Extra-gastrointestinal stromal tumour. Report of primary tumour in the omentum | Revista Médica del Hospital General de México

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Arellano-López, R. Solalinde-Vargas, J.I. Guzmán-Mejía, L.G. González-Pérez, A.C. Zamora-García" "autores" => array:5 [ 0 => array:2 [ "nombre" => "P.R." "apellidos" => "Arellano-López" ] 1 => array:2 [ "nombre" => "R." "apellidos" => "Solalinde-Vargas" ] 2 => array:2 [ "nombre" => "J.I." "apellidos" => "Guzmán-Mejía" ] 3 => array:2 [ "nombre" => "L.G." "apellidos" => "González-Pérez" ] 4 => array:2 [ "nombre" => "A.C." 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Note increased length and thickness of muscle wall." ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "L.P. Islas-Domínguez, J.R. Jiménez-Jiménez, J. Cruz-Díaz, K. Castellanos Reyes" "autores" => array:4 [ 0 => array:2 [ "nombre" => "L.P." "apellidos" => "Islas-Domínguez" ] 1 => array:2 [ "nombre" => "J.R." "apellidos" => "Jiménez-Jiménez" ] 2 => array:2 [ "nombre" => "J." "apellidos" => "Cruz-Díaz" ] 3 => array:2 [ "nombre" => "K." "apellidos" => "Castellanos Reyes" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0185106317300057?idApp=UINPBA00004N" "url" => "/01851063/0000008100000004/v1_201810010958/S0185106317300057/v1_201810010958/en/main.assets" ] "en" => array:19 [ "idiomaDefecto" => true "cabecera" => "Clinical case" "titulo" => "Extra-gastrointestinal stromal tumour. Report of primary tumour in the omentum" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "221" "paginaFinal" => "225" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "E.N. Valdes-Peregrina, M. Hernández-González, O. de León-Pacheco, S. Mendoza-Ramírez" "autores" => array:4 [ 0 => array:4 [ "nombre" => "E.N." "apellidos" => "Valdes-Peregrina" "email" => array:1 [ 0 => "valdestefanie@yahoo.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "M." "apellidos" => "Hernández-González" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "b" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "O." "apellidos" => "de León-Pacheco" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "c" "identificador" => "aff0015" ] ] ] 3 => array:3 [ "nombre" => "S." "apellidos" => "Mendoza-Ramírez" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "d" "identificador" => "aff0020" ] ] ] ] "afiliaciones" => array:4 [ 0 => array:3 [ "entidad" => "Department of Pathology, Hospital General de México “Dr. Eduardo Liceaga”, Mexico City, Mexico" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "UNAM Faculty of Medicine, Mexico City, Mexico" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Department of Surgical Oncology, Hospital General de México “Dr. Eduardo Liceaga”, Mexico City, Mexico" "etiqueta" => "c" "identificador" => "aff0015" ] 3 => array:3 [ "entidad" => "Department of Pathology, Hospital Regional 1° de Octubre, ISSSTE, Mexico City, Mexico" "etiqueta" => "d" "identificador" => "aff0020" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author at: Hospital General de México “Dr. Eduardo Liceaga”, Dr. Balmis 148, Col. Doctores, Cuauhtémoc, 06726 Mexico City, Mexico. Tel.: +52 2789 2000." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Tumor del estroma extra-gastrointestinal. Informe de un caso de tumor primario en epiplón" ] ] "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" => 480 "Ancho" => 951 "Tamanyo" => 81160 ] ] "descripcion" => array:1 [ "en" => "Omental tumour (external surface). Size: 50cm×30cm×15cm. 3.5kg." ] ] ] "textoCompleto" => "IntroductionGastrointestinal stromal tumours (GISTs) are the most common primary mesenchymal tumour in the gastrointestinal tract, with an incidence of 11–15 people per every 100,000. They account for 1% of gastrointestinal neoplasms, and most present in adults and in men,<a class="elsevierStyleCrossRef" href="#bib0100">2</a> but there are cases reported in children.<a class="elsevierStyleCrossRef" href="#bib0105">3</a> Most of the reported E-GIST cases are described in elderly patients.<a class="elsevierStyleCrossRefs" href="#bib0110">4–6</a>More than 50% of GISTs are found in the stomach, 25–30% in the small intestine (jejunum and ileum), 5% in the colon and rectum, and 1% in the oesophagus. Cases have been reported in the mesentery and omentum,<a class="elsevierStyleCrossRefs" href="#bib0095">1,2</a> as well as in other organs, such as the pancreas,<a class="elsevierStyleCrossRef" href="#bib0125">7</a> uterus<a class="elsevierStyleCrossRef" href="#bib0130">8</a> and vaginal wall.<a class="elsevierStyleCrossRef" href="#bib0135">9</a> They originate from the interstitial cells of Cajal and present mutations in the c-KIT genes and platelet-derived growth factor receptor (PDGFRA) which leads to tyrosine kinase activation.<a class="elsevierStyleCrossRef" href="#bib0140">10</a> The clinical presentation of these tumours depends on their location and size; however, in advanced stages, they occur with abdominal pain, ileus, bleeding, anaemia and weight loss.<a class="elsevierStyleCrossRef" href="#bib0125">7</a>The importance of this case of primary E-GIST in the omentum involves its location and unusual presentation, since there are few cases reported in the literature; thus there is no reported incidence.Case reportA 48-year-old woman who was admitted to the Oncology Department of the Hospital General de México, with symptoms starting two years prior characterised by repeated urinary tract infections, treated with antibiotics and analgesics with little response. An increase in her waist circumference occurred later on with colic-like pain predominantly in both iliac fossae, which did not ease when analgesics were administered, accompanied by episodes of fever, with no weight loss. No signs of intestinal occlusion or any other gastrointestinal symptoms were reported. A well defined, mobile tumour in the pelvis was palpated during a physical examination, located up to 3cm above the navel, which was impossible to rule out on palpation to examine the rest of the abdominal/pelvic organs. Therefore, a clinical diagnosis of ovarian cancer was established. Serum tumours markers were negative, and a computerised tomography scan showed a solid, heterogeneous lesion in the pelvis, which was multi-lobulated with cystic degeneration and calcifications measuring 17cm×12cm×6cm with 636 cubic cm of fluid. There was also mesenteric lymph node growth. The omentum that was surgically resected was described as a “cluster of grapes” with implants with the same characteristics in the surface of the mesocolon, uterine surface, both ovary surfaces and uterine tubes. In the gastrointestinal tract, only an ileal occlusion was reported due to a superficial