Scientific letter
Leishmania colitis: An uncommon cause of gastrointestinal bleeding in an immunosuppressed patient
Colitis por Leishmania: una causa rara de hemorragia digestiva en paciente inmunodeprimido
Francisco Vara-Luiza,b,
, Helena Piresc, Ivo Mendesa,b, Carolina Palmaa,b, Eduardo Fernandesd, Ana Elisa Telese, Francisco Martins do Valec, Marta Patitaa, Jorge Fonsecaa,b, Pedro Pinto-Marquesa
Corresponding author
a Gastroenterology Department, Hospital Garcia de Orta, Almada, Portugal
b Aging Lab, Egas Moniz Center for Interdisciplinary Research (CiiEM), Egas Moniz School of Health and Science, Almada, Portugal
c Infectious Diseases Department, Hospital Garcia de Orta, Almada, Portugal
d Radiology Department, Hospital Garcia de Orta, Almada, Portugal
e Pathology Department, Hospital Garcia de Orta, Almada, Portugal
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"entidad" => "Pathology Department, Hospital Garcia de Orta, Almada, Portugal" "etiqueta" => "e" "identificador" => "aff0025" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Colitis por Leishmania: una causa rara de hemorragia digestiva en paciente inmunodeprimido" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 2338 "Ancho" => 3292 "Tamanyo" => 1037726 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Computed tomography showing pronounced circumferential wall thickening of the recto-sigmoid junction, measuring up to 14<span class="elsevierStyleHsp" style=""></span>mm. (B) Colonoscopy revealing multiple circumferential irregular shaped ulcers and erosions with surrounding erythema, covered by white exudates in the recto-sigmoid junction. (C) Eosinophilic infiltration in the lamina propria and architectural distortion with granuloma formation and multinucleated giant (hematoxylin-eosin staining). (D) Intrahistiocytic microorganisms compatible with Leishmania amastigotes (Giemsa staining).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">A 67-year-old male with history of syndrome of remitting seronegative symmetrical synovitis with pitting edema taking prednisolone 20<span class="elsevierStyleHsp" style=""></span>mg/day, was admitted with fever and arthralgia. During hospitalization, the patient developed abdominal pain and hematochezia with hemodynamic instability. Abdominopelvic CT showed pronounced circumferential wall thickening of the recto-sigmoid junction, measuring up to 14<span class="elsevierStyleHsp" style=""></span>mm, without localized nodularity or mass effect (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A). The preservation of the stratified mural architecture, despite the thickening, suggested a potentially reversible inflammatory process. No significant lymphadenopathy or extraintestinal features were observed. Upper endoscopy showed no findings suggestive of upper gastrointestinal bleeding. Colonoscopy revealed loss of vascular markings, multiple circumferential irregular shaped ulcers and erosions with surrounding erythema, covered by white exudates, as well as loss of vascular markings and mucosal friability in the recto-sigmoid junction (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>B). Biopsies presented an acute colitis with eosinophilic infiltration in the lamina propria and architectural distortion with formation of granuloma and multinucleated giant cells (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>C). On Giemsa staining, intrahistiocytic microorganisms compatible with Leishmania amastigotes were identified, establishing the diagnosis (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>D). A total cumulative dose of 60<span class="elsevierStyleHsp" style=""></span>mg/kg of liposomal amphotericin B was administered, which resulted in clinical improvement and no rebleeding was observed. However, the patient died during hospitalization due to bacterial meningitis with septic shock.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Visceral leishmaniasis is a parasitic infection caused by <span class="elsevierStyleItalic">Leishmania</span> species. Leishmania infection is often asymptomatic, reflecting the ability of the host immune system to control the parasite.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> However, reactivation can occur in the setting of immunosuppression, namely in the context of uncontrolled human immunodeficiency virus infection. In such cases, gastrointestinal involvement may occur and can be manifested with diarrhea, dysphagia, and abdominal pain.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> Also, atypical presentation with intestinal involvement has been described in very few case reports, mainly in the small bowel.<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">3,4</span></a> The course is usually characterized by treatment failure and the prognosis is poor.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">The diagnosis of Leishmania relies mainly on visualization of the amastigote on histopathology. Other methods to establish the diagnosis are isolation by <span class="elsevierStyleItalic">in vitro</span> culture (only performed in reference laboratories), molecular detection of parasite DNA and serologic testing. If the parasite load is high enough, culture can be positive in two weeks but usually takes at least four weeks to obtain a result. In the absence of treatment, mortality rate can reach 90 percent. Liposomal amphotericin B has the highest therapeutic efficacy and the most favorable safety profile.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a> To the best of our knowledge, this is the first case reporting leishmania colitis manifested with lower gastrointestinal bleeding with hemodynamic instability, highlighting this rare entity in the differential diagnosis of acute colitis in immunosuppressed patients. Clinicians should maintain a high suspicion for unusual pathogens in an immunocompromised patient with intestinal lesions.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Statement of ethics</span><p id="par0020" class="elsevierStylePara elsevierViewall">The patient has given written informed consent for publication (including the publication of images).</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Funding</span><p id="par0025" class="elsevierStylePara elsevierViewall">The authors thank <span class="elsevierStyleGrantSponsor" id="gs1">FCT/MCTES</span> for the financial support to CiiEM (<span class="elsevierStyleGrantNumber" refid="gs1">UIDB/04585/2020</span>) through national funds.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Authors’ contributions</span><p id="par0030" class="elsevierStylePara elsevierViewall">FVL, HP, IM and CP wrote the manuscript. EF analyzed the computed tomography images. AET performed the histopathology evaluation. MP performed the colonoscopy. FMV performed clinical follow-up during hospitalization. JF and PPM critically reviewed the manuscript. All authors approved the final version of this paper.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Conflict of interest</span><p id="par0035" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Data availability statement</span><p id="par0040" class="elsevierStylePara elsevierViewall">The complete data of this study are not publicly available due to the patient's privacy but are available from the corresponding author upon reasonable request.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:6 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Statement of ethics" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Funding" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Authors’ contributions" ] 3 => array:2 [ "identificador" => "sec0020" "titulo" => "Conflict of interest" ] 4 => array:2 [ "identificador" => "sec0025" "titulo" => "Data availability statement" ] 5 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 2338 "Ancho" => 3292 "Tamanyo" => 1037726 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Computed tomography showing pronounced circumferential wall thickening of the recto-sigmoid junction, measuring up to 14<span class="elsevierStyleHsp" style=""></span>mm. (B) Colonoscopy revealing multiple circumferential irregular shaped ulcers and erosions with surrounding erythema, covered by white exudates in the recto-sigmoid junction. (C) Eosinophilic infiltration in the lamina propria and architectural distortion with granuloma formation and multinucleated giant (hematoxylin-eosin staining). (D) Intrahistiocytic microorganisms compatible with Leishmania amastigotes (Giemsa staining).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0030" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Leishmaniasis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "S. Burza" 1 => "S.L. Croft" 2 => "M. 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Jain" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:5 [ "tituloSerie" => "Clin Microbiol Infect" "fecha" => "2020" "volumen" => "26" "paginaInicial" => "1345" "paginaFinal" => "1346" ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0050" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Diagnosis and treatment of leishmaniasis: clinical practice guidelines by the Infectious Diseases Society of America (IDSA) and the American Society of Tropical Medicine and Hygiene (ASTMH)" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:3 [ 0 => "N. Aronson" 1 => "B.L. Herwaldt" 2 => "M. 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