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Tubular acute lymphangitis caused by Rickettsia sibirica mongolitimonae
Linfangitis aguda tubular por Rickettsia sibirica mongolitimonae
Sonia Miguélez Ferreiroa,
Corresponding author
tormentabranca@hotmail.com

Corresponding author.
, David Navalpotro Rodríguezb
a Servicio de Urgencias de Atención Primaria, Hospital General de Valencia, Valencia, Spain
b Servicio de Microbiología, Hospital General de Valencia, Valencia, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Acute lymphangitis is an acute inflammatory process in healthy or anatomically altered lymphatic vessels in either immunocompetent or immunocompromised patients&#46; It generally develops after inoculation of microorganisms into the lymphatic vessels through a skin defect or as a complication of a distal infection&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It is commonly caused by <span class="elsevierStyleItalic">Streptococcus pyogenes</span> and <span class="elsevierStyleItalic">Staphylococcus aureus&#59;</span> in immunocompromised patients&#44; Gram-negative bacteria&#59; <span class="elsevierStyleItalic">Pasteurella multocida</span> or <span class="elsevierStyleItalic">Spirillum minus</span> after dog&#44; cat or rat bites&#46; Less common culprits are mycobacteria&#44; viruses&#44; fungi&#44; parasites&#44; <span class="elsevierStyleItalic">Rickettsias</span> or non-infectious causes&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Clinically speaking&#44; acute lymphangitis may be&#58; reticular&#44; tubular&#44; nodular&#44; phlyctenular or necrotising&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">We report here a case of tubular acute lymphangitis caused by <span class="elsevierStyleItalic">Rickettsia sibirica mongolitimonae</span> in Valencia&#44; Spain&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">This was a 59-year-old man with no relevant medical history who consulted in April 2019 with a four-day history of pre-syncope&#44; general malaise&#44; headache&#44; arthralgia&#44; myalgia and fever of 38&#46;5<span class="elsevierStyleHsp" style=""></span>&#176;C&#46; Three days before the onset of his symptoms&#44; he developed an erythematous papule on his right leg&#46; In the previous 48<span class="elsevierStyleHsp" style=""></span>h it had become enlarged so&#44; suspecting cellulitis&#44; he was started on amoxicillin&#47;clavulanic acid 500<span class="elsevierStyleHsp" style=""></span>mg&#47;8<span class="elsevierStyleHsp" style=""></span>h&#46; Despite the antibiotic&#44; the lesion had worsened&#44; spreading to the knee&#44; and he had developed painful enlarged right inguinal lymph nodes&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">He stated that he had not travelled outside Valencia&#46; He had been in the countryside in an area with pigeons&#44; rabbits&#44; cats and dogs&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">On physical examination&#44; he was found to have a temperature of 37&#46;6<span class="elsevierStyleHsp" style=""></span>&#176;C&#44; the lesions shown in <a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a> and left inguinal lymphadenitis&#46; He had no peripheral lesions or rashes&#46; The only blood test finding was CRP 35<span class="elsevierStyleHsp" style=""></span>mg&#47;l&#46; Urinalysis and chest X-ray were normal&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">The appearance of the eschar raised the suspicion of rickettsiosis and he was started on treatment with oral doxycycline 100<span class="elsevierStyleHsp" style=""></span>mg&#47;12<span class="elsevierStyleHsp" style=""></span>h for five days&#46; Within 48<span class="elsevierStyleHsp" style=""></span>h his temperature had returned to normal and the rest of the symptoms and lesions gradually disappeared&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">For the aetiological diagnosis&#44; serological chemiluminescence immunoassay &#40;CLIA&#41; &#40;Vircell&#44; Granada&#44; Spain&#41; was performed for <span class="elsevierStyleItalic">Rickettsia conorii</span>&#44; <span class="elsevierStyleItalic">C&#46; burnetti</span> and <span class="elsevierStyleItalic">B&#46; burgdorferi</span>&#44; which were negative&#46; A sample of exudate was obtained from the eschar&#44; and Real-Time PCR &#40;Fast Track Diagnostic&#44; Luxembourg&#41; detected <span class="elsevierStyleItalic">Rickettsia</span> spp DNA&#46; Definitive species identification was obtained by molecular sequencing of a 525bp fragment of the <span class="elsevierStyleItalic">ompA gene</span>&#44; which confirmed the presence of <span class="elsevierStyleItalic">Rickettsia sibirica mongolitimonae</span> &#40;99&#46;7&#37; similarity with sequences deposited in GenBank&#174;&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> The serology was repeated at six weeks using CLIA and indirect immunofluorescence &#40;IIF&#41; &#40;Vircell&#44; Granada&#44; Spain&#41;&#58; IgG and IgM were positive against <span class="elsevierStyleItalic">Rickettsia conorii&#44;</span> with evidence of cross reactions between <span class="elsevierStyleItalic">R&#46; sibirica</span> and <span class="elsevierStyleItalic">R&#46; conorii</span>&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Acute tubular&#44; trunk- or branch-like lymphangitis affects the main superficial branches of the lymphatic vessels&#46; An erythematous plaque appears and an indurated&#44; erythematous&#44; warm&#44; palpable and painful sinuous tubular cord develops &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#44; extending proximally to the regional lymph nodes &#40;lymphadenitis&#41;&#46; It is usually accompanied by systemic symptoms&#58; chills&#44; fever&#44; general malaise&#44; headache&#44; arthralgia&#44; myalgia and nausea and&#47;or vomiting&#46; Laboratory tests may show leucocytosis&#44; increased inflammatory parameters&#44; and positive blood cultures&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It generally responds to empirical oral antibiotics&#46; Infants and small children may require hospital admission in the case of significant systemic involvement or antibiotic resistance&#46; Further exploratory tests are not usually necessary&#44; unless there is a lack of response or atypical aetiology is suspected&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The presence of lymphangitis&#44; fever and an inoculation eschar should make us suspect lymphangitis-associated rickettsiosis &#40;LAR&#41;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> and start treatment with doxycycline&#46; LAR do not have a specific clinical-aetiological correlation&#44; and can be caused by different species of <span class="elsevierStyleItalic">Rickettsiae</span>&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;6</span></a> One of these is <span class="elsevierStyleItalic">R&#46; sibirica mongolitimonae</span>&#44; which is involved in less than half of the published cases of LAR&#46;<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6&#44;7</span></a> This is a rare type of rickettsiosis&#44;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> which causes lymphangitis in approximately 35&#37; of cases&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> It tends to be transmitted in spring and summer by tick bites of the genera <span class="elsevierStyleItalic">Rhipicephalus</span> and <span class="elsevierStyleItalic">Hyalommas</span>&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> We confirmed a case of lymphangitis due to <span class="elsevierStyleItalic">R&#46; sibirica mongolitimonae</span> by PCR analysis of the eschar sample&#46; This technique allows the definitive diagnosis of rickettsiosis with precision&#44; speed and high sensitivity and specificity&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> Serology requires 2&#8211;3 weeks for seroconversion and shows cross-reactions&#44; as in our case&#46; The existence of cross reactions means that <span class="elsevierStyleItalic">R&#46; sibirica mongolitimonae</span> is probably underdiagnosed&#44; not only as a cause of LAR&#44; but also of other types of rickettsiosis&#44; as the microbiological diagnosis is based mainly on serology&#44; and in many cases it is only performed against <span class="elsevierStyleItalic">R&#46; conorii</span>&#46;</p></span>"
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