tumour implant, thus the superficial ileum implant and the rest of the implants were surgically resected and sent to the Surgical Pathology department for pathological study along with the omentum.In the surgery, the liver and stomach did not show any macroscopic abnormalities in their morphologies, nor any superficial implants in the omentum tumour. As mentioned above, the established clinical diagnosis was undifferentiated omentum carcinoma. It is important to mention that, in the imaging studies and during the surgery, no evidence was found of a tumour in the gastrointestinal tract.The Surgical Pathology department received the omentum (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), which measured 50cm×30cm×15cm and weight 3,500g. Its exterior surface presented multiple, well-defined nodules measuring from 1 to 15cm on the major axis that were light yellow in colour. On the slice surface, most of the nodules were solid, some with necrosis and haemorrhage, and one was cystic with blood inside (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Implants with the same characteristics were found on the round ligaments and the ileal serosa. There were no macroscopic abnormalities to the ovaries. A diffuse neoplasm composed of spindle cells and some epithelioid cells with clear cytoplasm and prominent nucleoli was found in the microscopic study, with 3 mitoses present in 50 high-power fields (<a class="elsevierStyleCrossRefs" href="#fig0015">Figs. 3 and 4</a>). There were areas of haemorrhage and liquefactive necrosis. The ovaries did not show microscopic abnormalities; therefore, a primary lesion of the ovaries was ruled out.<elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><elsevierMultimedia ident="fig0020"></elsevierMultimedia>Immunohistochemical studies were ordered with the following markers (<a class="elsevierStyleCrossRef" href="#tbl0005">Table 1</a>), which were positive for CD117 and DOG-1 (<a class="elsevierStyleCrossRef" href="#fig0025">Fig. 5</a>A and B). With all the above information, a primary extra-gastrointestinal stromal tumour in the omentum was diagnosed. Peritoneal leiomyomatosis was ruled out since the smooth muscle actin and muscle-specific actin markers were negative, as well as spindle cell peritoneal mesothelium since the calretinin and WT1 markers were negative.<elsevierMultimedia ident="tbl0005"></elsevierMultimedia><elsevierMultimedia ident="fig0025"></elsevierMultimedia>The patient progressed adequately, with no signs of intestinal obstruction and no abdominal pain, with no clinical or laboratory signs of urinary tract infection. She was discharged 10 days after the surgery due to improvement. She later started treatment with imatinib as she was a candidate since the CD117 marker was positive. Three months later, she reported episodes of bleeding when defecating, bone pain, and “hot flushes” which stopped, and she continued the treatment with imatinib. At four months, she presented with signs of drug-induced grade IV neutropenia, as well as late, postprandial colic-like pain, with no signs of peritoneal irritation and the treatment was discontinued. One week later, she recovered from the neutropenia, and reported dysuria on one occasion and pain in the left flank near the kidneys. The last follow-up was at 5 months, and she reported generalised pain. No palpable tumour was found during the physical examination nor were any signs of clinical tumour activity, with signs of grade II neutropenia. Plans were made to reinitiate treatment with imatinib.DiscussionMacroscopically, GISTs present as nodules in the stroma or subserosa in the gastrointestinal tract.<a class="elsevierStyleCrossRef" href="#bib0100">2</a> The E-GISTs reported in the literature are very similar to that of our case, and are described as a large tumour with multiple nodules, some red in colour, others a light yellow, with areas of haemorrhage and necrosis.<a class="elsevierStyleCrossRefs" href="#bib0095">1,2,4,6,11,12</a>Microscopically, these tumours can be classified as spindle cell (70%), epithelioid (20%), and mixed (10%).<a class="elsevierStyleCrossRef" href="#bib0135">9</a> This case was classified as mixed since she had both types of cells. Some had enlarged nuclei and were hyperchromic which is associated with higher tumour aggression. The mitosis can vary from little to abundant, and some can present atypical mitoses.<a class="elsevierStyleCrossRef" href="#bib0100">2</a>These tumours are characterised by c-KIT gene mutations, or PDGFRA mutations; both genes lead to tyrosine kinase activation.<a class="elsevierStyleCrossRef" href="#bib0140">10</a> The cases with c-KIT mutation are positive for the CD117 marker,<a class="elsevierStyleCrossRefs" href="#bib0095">1,2</a> and adequately response to imatinib therapy.<a class="elsevierStyleCrossRef" href="#bib0140">10</a> Cases with PDGFRA mutations are usually negative for the CD117 marker and are called “wild-type”. In these cases, another marker such as DOG-1 (discovered in GIST-1) has been found to have a higher specificity and sensitivity than CD117,<a class="elsevierStyleCrossRef" href="#bib0155">13</a> and is used to confirm GIST diagnosis.<a class="elsevierStyleCrossRefs" href="#bib0100">2,10</a> It cannot be stated that CD117 or DOG-1 are expressed exclusively in GISTs, since although they are highly specific for them, they are also rarely expressed in other tumours such as leiomyosarcomas, leiomyomas, and leiomyoblastomas, among others.<a class="elsevierStyleCrossRef" href="#bib0160">14</a> The CD34 marker can also be used in GISTs, but it is not specific and is only expressed in 25% of cases.<a class="elsevierStyleCrossRefs" href="#bib0100">2,5,12</a> In E-GIST cases where the CD34 marker test has been performed, the results have been reported to be negative,<a class="elsevierStyleCrossRefs" href="#bib0115">5,12</a> as in our case.Risk stratification in these tumours has been classified based on the tumour size (0.1, 2, 5, 10, 15 and 25cm), the number of mitoses (0, 2, 5, 10, 15, 25 and 50 mitoses in 50 high-power fields) and primary tumours site (gastric with or without rupture, non-gastric with or without rupture, and extra-gastrointestinal with or without rupture).<a class="elsevierStyleCrossRef" href="#bib0100">2</a> In this case, due to the large size over 10cm, with fewer than 5 mitoses and being extra-gastrointestinal without rupture, it was considered high risk. The treatment of choice is surgical resection and treatment with tyrosine kinase inhibitors such as imatinib.<a class="elsevierStyleCrossRefs" href="#bib0095">1,2,15</a> In advanced cases, liver metastasis is present most of the time, whereas metastasis occurs less frequently in other organs such as in the ovaries. Metastasis in the lymph nodes is rare.