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=> array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">d</span>" "identificador" => "aff0020" ] ] ] 11 => array:3 [ "nombre" => "Jorge" "apellidos" => "Fonseca" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:4 [ 0 => array:3 [ "entidad" => "Gastroenterology Department, Hospital Garcia de Orta, Almada, Portugal" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Egas Moniz Center for Interdisciplinary Research (CiiEM), Egas Moniz School of Health and Science, Caparica, Portugal" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Pathology Department, Hospital Garcia de Orta, Almada, Portugal" "etiqueta" => "c" "identificador" => "aff0015" ] 3 => array:3 [ "entidad" => "Surgery Department, Hospital Garcia de Orta, Almada, Portugal" "etiqueta" => "d" "identificador" => "aff0020" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Transformación maligna en fístula tras proctosigmoidectomía: una rara complicación de la enfermedad de Crohn de larga evolución" ] ] "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" => 1190 "Ancho" => 1674 "Tamanyo" => 293654 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Pelvic magnetic resonance imaging revealing a 3.9<span class="elsevierStyleHsp" style=""></span>cm nodular image with necrotic center. (B) Histopathology showing signet ring mucinous adenocarcinoma. (C) Endoscope-guided gynecological examination showing an endoscope an ulcerated and infiltrative lesion in the vaginal wall. (D) Bone scan with multiple bone metastasis.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">In Crohn's disease (CD), fistula formation is an aggressive and difficult to treat phenotype that affects up to 50% of patients and requires a multidisciplinary approach.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">1,2</span></a> Malignant transformation of CD fistulas is rare, and pathogenesis is not fully understood.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> Data concerning incidence, diagnosis, and treatment is scarce.</p><p id="par0010" class="elsevierStylePara elsevierViewall">We report the case of a 56-year-old female with penetrating CD (A2, L2, pB3), diagnosed at the age of 22. She was initially treated with infliximab, but therapy was switched to adalimumab and azathioprine as a result of severe infusion reaction (glottic edema). Due to refractory perianal disease, with multiple complex perianal fistulas, a diverting stoma was created at the age of 43 and was submitted to proctosigmoidectomy five years later. A fistula between the vagina and the supra-anal space persisted, but as the patient was asymptomatic, the previous therapy with adalimumab and azathioprine was maintained and she was kept on regular clinical, laboratory and imaging follow-up with magnetic resonance imaging (MRI), uneventfully. Eight years after the last surgery, she was admitted in gastroenterology ward due to pelvic pain and bloody fetid vaginal discharge. A pelvic MRI revealed a 3.9<span class="elsevierStyleHsp" style=""></span>cm nodular image with necrotic center, suggestive of an abscess in the posterior vaginal wall (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A). Abscess drainage under general anesthesia was performed, and biopsies of the fistula through the remnant rectovaginal septum revealed vaginal fistula tissue with marked infiltration by mucinous adenocarcinoma with an extensive component of signet-ring cells. At higher magnification, signet-ring cells with prominent intracytoplasmic mucin, displacing the nucleus to the periphery with nuclear molding (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>B). Gynecological examination using an endoscope revealed an ulcerated and infiltrative lesion in the vaginal wall (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>C). Due to non-specific bone pain, a bone scintigraphy was performed, which revealed multiple metastasis (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>D). She underwent pelvic palliative radiotherapy but died eight months after the diagnosis due to progression of the disease.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Malignant transformation, although rare, should be considered in the context of a non-healing fistula in CD.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a> There is no surveillance protocol, but a high index of suspicion and close follow-up is mandatory for an early diagnosis. Since symptoms are non-specific, high-risk patients (female gender, early-onset disease, long-standing Crohn's disease, chronic fistula and worsening of symptoms) should be regularly screened, including MRI, the best imaging modality, and biopsies under anesthesia of any masses or accessible fistulous tracts are mandatory for an early diagnosis.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a> The authors highlight this rare case of malignant transformation in a persistent fistula after surgery.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Informed consent</span><p id="par0020" class="elsevierStylePara elsevierViewall">The patient's relative 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">Francisco Vara-Luiz, Ivo Mendes, Carla Oliveira, Ana Pascoal and Cláudia Afonso wrote the manuscript; Ana Elisa Teles performed the histopathology evaluation; Marta Patita, Irina Mocanu and Ana Vieira performed clinical follow-up; Pedro Moniz Pereira performed the abscess drainage and biopsies; Vítor Fernandes and Jorge Fonseca 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>" "textoCompletoSecciones" => array:1 [ "secciones" => array:5 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Informed consent" ] 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: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" => 1190 "Ancho" => 1674 "Tamanyo" => 293654 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Pelvic magnetic resonance imaging revealing a 3.9<span class="elsevierStyleHsp" style=""></span>cm nodular image with necrotic center. (B) Histopathology showing signet ring mucinous adenocarcinoma. (C) Endoscope-guided gynecological examination showing an endoscope an ulcerated and infiltrative lesion in the vaginal wall. (D) Bone scan with multiple bone metastasis.</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" => "Perianal Crohn's fistula with malignant transformation" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "Z.Q. Ng" 1 => "N. Hew" 2 => "R. 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Cagir" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:5 [ "tituloSerie" => "J Gastrointest Surg" "fecha" => "2010" "volumen" => "14" "paginaInicial" => "66" "paginaFinal" => "73" ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0050" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Fistula-related cancer in Crohn's disease: a systematic review" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "A. Kotsafti" 1 => "M. Scarpa" 2 => "I. Angriman" 3 => "I. Castagliuolo" 4 => "A. Caruso" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:4 [ "tituloSerie" => "Cancers (Basel)" "fecha" => "2021" "volumen" => "13" "paginaInicial" => "1445" ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/02105705/0000004700000008/v3_202410150442/S0210570524000335/v3_202410150442/en/main.assets" "Apartado" => array:4 [ "identificador" => "9030" "tipo" => "SECCION" "es" => array:2 [ "titulo" => "Cartas científicas" "idiomaDefecto" => true ] "idiomaDefecto" => "es" ] "PDF" => "https://static.elsevier.es/multimedia/02105705/0000004700000008/v3_202410150442/S0210570524000335/v3_202410150442/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0210570524000335?idApp=UINPBA00004N" ]
Journal Information
Scientific letters
Malignant transformation in fistula after proctosigmoidectomy: A rare complication of long-standing Crohn's disease
Transformación maligna en fístula tras proctosigmoidectomía: una rara complicación de la enfermedad de Crohn de larga evolución
Francisco Vara-Luiza,b,
, Ivo Mendesa, Ana Elisa Telesc, Carla Oliveiraa, Ana Pascoala, Cláudia Afonsoa, Marta Patitaa, Irina Mocanua, Ana Vieiraa, Vítor Fernandesa, Pedro Moniz Pereirad, Jorge Fonsecaa,b
Corresponding author
a Gastroenterology Department, Hospital Garcia de Orta, Almada, Portugal
b Egas Moniz Center for Interdisciplinary Research (CiiEM), Egas Moniz School of Health and Science, Caparica, Portugal
c Pathology Department, Hospital Garcia de Orta, Almada, Portugal
d Surgery Department, Hospital Garcia de Orta, Almada, Portugal