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"documento" => "article" "crossmark" => 0 "subdocumento" => "fla" "cita" => "Rev Clin Esp. 2014;214:31-7" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:2 [ "total" => 287 "HTML" => 287 ] "en" => array:13 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Special article</span>" "titulo" => "Guidelines on the management of implantable cardioverter defibrillators at the end of life" "tienePdf" => "en" "tieneTextoCompleto" => "en" "tieneResumen" => array:2 [ 0 => "en" 1 => "es" ] "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "31" "paginaFinal" => "37" ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Guía sobre el manejo de desfibriladores automáticos implantables al final de la vida" ] ] "contieneResumen" => array:2 [ "en" => true "es" => true ] "contieneTextoCompleto" => array:1 [ "en" => true ] "contienePdf" => array:1 [ "en" => true ] "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" => 2228 "Ancho" => 3000 "Tamanyo" => 432302 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Algorithm with the steps to take when the patient requests the disconnection of the implantable cardioverter defibrillator (ICD) until the deactivation has been performed.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "T. Datino, L. Rexach, M.T. Vidán, A. Alonso, Á. Gándara, J. Ruiz-García, B. Fontecha, M. Martínez-Sellés" "autores" => array:8 [ 0 => array:2 [ "nombre" => "T." "apellidos" => "Datino" ] 1 => array:2 [ "nombre" => "L." "apellidos" => "Rexach" ] 2 => array:2 [ "nombre" => "M.T." "apellidos" => "Vidán" ] 3 => array:2 [ "nombre" => "A." "apellidos" => "Alonso" ] 4 => array:2 [ "nombre" => "Á." "apellidos" => "Gándara" ] 5 => array:2 [ "nombre" => "J." "apellidos" => "Ruiz-García" ] 6 => array:2 [ "nombre" => "B." "apellidos" => "Fontecha" ] 7 => array:2 [ "nombre" => "M." "apellidos" => "Martínez-Sellés" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S0014256513002828" "doi" => "10.1016/j.rce.2013.08.009" "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/S0014256513002828?idApp=WRCEE" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S225488741300129X?idApp=WRCEE" "url" => "/22548874/0000021400000001/v1_201402030902/S225488741300129X/v1_201402030902/en/main.assets" ] "itemAnterior" => array:19 [ "pii" => "S2254887413000982" "issn" => "22548874" "doi" => "10.1016/j.rceng.2013.01.006" "estado" => "S300" "fechaPublicacion" => "2014-01-01" "aid" => "738" "copyright" => "Elsevier España, S.L." "documento" => "article" "crossmark" => 0 "subdocumento" => "fla" "cita" => "Rev Clin Esp. 2014;214:17-23" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:2 [ "total" => 267 "HTML" => 267 ] "en" => array:12 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Original article</span>" "titulo" => "Comprehensive care program for elderly patients over 65 years with hip fracture" "tienePdf" => "en" "tieneTextoCompleto" => "en" "tieneResumen" => array:2 [ 0 => "en" 1 => "es" ] "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "17" "paginaFinal" => "23" ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Programa de atención integral a pacientes mayores de 65 años con fractura de cadera" ] ] "contieneResumen" => array:2 [ "en" => true "es" => true ] "contieneTextoCompleto" => array:1 [ "en" => true ] "contienePdf" => array:1 [ "en" => true ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "A. Fernández-Moyano, R. Fernández-Ojeda, V. Ruiz-Romero, B. García-Benítez, C. Palmero-Palmero, R. Aparicio-Santos" "autores" => array:6 [ 0 => array:2 [ "nombre" => "A." "apellidos" => "Fernández-Moyano" ] 1 => array:2 [ "nombre" => "R." "apellidos" => "Fernández-Ojeda" ] 2 => array:2 [ "nombre" => "V." "apellidos" => "Ruiz-Romero" ] 3 => array:2 [ "nombre" => "B." "apellidos" => "García-Benítez" ] 4 => array:2 [ "nombre" => "C." "apellidos" => "Palmero-Palmero" ] 5 => array:2 [ "nombre" => "R." "apellidos" => "Aparicio-Santos" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S0014256513000714" "doi" => "10.1016/j.rce.2013.01.011" "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/S0014256513000714?idApp=WRCEE" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2254887413000982?idApp=WRCEE" "url" => "/22548874/0000021400000001/v1_201402030902/S2254887413000982/v1_201402030902/en/main.assets" ] "en" => array:18 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Clinical up-date</span>" "titulo" => "Cryptogenia multifocal ulcerous stenosing enteritis: An entity on its own as a cause of abdominal pain, iron deficiency anemia and protein-losing enteropathy" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "26" "paginaFinal" => "30" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "P. Guisado Vasco, G. Fraile Rodríguez" "autores" => array:2 [ 0 => array:4 [ "nombre" => "P." "apellidos" => "Guisado Vasco" "email" => array:1 [ 0 => "pablogvasco@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">¿</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "G." "apellidos" => "Fraile