<a class="elsevierStyleCrossRefs" href="#bib0095">1,2</a> In this case, the patient presented multiple superficial abdominal implants.The risk of recurrence has been associated with the loss of the c-KIT gene.<a class="elsevierStyleCrossRef" href="#bib0100">2</a> It usually occurs in the first two years,<a class="elsevierStyleCrossRef" href="#bib0095">1</a> involving the liver and the peritoneal surface.<a class="elsevierStyleCrossRef" href="#bib0105">3</a>The literature describes primary carcinoma of the peritoneum as a rare tumour that originates in the pelvic and abdominal peritoneum. It is believed to come from a secondary Müllerian system that triggers the start of a primary carcinoma resembling serous papillary carcinoma of the ovary in its histological appearance, spread, treatment and prognosis.<a class="elsevierStyleCrossRef" href="#bib0170">16</a> In this case, the initial focus was as a primary neoplasm of the omentum with a spindle cell pattern. It was thought to be a diffuse peritoneal leiomyomatosis due to the predominance of spindle cells, low mitotic count and no pleomorphism. This was ruled out with the negative markers for smooth muscle actin and muscle-specific actin.<a class="elsevierStyleCrossRef" href="#bib0175">17</a> It was also thought to be peritoneal spindle cell mesothelioma, which was ruled out with the negative markers for calretinin and WT1, which are macroscopically very similar and present as peritoneal nodules.<a class="elsevierStyleCrossRef" href="#bib0180">18</a> As in this case, carcinomas and leiomyosarcomas should be ruled out for large tumours in the omentum.<a class="elsevierStyleCrossRef" href="#bib0100">2</a> Since there is little information about this tumour, it should be considered in the differential diagnosis of patients with large, multi-nodular, primary lesions of the mesentery and omentum.<a class="elsevierStyleCrossRef" href="#bib0095">1</a>As for patient symptoms, there is no correlation with GISTs in the gastrointestinal tract.ConclusionE-GISTs are very rare. This group of tumours are not well known and involve a large number of diagnostic and therapeutic issues due to the lack of information about their behaviour. Despite generalised knowledge about gastrointestinal stromal tumours, there is a lack of data on patients with E-GIST. Therefore, this type of publication can help to provide useful information on its diagnosis, prognosis and treatment. The diagnosis was of great importance since the treatment changes from one tumour (peritoneal leiomyomatosis or peritoneal mesothelioma) to the next (E-GIST).Ethical disclosuresProtection of human and animal subjectsThe authors declare that no experiments were performed on humans or animals for this study.Confidentiality of dataThe authors declare that they have followed the protocols of their work centre on the publication of patient data.Right to privacy and informed consentThe authors have obtained the written informed consent of the patients or subjects mentioned in the article. The corresponding author is in possession of this document.Conflict of interestThe authors declare that they have no conflict of interests." 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More than half of these originate in the stomach, the rest in the small intestine, colon, rectum and oesophagus, and rarely in the omentum. Case reportA 48-year-old woman, with painful abdominal growth developing over two years. Radiological studies showed a heterogeneous solid lesion in the pelvis, so she underwent surgery and the omentum was resected, no evidence of a tumour was found in the gastrointestinal tract. The omentum measured 50cm×30cm×15cm. Its surface was heterogeneous and showed multiple nodules of varying sizes. A microscopically diffuse pattern of spindle cells was observed, some of them epithelioid, with three mitoses in 50 high-power fields. The immunohistochemical study is positive for CD117 and DOG-1.E-GIST should be considered for the differential diagnosis of patients with multinodular lesions in the omentum and mesentery." "secciones" => array:2 [ 0 => array:1 [ "identificador" => "abst0005" ] 1 => array:2 [ "identificador" => "abst0010" "titulo" => "Case report" ] ] ] "es" => array:3 [ "titulo" => "Resumen" "resumen" => "El tumor del estroma gastrointestinal (GIST) es el tumor mesenquimatoso más frecuente del tubo digestivo. Más de la mitad de estos se originan en estómago, el resto en intestino delgado, colon, recto y esófago; excepcionalmente en epiplón. Caso clínicoMujer de 48 años con crecimiento doloroso abdominal de 2 años de evolución. Los estudios radiológicos mostraron una lesión sólida heterogénea en pelvis, por lo que fue intervenida quirúrgicamente y se resecó epiplón, no se encontró evidencia de tumor en el tracto gastrointestinal. El epiplón midió 50×30×15cm. Su superficie es heterogénea, presenta múltiples nódulos de diferentes tamaños. Microscópicamente se observa un patrón difuso de células fusiformes, algunas de ellas epitelioides con 3 mitosis en 50 campos de alta resolución. El estudio de inmunohistoquímica es positivo para CD117 y DOG-1.El E-GIST, se debe considerar en el diagnóstico diferencial de pacientes con lesiones multinodulares en epiplón y mesenterio." "secciones" => array:2 [ 0 => array:1 [ "identificador" => "abst0015" ] 1 => array:2 [ "identificador" => "abst0020" "titulo" => "Caso clínico" ] ] ] ] "multimedia" => array:6 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 480 "Ancho" => 951 "Tamanyo" => 81160 ] ] "descripcion" => array:1 [ "en" => "Omental tumour (external surface). Size: 50cm×30cm×15cm. 3.5kg." ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 608 "Ancho" => 975 "Tamanyo" => 89090 ] ] "descripcion" => array:1 [ "en" => "Omental tumour (cut surface)." ] ] 2 => array:7 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 700 "Ancho" => 975 "Tamanyo" => 212798 ] ] "descripcion" => array:1 [ "en" => "Optical microscopy of solid spindle cell neoplasm (10×)." ] ] 3 => array:7 [ "identificador" => "fig0020" "etiqueta" => "Figure 4" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr4.jpeg" "Alto" => 688 "Ancho" => 975 "Tamanyo" => 139046 ] ] "descripcion" => array:1 [ "en" => "Optical microscopy of solid epithelial neoplasm (40×)." ] ] 4 => array:7 [ "identificador" => "fig0025" "etiqueta" => "Figure 5" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr5.jpeg" "Alto" => 513 "Ancho" => 1500 "Tamanyo" => 273790 ] ] "descripcion" => array:1 [ "en" => "(A) Positive CD117 marker (cytoplasm). 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Clinical case

