; stercoralis</span> &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#41;&#46; Stool culture was negative&#46; Stool examination identified rare larvae of <span class="elsevierStyleItalic">S&#46; stercolaris</span> and treatment with albendazole for 10 days was prescribed&#44; followed by another cycle one week after&#44; while waiting for evermectine pharmacy approval&#46; <span class="elsevierStyleItalic">Strongyloides</span> serology was not available&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Discussion</span><p id="par0030" class="elsevierStylePara elsevierViewall">The presented case concerns a man with recurrent bacterial meningitis episodes without any apparent immunosuppression&#46; Investigation revealed strongyloidiasis and HTLV-1 infection&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">S&#46; stercoralis</span> is endemic in Brazil&#44; where the patient was born&#59; nevertheless is frequently neglected in industrialized countries&#44; leading to recurrent and severe Gram negative bacterial infections&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Epidemiological European studies about HTVL-1 are scarce&#44; reporting infection mostly in blood donors and pregnant women&#44; as they are mostly tested&#46; In Portugal HTLV-1 seropositivity is related to African or South American migrants&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> There is no specific therapy or chemoprophylaxis for HTLV-1&#59; prevention and counseling about transmition routes have been the base the past years&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">A correlation between HTLV-1 and strongyloidiasis has been shown&#44; with a high percentage of <span class="elsevierStyleItalic">S&#46; stercoralis</span> carriers also infected with HTLV-1&#46; Studies in S&#227;o Paulo demonstrated an immunological impairment in co-infected individuals with more severe HIS cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">7&#44;8</span></a> This could be explained by the fact that by infecting T cells lymphocytes&#44; HTLV-1 alters the humoral immunity with an increase of Th1-type &#40;INF-&#947; and TNF-&#945;&#41; and decrease of Th2-type &#40;IL-4 and IL-10&#41; responses&#46; A decreased IgE secretion&#44; required to the parasitic infection control is observed&#44;<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">6&#44;7&#44;10</span></a> contributing to severe helminthic infections&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">S&#46; stercoralis</span>&#44; when associated with immunosuppression&#44; has the ability of acting as an opportunistic agent&#46; Severe strongyloidiasis cases associated to short courses of corticotherapy have been reported&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;3</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Our patient&#44; presented a strongyloidiasis and HTLV-1 infection with a probable impaired Th2 response&#44; that together could justify the recurrent meningitis episodes&#46; Therapy with dexametasone during admission was a major trigger to larvae burden increase and to a nosocomial meningitis episode&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">This case highlights the aspects of two infectious diseases mostly neglected in developed countries and how they can influence the development of a life-threatening disease&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">Although we have been observing an increased knowledge about these pathogens&#44; routine screening guidelines are not established&#46; Imunossupressed patients who present a high risk of exposure to <span class="elsevierStyleItalic">S&#46; stercoralis</span> &#40;who have visited or native from endemic areas&#41; should be screened for strongyloidiasis&#46; Individuals starting immunussupressive drugs or chemotherapy are at risk and eosinophil levels&#44; stool examination and serology should be done&#46; Risk categories should be established and European guidelines created due to recent incidence increase in Europe&#44; mostly due to migration&#46; Migrants at risk with previous hyperinfection syndrome should be investigated for immunosuppressive conditions&#44; including HTLV-1&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflict of interest</span><p id="par0070" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflict of interest and did not receive any financial support&#46; All authors contributed equally to this article&#46; The authors followed their institutions&#8217; protocols to access patient&#39;s data&#44; with the unique purpose of scientific investigation and disclosure&#46; All necessary ethical approval and consents were followed&#46;</p></span></span>"
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