Rodríguez" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Medicina Interna, Hospital Universitario Ramón y Cajal, Madrid, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Úlceras estenosantes, múltiples, recurrentes y crónicas del intestino delgado: una entidad propia como causa de dolor abdominal, anemia ferropénica y enteropatía pierde proteínas" ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Background</span><p id="par0005" class="elsevierStylePara elsevierViewall">A 25-year-old male was admitted to the internal medicine outpatient center for swelling in the legs and periorbital region, general fatigue and recurring bouts of abdominal distension and pain in recent years, with no other accompanying symptoms. Patient stated that he was not taking anti-inflammatory agents or other drugs on a regular basis. He was admitted on several occasions to various emergency departments and was diagnosed with bouts of partial bowel obstruction, which were resolved without surgery. The general laboratory study showed only hypoproteinemia and iron deficiency anemia. There were no inflammatory parameters, and the studies on systemic and intestinal infections, autoimmune diseases, celiac disease, lymphoma and solid tumors were negative. Colonoscopy with ileoscopy and the abdominal computed tomography results were normal. In the oral endoscopy, an ulcerated stenosis was observed in the posterior area of the duodenal bulb, whose pathological study revealed superficial ulceration, along with a slight nonspecific inflammatory infiltrate of the intestinal mucosa. Finally, in the gastrointestinal transit time using barium and capsule endoscopy, we observed several short stenoses of the small intestine and superficial ulcers.</p><p id="par0010" class="elsevierStylePara elsevierViewall">What disease does this patient have that is causing anemia, anasarca and abdominal pain? What are the diagnostic possibilities in this case? What would be the most beneficial treatment? What could surgery provide?</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">The clinical problem</span><p id="par0015" class="elsevierStylePara elsevierViewall">Numerous chronic ulcerations of the small intestine with nonspecific etiology were described in the mid to late 1960s by French and Japanese scientists.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–4</span></a> It is a disease whose incidence and prevalence is unknown. It is currently a rare condition, but its detection is important for clinicians because it presents a number of specific characteristics. The syndrome is defined by the presence of numerous ulcers in the small proximal intestine (ileum, jejunum, and duodenum), which typically measure less than 4<span class="elsevierStyleHsp" style=""></span>cm and have a geographical or linear form. The predominant symptoms are related to anemia, fatigue, weight loss and/or delayed growth, abdominal pain, bowel obstruction conditions and peripheral edema as manifestations of hypoalbuminemia and protein-losing enteropathy. The ulcers affect the mucosa and submucosa but typically do not affect the muscle layer itself. The mucosa shows a nonspecific inflammatory infiltrate of plasma cells, lymphocytes and scarce eosinophils, with an absence of granulomas. A submucous fibrosis process can also be seen in the ulceration area.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Currently, due to the increasingly extensive use of capsule endoscopy<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> and double balloon endoscopy, clinicians can study the small intestine more precisely as a cause of occult gastrointestinal bleeding. In general, vascular causes (telangiectasia, etc.) tend to be the most common, but the presence of ulcers in the intestinal mucosa can also be visualized with these techniques.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Various terms have been used in the scientific literature to name this disease. On occasions some have overlapped others, although all include a number of common characteristics with the terms of cryptogenic enteritis with stenotic ulcers (CMUSE, the term most known in the West) or multiple, nonspecific and chronic ulcers of the small intestine, in its Asian terminology. Its primary differential diagnosis is established with nonsteroidal anti-inflammatory drug (NSAID)-induced enteropathy, Crohn's disease (CD) and intestinal tuberculosis.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Diagnosis</span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Signs and symptoms</span><p id="par0030" class="elsevierStylePara elsevierViewall">The main manifestations of this condition are abdominal pain, fatigue, weight loss, delayed growth and peripheral edema.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,7</span></a> It is important to note the lack of inflammatory parameters in laboratory tests (except for a case reported by Kohotová et al.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a>); however, hypoalbuminemia, hypoproteinemia and iron deficiency anemia are often found.