Extra-gastrointestinal stromal tumour. Report of primary tumour in the omentum

Tumor del estroma extra-gastrointestinal. Informe de un caso de tumor primario en epiplón

E.N. Valdes-Peregrinaa,

Corresponding author

valdestefanie@yahoo.com

Corresponding author at: Hospital General de México “Dr. Eduardo Liceaga”, Dr. Balmis 148, Col. Doctores, Cuauhtémoc, 06726 Mexico City, Mexico. Tel.: +52 2789 2000.

, M. Hernández-Gonzáleza,b, O. de León-Pachecoc, S. Mendoza-Ramíreza,d

a Department of Pathology, Hospital General de México “Dr. Eduardo Liceaga”, Mexico City, Mexico

b UNAM Faculty of Medicine, Mexico City, Mexico

c Department of Surgical Oncology, Hospital General de México “Dr. Eduardo Liceaga”, Mexico City, Mexico

d Department of Pathology, Hospital Regional 1° de Octubre, ISSSTE, Mexico City, Mexico

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of GISTs are found in the stomach&#44; 25&#8211;30&#37; in the small intestine &#40;jejunum and ileum&#41;&#44; 5&#37; in the colon and rectum&#44; and 1&#37; in the oesophagus&#46; Cases have been reported in the mesentery and omentum&#44;<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">1&#44;2</span></a> as well as in other organs&#44; such as the pancreas&#44;<a class="elsevierStyleCrossRef" href="#bib0125"><span class="elsevierStyleSup">7</span></a> uterus<a class="elsevierStyleCrossRef" href="#bib0130"><span class="elsevierStyleSup">8</span></a> and vaginal wall&#46;<a class="elsevierStyleCrossRef" href="#bib0135"><span class="elsevierStyleSup">9</span></a> They originate from the interstitial cells of Cajal and present mutations in the c-KIT genes and platelet-derived growth factor receptor &#40;PDGFRA&#41; which leads to tyrosine kinase activation&#46;<a class="elsevierStyleCrossRef" href="#bib0140"><span class="elsevierStyleSup">10</span></a> The clinical presentation of these tumours depends on their location and size&#59; however&#44; in advanced stages&#44; they occur with abdominal pain&#44; ileus&#44; bleeding&#44; anaemia and weight loss&#46;<a class="elsevierStyleCrossRef" href="#bib0125"><span class="elsevierStyleSup">7</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">The importance of this case of primary E-GIST in the omentum involves its location and unusual presentation&#44; since there are few cases reported in the literature&#59; thus there is no reported incidence&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Case report</span><p id="par0020" class="elsevierStylePara elsevierViewall">A 48-year-old woman who was admitted to the Oncology Department of the Hospital General de M&#233;xico&#44; with symptoms starting two years prior characterised by repeated urinary tract infections&#44; treated with antibiotics and analgesics with little response&#46; An increase in her waist circumference occurred later on with colic-like pain predominantly in both iliac fossae&#44; which did not ease when analgesics were administered&#44; accompanied by episodes of fever&#44; with no weight loss&#46; No signs of intestinal occlusion or any other gastrointestinal symptoms were reported&#46; A well defined&#44; mobile tumour in the pelvis was palpated during a physical examination&#44; located up to 3<span class="elsevierStyleHsp" style=""></span>cm above the navel&#44; which was impossible to rule out on palpation to examine the rest of the abdominal&#47;pelvic organs&#46; Therefore&#44; a clinical diagnosis of ovarian cancer was established&#46; Serum tumours markers were negative&#44; and a computerised tomography scan showed a solid&#44; heterogeneous lesion in the pelvis&#44; which was multi-lobulated with cystic degeneration and calcifications measuring 17<span class="elsevierStyleHsp" style=""></span>cm<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>12<span class="elsevierStyleHsp" style=""></span>cm<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>6<span class="elsevierStyleHsp" style=""></span>cm with 636 cubic cm of fluid&#46; There was also mesenteric lymph node growth&#46; The omentum that was surgically resected was described as a &#8220;cluster of grapes&#8221; with implants with the same characteristics in the surface of the mesocolon&#44; uterine surface&#44; both ovary surfaces and uterine tubes&#46; In the gastrointestinal tract&#44; only an ileal occlusion was reported due to a superficial tumour implant&#44; thus the superficial ileum implant and the rest of the implants were surgically resected and sent to the Surgical Pathology department for pathological study along with the omentum&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">In the surgery&#44; the liver and stomach did not show any macroscopic abnormalities in their morphologies&#44; nor any superficial implants in the omentum tumour&#46; As mentioned above&#44; the established clinical diagnosis was undifferentiated omentum carcinoma&#46; It is important to mention that&#44; in the imaging studies and during the surgery&#44; no evidence was found of a tumour in the gastrointestinal tract&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">The Surgical Pathology department received the omentum &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#44; which measured 50<span class="elsevierStyleHsp" style=""></span>cm<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>30<span class="elsevierStyleHsp" style=""></span>cm<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>15<span class="elsevierStyleHsp" style=""></span>cm and weight 3&#44;500<span class="elsevierStyleHsp" style=""></span>g&#46; Its