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Gastritis cystica profunda mimicking a GIST – A diagnostic challenge
Gastritis cystica profunda simulando GIST – un caso de desafío diagnóstico
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(D) Immunoexpression in eccrine gland lumina of carcinoembryonic antigen (CEA× 200) (D1); tumoural areas with clear-cell change (H&E× 200) (D2); intense, diffuse positivity for p53 (p53× 200) (D3); and a neoplastic component with positive immunostaining for cytokeratin 8 (CK8× 200) (D4).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Ángel Pareja-López, Manuel Ferrer-Márquez, María del Mar Berenguel-Ibáñez, Natalia Espínola-Cortés, Francisco Javier Velasco-Albendea" "autores" => array:5 [ 0 => array:2 [ "nombre" => "Ángel" "apellidos" => "Pareja-López" ] 1 => array:2 [ "nombre" => "Manuel" "apellidos" => "Ferrer-Márquez" ] 2 => array:2 [ "nombre" => "María del Mar" "apellidos" => "Berenguel-Ibáñez" ] 3 => array:2 [ "nombre" => "Natalia" "apellidos" => "Espínola-Cortés" ] 4 => array:2 [ "nombre" => "Francisco Javier" "apellidos" => "Velasco-Albendea" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S021057051730167X" "doi" => "10.1016/j.gastrohep.2017.07.002" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S021057051730167X?idApp=UINPBA00004N" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S244438241830107X?idApp=UINPBA00004N" "url" => "/24443824/0000004100000007/v2_201810040621/S244438241830107X/v2_201810040621/en/main.assets" ] "itemAnterior" => array:19 [ "pii" => "S2444382418301287" "issn" => "24443824" "doi" => "10.1016/j.gastre.2018.07.001" "estado" => "S300" "fechaPublicacion" => "2018-08-01" "aid" => "1177" "copyright" => "Elsevier España, S.L.U." 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(A) Serous cystadenoma: multiple cystic structures, covered by cubic or cylindrical epithelial cells, with a clear cytoplasm and a round nucleus without atypia. 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Catarina Quadros, Liliane Meireles, Irina Alves, Paula Moura dos Santos, Fátima Serejo, Cristina Ferreira, José Paulo Freire, José Velosa" "autores" => array:9 [ 0 => array:4 [ "nombre" => "Joana Rita" "apellidos" => "Carvalho" "email" => array:1 [ 0 => "joana.rita.carvalho@gmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "Ana Catarina" "apellidos" => "Quadros" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "Liliane" "apellidos" => "Meireles" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 3 => array:3 [ "nombre" => "Irina" "apellidos" => "Alves" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 4 => array:3 [ "nombre" => "Paula" "apellidos" => "Moura dos Santos" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 5 => array:3 [ "nombre" => "Fátima" "apellidos" => "Serejo" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 6 => array:3 [ "nombre" => "Cristina" "apellidos" => "Ferreira" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 7 => array:3 [ "nombre" => "José Paulo" "apellidos" => "Freire" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] 8 => array:3 [ "nombre" => "José" "apellidos" => "Velosa" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Department of Gastroenterology and Hepatology, North Lisbon Hospital Center, Portugal" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Department of Pathology, North Lisbon Hospital Center, Portugal" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Department of General Surgery, North Lisbon Hospital Center, Portugal" "etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Gastritis cystica profunda simulando GIST – un caso de desafío diagnóstico" ] ] "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" => 636 "Ancho" => 2333 "Tamanyo" => 180829 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Upper gastrointestinal endoscopy showing a 40<span class="elsevierStyleHsp" style=""></span>mm polypoid lesion in the gastric body with normal mucosa surface and a central 15<span class="elsevierStyleHsp" style=""></span>mm ulcerated bleeding suggestive of a gastrointestinal stromal tumor.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Gastritis cystica profunda (GCP) is a rare hyperplastic lesion with unclear pathogenesis histologically characterized by the presence of gastric glands in the submucosa and even muscularis propria of the stomach with normal overlying mucosa.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,2</span></a> There are two histological patterns of gastritis cystica poliposa: gastritis cystica superficialis, in which cystic glands are limited to the mucosal layer; and GCP in which the cystic lesion locates within the submucosa and muscularis propria.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> Clinical manifestations of GCP are variable and can include gastrointestinal bleeding, epigastric pain and weight loss.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> An unspecified mucosal insult or injury is widely accepted as etiological mechanism but the pathophysiology is unknown.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1–4</span></a> We present a case of acute gastrointestinal bleeding caused by gastritis cystica profunda mimicking a gastrointestinal stromal tumor in a patient without previous gastric surgery.</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 67 year-old man was admitted for melena during 4 days associated with syncope. He had a previous history of atrial fibrillation and was medicated with warfarin. At admission, he was hemodynamically stable and had no abdominal pain. Laboratory tests revealed normocytic anemia with hemoglobin 8.8<span class="elsevierStyleHsp" style=""></span>g/dl (previous value: 13.5<span class="elsevierStyleHsp" style=""></span>g/dl) and an INR of 2.3. Upper gastrointestinal endoscopy (UGIE) revealed a 40<span class="elsevierStyleHsp" style=""></span>mm polypoid lesion in the gastric body with normal mucosa surface and a central 15<span class="elsevierStyleHsp" style=""></span>mm ulcerated bleeding as which was very suggestive of a gastrointestinal stromal tumor (GIST) (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). There were no esophageal or duodenal visible bleeding lesions. Biopsies performed during UGIE revealed aspects of chronic active gastritis and <span class="elsevierStyleItalic">Helicobacter pylori</span> was