</p><p id="par0035" class="elsevierStylePara elsevierViewall">The presence of extra-abdominal symptoms (fever, dry syndrome, Raynaud's phenomenon, polyneuropathy) has only been reported in isolated cases.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,6,7</span></a> A recessive reticular pigmentary disorder associated with chromosome X (region Xp22-p21, IMO 301220) has been reported in a patient with CMUSE.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">In the cases published in the literature, CMUSE has more points in common than phenotypic differences in patients of Eastern and Western descent. The age at diagnosis is younger in Asian patients, and they therefore have longer follow-up periods. In Western cases, reports indicate that men are more frequently affected and that there are a greater number of intestinal stenoses and cases of abdominal pain.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Endoscopy and imaging tests</span><p id="par0045" class="elsevierStylePara elsevierViewall">The development of capsule endoscopy and double balloon endoscopy allows clinicians to examine the small intestine with greater precision in patients with bleeding of uncertain origin. It is therefore expected that in the coming years the visualization of intestinal lesions using these techniques will increase and that CMUSE will require an accurate differential diagnosis. The technique of functional radiology with oral barium contrast is well documented in the diagnosis of these patients and can be an easier and more economical initial diagnostic, although it probably does not preclude the implementation of other techniques for obtaining tissue samples. However, intestinal lesions can be difficult to recognize, since the ulcers can be very narrow.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The terminal ileum is usually spared, although there are reported cases of its involvement. The involvement of the small intestine at several levels is the most frequently reported. Abdominal CT results are not conclusive for this disease.<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">8–12</span></a> Although capsule endoscopy can diagnose more patients with this disorder, this technique also has potential complications, given that the stenoses are very narrow and short and the capsule can be retained in them.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">The endoscopic appearance of the CMUSE lesions has been well characterized.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,13</span></a> These are linear or triangular erosions or ulcers that occasionally fuse and acquire a geographical configuration. The general rule is to find multiple short, narrow ulcers separated by more than 4<span class="elsevierStyleHsp" style=""></span>cm from each another. The ulcers are also more distal in the small intestinal tract in comparison with NSAID-induced disease.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">14</span></a> The ulcers do not reach the lamina propria. Sometimes the stenoses are not only concentric but can also be oblique or spiral. Finally, findings compatible with a healing phase of the stenoses can be found.</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Histology</span><p id="par0055" class="elsevierStylePara elsevierViewall">Mucosal ulcers are accompanied by a mild inflammatory infiltrate of plasma cells, lymphocytes and eosinophils. Occasionally, lymphoid follicles can be observed. The margins of the ulcer are clearly defined and surrounded by mucosa with no disorders in the villi. A certain submucosal fibrosis can also be observed. The inflammatory infiltrate reaches up to the submucosa but characteristically does not affect the deepest layers. Granulomas, fissures, thrush type ulcers and local adenopathies are not observed.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,7,8,15</span></a></p></span></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Differential diagnosis</span><p id="par0060" class="elsevierStylePara elsevierViewall">Small bowel diaphragm disease (DD or NSAID-associated enteropathy) is an uncommon condition characterized by the presence of numerous circumferential stenotic lesions in a diaphragm shape in the small intestine of patients undergoing prolonged use of NSAIDs.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">16</span></a> These are short annular ulcers, with superficial involvement of the apex of the villi, reactive enterocytes, mild to moderate inflammatory infiltrate and submucosal fibrosis.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">17</span></a> Cryptitis, lymphoplasmacytic infiltrate of the lumen and the presence of eosinophils in the mucosal layer can also be observed. Pathology studies have not identified transmural inflammation, fistulized ulcers or lymphocytic aggregates throughout the muscularis propria layer.