exterior surface presented multiple&#44; well-defined nodules measuring from 1 to 15<span class="elsevierStyleHsp" style=""></span>cm on the major axis that were light yellow in colour&#46; On the slice surface&#44; most of the nodules were solid&#44; some with necrosis and haemorrhage&#44; and one was cystic with blood inside &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Implants with the same characteristics were found on the round ligaments and the ileal serosa&#46; There were no macroscopic abnormalities to the ovaries&#46; A diffuse neoplasm composed of spindle cells and some epithelioid cells with clear cytoplasm and prominent nucleoli was found in the microscopic study&#44; with 3 mitoses present in 50 high-power fields &#40;<a class="elsevierStyleCrossRefs" href="#fig0015">Figs&#46; 3 and 4</a>&#41;&#46; There were areas of haemorrhage and liquefactive necrosis&#46; The ovaries did not show microscopic abnormalities&#59; therefore&#44; a primary lesion of the ovaries was ruled out&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><elsevierMultimedia ident="fig0020"></elsevierMultimedia><p id="par0035" class="elsevierStylePara elsevierViewall">Immunohistochemical studies were ordered with the following markers &#40;<a class="elsevierStyleCrossRef" href="#tbl0005">Table 1</a>&#41;&#44; which were positive for CD117 and DOG-1 &#40;<a class="elsevierStyleCrossRef" href="#fig0025">Fig&#46; 5</a>A and B&#41;&#46; With all the above information&#44; a primary extra-gastrointestinal stromal tumour in the omentum was diagnosed&#46; Peritoneal leiomyomatosis was ruled out since the smooth muscle actin and muscle-specific actin markers were negative&#44; as well as spindle cell peritoneal mesothelium since the calretinin and WT1 markers were negative&#46;</p><elsevierMultimedia ident="tbl0005"></elsevierMultimedia><elsevierMultimedia ident="fig0025"></elsevierMultimedia><p id="par0040" class="elsevierStylePara elsevierViewall">The patient progressed adequately&#44; with no signs of intestinal obstruction and no abdominal pain&#44; with no clinical or laboratory signs of urinary tract infection&#46; She was discharged 10 days after the surgery due to improvement&#46; She later started treatment with imatinib as she was a candidate since the CD117 marker was positive&#46; Three months later&#44; she reported episodes of bleeding when defecating&#44; bone pain&#44; and &#8220;hot flushes&#8221; which stopped&#44; and she continued the treatment with imatinib&#46; At four months&#44; she presented with signs of drug-induced grade IV neutropenia&#44; as well as late&#44; postprandial colic-like pain&#44; with no signs of peritoneal irritation and the treatment was discontinued&#46; One week later&#44; she recovered from the neutropenia&#44; and reported dysuria on one occasion and pain in the left flank near the kidneys&#46; The last follow-up was at 5 months&#44; and she reported generalised pain&#46; No palpable tumour was found during the physical examination nor were any signs of clinical tumour activity&#44; with signs of grade II neutropenia&#46; Plans were made to reinitiate treatment with imatinib&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Discussion</span><p id="par0045" class="elsevierStylePara elsevierViewall">Macroscopically&#44; GISTs present as nodules in the stroma or subserosa in the gastrointestinal tract&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">2</span></a> The E-GISTs reported in the literature are very similar to that of our case&#44; and are described as a large tumour with multiple nodules&#44; some red in colour&#44; others a light yellow&#44; with areas of haemorrhage and necrosis&#46;<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">1&#44;2&#44;4&#44;6&#44;11&#44;12</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Microscopically&#44; these tumours can be classified as spindle cell &#40;70&#37;&#41;&#44; epithelioid &#40;20&#37;&#41;&#44; and mixed &#40;10&#37;&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0135"><span class="elsevierStyleSup">9</span></a> This case was classified as mixed since she had both types of cells&#46; Some had enlarged nuclei and were hyperchromic which is associated with higher tumour aggression&#46; The mitosis can vary from little to abundant&#44; and some can present atypical mitoses&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">2</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">These tumours are characterised by c-KIT gene mutations&#44; or PDGFRA mutations&#59; both genes lead to tyrosine kinase activation&#46;<a class="elsevierStyleCrossRef" href="#bib0140"><span class="elsevierStyleSup">10</span></a> The cases with c-KIT mutation are positive for the CD117 marker&#44;<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">1&#44;2</span></a> and adequately response to imatinib therapy&#46;<a class="elsevierStyleCrossRef" href="#bib0140"><span class="elsevierStyleSup">10</span></a> Cases with PDGFRA mutations are usually negative for the CD117 marker and are called &#8220;wild-type&#8221;&#46; In these cases&#44; another marker such as DOG-1 &#40;discovered in GIST-1&#41; has been found to have a higher specificity and sensitivity than CD117&#44;<a class="elsevierStyleCrossRef" href="#bib0155"><span class="elsevierStyleSup">13</span></a> and is used to confirm GIST diagnosis&#46;<a class="elsevierStyleCrossRefs" href="#bib0100"><span class="elsevierStyleSup">2&#44;10</span></a> It cannot be stated that CD117 or DOG-1 are expressed exclusively