identified. Upper endoscopic ultrasonography (EUS) showed 40<span class="elsevierStyleHsp" style=""></span>mm submucosal a hypoechogenic and heterogeneous mass with cystic areas and no perilesional adenopathies. The EUS findings could also correspond to a GIST. EUS guided fine-needle aspiration (FNA) using 19 gauge needle was preformed but the sample was insufficient for evaluation. A full body computed tomography was performed and revealed a 4<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>6<span class="elsevierStyleHsp" style=""></span>cm gastric intraluminal lesion without signs of invasion or metastatic disease. The diagnosis of a gastric GIST was assumed and the patient was proposed to surgical resection of the lesion. Macroscopically, a polypoid lesion with a nodular surface and a central ulcer was observed, with multiple cysts and solid areas on cross-section (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Histologically (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>), the gastric mucosa showed focal lesions of chronic atrophic gastritis with activity, hemorrhage and ulceration, the submucosa and muscular propria displayed an abundant cystically dilated pyloric-type and foveolar-type glandular proliferation, without mitoses or atypia; surrounding the glands there was a thin layer of lamina propria and fibromuscular hyperplasia. The diagnosis of Gastritis cystica profunda was made. Surgical margins were free of lesion.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">In the majority of reported cases, GCP occurs in patients with a history of gastric surgery, in particular Billroth II procedure.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2,3</span></a> It is unclear if it is secondary to chronic inflammation as consequence of duodenal reflux, foreign body reaction or ischemic injury as a result of the surgery.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,2,5,6</span></a> Nevertheless, the interruption of the muscularis mucosae appears to allow migration of epithelial cells into the submucosal layer and subsequent cystic dilation.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2,7</span></a> In the unoperated stomach, the cause may be congenital in origin in patients with no prior gastric ulceration or trauma history.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> It is more common in men and most frequently develops in the gastric body,<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> as seen in this case. Our patient had a history of chronic atrophic gastritis with <span class="elsevierStyleItalic">H. pylori</span> infection which in this case can be considered as a possible etiological factor. The presentation symptoms are not specific and, endoscopically it is impossible to differentiate from other entities like polyps or GIST.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> As biopsy samples are restricted to the spared mucosal the results are often not diagnostic.<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3,8</span></a> CT can show a heterogeneously iso- to hypoattenuating intraluminal lesion with multiple small cysts but the appearance may be similar to GIST.<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3,9</span></a> On EUS, GCP can appear as a polymorphic, homogeneous cystic mass with a minimal solid component within the gastric mucosae which is also not specific of GCP.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,3</span></a> However, the accuracy of EUS-FNA with immunostaining in preoperative GIST, diagnosis has been reported at 91%–100%.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> Some patients might have to undergo gastric resection when it is impossible to make a definite diagnosis with radiologic study or endoscopic biopsy, as seen in this case. Case reports have revealed the possibility of a malignant transformation from GCP, even in an unoperated stomach, but the incidence of malignancy in GCP patients remains unknown.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> Epstein–Barr virus might have a role as a premalignant factor in cancer tissue with GCP.<span class="elsevierStyleSup">10</span> Given the lack of a pathognomonic endoscopic or radiographic appearance of GCP, diagnostic and surveillance guidelines are not available and further studies are required.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Funding</span><p id="par0020" class="elsevierStylePara elsevierViewall">No funding.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Conflicts of interest</span><p id="par0025" class="elsevierStylePara elsevierViewall">No conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:3 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Funding" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Conflicts of interest" ] 2 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 636 "Ancho" => 2333 "Tamanyo" => 180829 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Upper gastrointestinal endoscopy showing a 40<span class="elsevierStyleHsp" style=""></span>mm polypoid lesion in the gastric body with normal mucosa surface and a central 15<span class="elsevierStyleHsp" style=""></span>mm ulcerated bleeding suggestive of a gastrointestinal stromal tumor.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 2090 "Ancho" => 2333 "Tamanyo" => 763779 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Cross-section shows multiple cysts with diameters between 0.2 and 1.5<span class="elsevierStyleHsp" style=""></span>cm, filled with transparent and mucinous-looking liquid. (a) (H&E ×20): gastric wall including mucosa (#), submucosa (>) and muscularis propria (*) with abundant cystic glandular proliferation; (b) (H&E ×40): surrounding the glands there is a thin layer of lamina propria and fibromuscular hyperplasia; (c) (H&E ×100): the glands are lined by pyloric-type and foveolar-type epithelium, without mitoses or atypia.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Gastritis cystica profunda: endoscopic ultrasound findings and review of the literature" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "J. Machicado" 1 => "J. Shroff" 2 => "A. Quesada" 3 => "K. Jelinek" 4 => "M.P. Spinn" 5 => "L.D. 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| Year/Month | Html | Total | |
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