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">18</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">The clinical and radiological presentation is similar in both conditions. The differential diagnosis with CMUSE can be established by anamnesis (in the absence of taking NSAIDs), the possible involvement of the colon<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">19</span></a> and the greater age of patients with DD.</p><p id="par0070" class="elsevierStylePara elsevierViewall">Ulcerative and stenotic lesions of the small intestine can also be interpreted as involvement by Crohn's disease (CD). Although the lack of laboratory inflammatory parameters is very suggestive of CMUSE, persistently low CRP levels have been reported in patients with CD,<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">20</span></a> above all in ileal involvement. The arrangement of CMUSE stenoses is circular or oblique, geographical or linear, and the stenoses are short (less than 4<span class="elsevierStyleHsp" style=""></span>cm) and are surrounded by healthy mucosa. However, in CD,<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">21</span></a> the ulcers begin as small red points or depressions in the mucosa, which can deepen and acquire a star distribution or merge into longitudinal ulcers, surrounded by edematous mucosa with a cobblestone appearance. Transmural involvement, fistulization, fissures, giant-cell granuloma<a class="elsevierStyleCrossRef" href="#bib0110"><span class="elsevierStyleSup">22</span></a> and cryptitis have not been observed in the histopathology of CMUSE. The involvement of other gastrointestinal areas and extraintestinal manifestations (uveitis, episcleritis, erythema nodosum, pyoderma gangrenosum, etc.) are more typical of CD. In terms of treatment, CMUSE rarely responds (and then only for a few months) to corticosteroid therapy.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0075" class="elsevierStylePara elsevierViewall">Intestinal infections, especially by <span class="elsevierStyleItalic">Mycobacterium tuberculosis</span>,<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,21</span></a> can cause chronic inflammation of the ileum. Unlike CMUSE, these ulcers have irregular margins, a dense mucosal exudate and several inflammatory stages. These lesions can be accompanied by ascites and/or locoregional adenopathies. Histopathology usually reveals the presence of epithelioid granulomas with central caseous necrosis. The presence of fever and sweating should make us further consider an infectious cause.<a class="elsevierStyleCrossRef" href="#bib0110"><span class="elsevierStyleSup">22</span></a> CMUSE lesions do not respond to treatment with tuberculosis drugs.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0080" class="elsevierStylePara elsevierViewall">In certain patients, ulcerative jejunitis associated with celiac disease<a class="elsevierStyleCrossRefs" href="#bib0115"><span class="elsevierStyleSup">23,24</span></a> and intestinal infections by <span class="elsevierStyleItalic">Campylobacter</span> or <span class="elsevierStyleItalic">Shigella</span> spp. can be also included in the differential diagnosis with CMUSE.<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">21</span></a></p><p id="par0085" class="elsevierStylePara elsevierViewall">Moreover, vasculitis<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> can imitate CMUSE (or have an undemonstrated direct relationship) and, in these cases, can respond well to corticosteroids (both immediately and over time).</p></span><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Etiology</span><p id="par0090" class="elsevierStylePara elsevierViewall">Until a few years ago, data were scarce on the origin and pathogenesis of CMUSE. Perlemuter et al.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> described a case with a type I heterozygous deficiency of the C2 component of complement (genetic deletion of 28 base pairs) in a patient with CMUSE, extraintestinal symptoms and mesenteric aneurysms. These authors proposed the hypothesis that CMUSE is a form of expression of vasculitis (itself or as a variant of polyarteritis nodosa).</p><p id="par0095" class="elsevierStylePara elsevierViewall">The first genetic evidence of the cause of this disease was reported by Adler et al. in 2008.<a class="elsevierStyleCrossRef" href="#bib0125"><span class="elsevierStyleSup">25</span></a> The authors presented the case of a 45-year-old male of Italian descent, with chronic iron deficiency anemia, recurring abdominal pain from childhood and gastrointestinal bleeding of uncertain origin. The patient had multiple stenoses in the jejunum and ileum. The authors suspected that the patient had an inherited prostanoid biosynthesis deficiency, given that the platelet COX-1 activity was normal. In the laboratory studies, they demonstrated a marked deficit of thromboxane A2, leukotriene E4 and B4 and 12-hydroxyeicosatetraenoic acid (12-HETE) in the presence of whole blood activated by calcium ionophore. These findings were consistent with the virtual absence of arachidonic