in GISTs&#44; since although they are highly specific for them&#44; they are also rarely expressed in other tumours such as leiomyosarcomas&#44; leiomyomas&#44; and leiomyoblastomas&#44; among others&#46;<a class="elsevierStyleCrossRef" href="#bib0160"><span class="elsevierStyleSup">14</span></a> The CD34 marker can also be used in GISTs&#44; but it is not specific and is only expressed in 25&#37; of cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0100"><span class="elsevierStyleSup">2&#44;5&#44;12</span></a> In E-GIST cases where the CD34 marker test has been performed&#44; the results have been reported to be negative&#44;<a class="elsevierStyleCrossRefs" href="#bib0115"><span class="elsevierStyleSup">5&#44;12</span></a> as in our case&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">Risk stratification in these tumours has been classified based on the tumour size &#40;0&#46;1&#44; 2&#44; 5&#44; 10&#44; 15 and 25<span class="elsevierStyleHsp" style=""></span>cm&#41;&#44; the number of mitoses &#40;0&#44; 2&#44; 5&#44; 10&#44; 15&#44; 25 and 50 mitoses in 50 high-power fields&#41; and primary tumours site &#40;gastric with or without rupture&#44; non-gastric with or without rupture&#44; and extra-gastrointestinal with or without rupture&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">2</span></a> In this case&#44; due to the large size over 10<span class="elsevierStyleHsp" style=""></span>cm&#44; with fewer than 5 mitoses and being extra-gastrointestinal without rupture&#44; it was considered high risk&#46; The treatment of choice is surgical resection and treatment with tyrosine kinase inhibitors such as imatinib&#46;<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">1&#44;2&#44;15</span></a> In advanced cases&#44; liver metastasis is present most of the time&#44; whereas metastasis occurs less frequently in other organs such as in the ovaries&#46; Metastasis in the lymph nodes is rare&#46;<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">1&#44;2</span></a> In this case&#44; the patient presented multiple superficial abdominal implants&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">The risk of recurrence has been associated with the loss of the c-KIT gene&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">2</span></a> It usually occurs in the first two years&#44;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">1</span></a> involving the liver and the peritoneal surface&#46;<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">3</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">The literature describes primary carcinoma of the peritoneum as a rare tumour that originates in the pelvic and abdominal peritoneum&#46; It is believed to come from a secondary M&#252;llerian system that triggers the start of a primary carcinoma resembling serous papillary carcinoma of the ovary in its histological appearance&#44; spread&#44; treatment and prognosis&#46;<a class="elsevierStyleCrossRef" href="#bib0170"><span class="elsevierStyleSup">16</span></a> In this case&#44; the initial focus was as a primary neoplasm of the omentum with a spindle cell pattern&#46; It was thought to be a diffuse peritoneal leiomyomatosis due to the predominance of spindle cells&#44; low mitotic count and no pleomorphism&#46; This was ruled out with the negative markers for smooth muscle actin and muscle-specific actin&#46;<a class="elsevierStyleCrossRef" href="#bib0175"><span class="elsevierStyleSup">17</span></a> It was also thought to be peritoneal spindle cell mesothelioma&#44; which was ruled out with the negative markers for calretinin and WT1&#44; which are macroscopically very similar and present as peritoneal nodules&#46;<a class="elsevierStyleCrossRef" href="#bib0180"><span class="elsevierStyleSup">18</span></a> As in this case&#44; carcinomas and leiomyosarcomas should be ruled out for large tumours in the omentum&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">2</span></a> Since there is little information about this tumour&#44; it should be considered in the differential diagnosis of patients with large&#44; multi-nodular&#44; primary lesions of the mesentery and omentum&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">1</span></a></p><p id="par0075" class="elsevierStylePara elsevierViewall">As for patient symptoms&#44; there is no correlation with GISTs in the gastrointestinal tract&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Conclusion</span><p id="par0080" class="elsevierStylePara elsevierViewall">E-GISTs are very rare&#46; This group of tumours are not well known and involve a large number of diagnostic and therapeutic issues due to the lack of information about their behaviour&#46; Despite generalised knowledge about gastrointestinal stromal tumours&#44; there is a lack of data on patients with E-GIST&#46; Therefore&#44; this type of publication can help to provide useful information on its diagnosis&#44; prognosis and treatment&#46; The diagnosis was of great importance since the treatment changes from one tumour &#40;peritoneal leiomyomatosis or peritoneal mesothelioma&#41; to the next &#40;E-GIST&#41;&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Ethical disclosures</span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Protection of human and animal subjects</span><p id="par0085" class="elsevierStylePara elsevierViewall">The authors declare that no experiments were performed on humans or animals for this study&#46;</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0065">Confidentiality of data</span><p id="par0090" class="elsevierStylePara elsevierViewall">The authors declare that they have followed the protocols of their work centre on the publication of patient data&#46;</p></span><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0070">Right to privacy and informed consent</span><p id="par0095" class="elsevierStylePara elsevierViewall">The authors have obtained the written informed consent of the patients or subjects mentioned in the article&#46; The corresponding author is in possession of this document&#46;</p></span></span><span id="sec0045" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0075">Conflict of interest</span><p id="par0100" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflict of interests&#46;</p></span></span>"
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        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Gastrointestinal stromal tumours &#40;GISTs&#41; are the most common mesenchymal tumour in the gastrointestinal tract&#46; More than half of these originate in the stomach&#44; the rest in the small intestine&#44; colon&#44; rectum and oesophagus&#44; and rarely in the omentum&#46;</p></span> <span id="abst0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Case report</span><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A 48-year-old woman&#44; with painful abdominal growth developing over two years&#46; Radiological studies showed a heterogeneous solid lesion in the pelvis&#44; so she underwent surgery and the omentum was resected&#44; no evidence of a tumour was found in the gastrointestinal tract&#46; The omentum measured 50<span class="elsevierStyleHsp" style=""></span>cm<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>30<span class="elsevierStyleHsp" style=""></span>cm<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>15<span class="elsevierStyleHsp" style=""></span>cm&#46; Its surface was heterogeneous and showed multiple nodules of varying sizes&#46; A microscopically diffuse pattern of spindle cells was observed&#44; some of them epithelioid&#44; with three mitoses in 50 high-power fields&#46; The immunohistochemical study is positive for CD117 and DOG-1&#46;</p><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">E-GIST should be considered for the differential diagnosis of patients with multinodular lesions in the omentum and mesentery&#46;</p></span>"
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        "resumen" => "<span id="abst0015" class="elsevierStyleSection elsevierViewall"><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">El tumor del estroma gastrointestinal &#40;GIST&#41; es el tumor mesenquimatoso m&#225;s frecuente del tubo digestivo&#46; M&#225;s de la mitad de estos se originan en est&#243;mago&#44; el resto en intestino delgado&#44; colon&#44; recto y es&#243;fago&#59; excepcionalmente en epipl&#243;n&#46;</p></span> <span id="abst0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Caso cl&#237;nico</span><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Mujer de 48 a&#241;os con crecimiento doloroso abdominal de 2 a&#241;os de evoluci&#243;n&#46; Los estudios radiol&#243;gicos mostraron una lesi&#243;n s&#243;lida heterog&#233;nea en pelvis&#44; por lo que fue intervenida quir&#250;rgicamente y se resec&#243; epipl&#243;n&#44; no se encontr&#243; evidencia de tumor en el tracto gastrointestinal&#46; El epipl&#243;n midi&#243; 50<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>30<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>15<span class="elsevierStyleHsp" style=""></span>cm&#46; Su superficie es heterog&#233;nea&#44; presenta m&#250;ltiples n&#243;dulos de diferentes tama&#241;os&#46; Microsc&#243;picamente se observa un patr&#243;n difuso de c&#233;lulas fusiformes&#44; algunas de ellas epitelioides con 3 mitosis en 50 campos de alta resoluci&#243;n&#46; El estudio de inmunohistoqu&#237;mica es positivo para CD117 y DOG-1&#46;</p><p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">El E-GIST&#44; se debe considerar en el diagn&#243;stico diferencial de pacientes con lesiones multinodulares en epipl&#243;n y mesenterio&#46;</p></span>"
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                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Primary extra-gastrointestinal stromal tumor &#40;GIST&#41; arising from mesentery of small bowel and presenting as abdominal mass&#58; a rare entity"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => true
                          "autores" => array:3 [
                            0 => "K&#46;T&#46; Alok"
                            1 => "K&#46;C&#46; Anil"
                            2 => "K&#46; Hemant"
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                    ]
                  ]
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                    0 => array:1 [
                      "Revista" => array:5 [
                        "tituloSerie" => "Open J Gastroenterol"
                        "fecha" => "2013"
                        "volumen" => "3"
                        "paginaInicial" => "267"
                        "paginaFinal" => "271"
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                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "The changing face of GIST&#58; implications for pathologists"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:2 [
                            0 => "H&#46; Chris"
                            1 => "Y&#46; Desmond"
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Article information

ISSN: 01851063
Original language: English

DOI:10.1016/j.hgmx.2016.12.002

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Year/Month	Html	Pdf	Total

2024 October	35	15	50
2024 September	120	20	140
2024 August	118	16	134
2024 July	118	17	135
2024 June	116	18	134
2024 May	113	17	130
2024 April	89	20	109
2024 March	104	17	121
2024 February	102	19	121
2024 January	120	17	137
2023 December	95	15	110
2023 November	135	17	152
2023 October	153	34	187
2023 September	153	23	176
2023 August	87	15	102
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2023 April	53	23	76
2023 March	67	21	88
2023 February	59	5	64
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2022 December	87	18	105
2022 November	56	9	65
2022 October	61	8	69
2022 September	61	17	78
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2022 May	57	11	68
2022 April	72	23	95
2022 March	67	16	83
2022 February	74	18	92
2022 January	73	13	86
2021 December	68	18	86
2021 November	67	16	83
2021 October	66	26	92
2021 September	67	22	89
2021 August	48	14	62
2021 July	54	11	65
2021 June	68	15	83
2021 May	67	11	78
2021 April	127	8	135
2021 March	80	17	97
2021 February	56	17	73
2021 January	54	16	70
2020 December	58	13	71
2020 November	48	11	59
2020 October	33	4	37
2020 September	42	9	51
2020 August	41	14	55
2020 July	32	11	43
2020 June	33	11	44
2020 May	52	6	58
2020 April	36	10	46
2020 March	37	15	52
2020 February	47	7	54
2020 January	37	10	47
2019 December	40	7	47
2019 November	24	2	26
2019 October	31	15	46
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2019 August	16	6	22
2019 July	32	17	49
2019 June	70	17	87
2019 May	180	15	195
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2018 December	22	3	25
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2018 October	35	7	42
2018 September	5	5	10
2018 August	0	6	6
2018 July	1	2	3
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2018 May	2	2	4
2018 April	1	1	2
2018 March	1	1	2
2018 January	4	1	5
2017 December	0	3	3
2017 November	5	0	5
2017 October	11	1	12
2017 September	6	7	13
2017 August	3	2	5
2017 July	4	1	5
2017 June	1	8	9
2017 May	6	6	12
2017 April	6	4	10
2017 March	12	20	32
2017 February	2	8	10

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