acid (AA) liberation during platelet activation (and, therefore, the platelets also showed reduced total activity). Platelet aggregation and degranulation in response to adenosine diphosphate (ADP) was virtually absent and was corrected by adding AA to the patient's platelets. Following these studies, the authors identified a heterozygous missense mutation in the patient's cPLA2α gene (PLA2G4A; OMIM 600522): S11P (600522.0001) (a rare allele inherited from the mother) and R485H (600522.0002) (a rare variant) and, in the same allele as R485H, a known variant of the SNP allele K651R (inherited from the father). The patient's sister and mother (who were asymptomatic) had intermediate levels of thromboxane B2 and low levels (or at the lower limit of normality) of 12-HETE. Both were heterozygous for one of the mutations found in the patient. An interesting conclusion of this study is that the synthesis of eicosanoids<a class="elsevierStyleCrossRefs" href="#bib0130"><span class="elsevierStyleSup">26,27</span></a> by human platelets and leukocytes results entirely from the cPLA2α pathway and suggests that this phospholipase contributes decisively to the integrity of the small intestine. Other authors have suggested that consanguinity can play a role in CMUSE.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">18</span></a></p><p id="par0100" class="elsevierStylePara elsevierViewall">Recently, Brooke et al.,<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">11</span></a> in 2012, reported 2 new cases with a homozygous mutation of the PLA2G4A gene (deletion of 4 base pairs [g.155574_77delGTAA], in exon 17) in 2 siblings (1 male and 1 female) with signs and symptoms of CMUSE. They also demonstrated that the activity of cPLA2α was virtually absent.</p></span><span id="sec0045" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0065">Treatment and prognosis</span><p id="par0105" class="elsevierStylePara elsevierViewall">Medical therapy (corticosteroids and immunosuppressants) is seldom effective<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,7,28,29</span></a> in patients affected by this disease. Occasionally, control of the disease can be achieved for a few months, although sustained remission for more than 1 year is not common. Surgery can resolve the symptoms in a considerable number of cases, although relapses are not rare, and new laparotomies are required during follow-up of these cases. Nutrition support therapy and treatment of the symptomatic anemia are therapeutic strategies that should always be maintained. NSAIDs should be avoided as they theoretically can cause new outbreaks of this disease. Immunosuppressants and corticosteroids should be used prudently because the side effects (infections, metabolic, etc.) can subject patients to increased morbidity and mortality.</p><p id="par0110" class="elsevierStylePara elsevierViewall">The long-term prognosis is good. The mortality directly attributable to CMUSE is low, although it is unknown whether CMUSE predisposes patients to increased frequency of tumors and other morbidity.</p></span><span id="sec0050" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0070">Areas of uncertainty</span><p id="par0115" class="elsevierStylePara elsevierViewall">There is currently no specific therapy for this condition and to date, we have found no study underway on this topic. Although it has been reported in isolated cases, it is possible that the condition is caused by mutations in the PLA2G4A gene, which cause dysfunction in eicosanoid biosynthesis. Moreover, the terms “cryptogenic” and “nonspecific” do not seem appropriate at present. The scientific terms are confusing in many cases and can also be confused with other diseases. The terminology of chronic, multiple, recurrent and stenosing ulcers of the small intestine is probably more exact for defining CMUSE, in light of current knowledge of this disease.</p></span><span id="sec0055" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0075">Conclusions</span><p id="par0120" class="elsevierStylePara elsevierViewall">After the case was studied, the diagnosis was CMUSE, and the hypoproteinemia and iron deficiency anemia were interpreted as secondary to intestinal losses due to the superficial mucosal ulcers typical of this disease. The type of short stenoses, the lack of inflammation and the histological findings were not consistent with Crohn's disease.<a class="elsevierStyleCrossRefs" href="#bib0150"><span class="elsevierStyleSup">30–32</span></a> Adequate nutrition, iron replacement therapy and low-dose steroid therapy were begun, which did not substantially change the symptoms. The patient was not subjected to any abdominal surgery.</p><p id="par0125" class="elsevierStylePara elsevierViewall">Therefore, CMUSE is a disease with unique characteristics, probably caused by mutations in the PLA2G4A gene, and whose prevalence is unknown. CMUSE should be included in the differential diagnosis of patients with abdominal pain of unknown etiology (both with and without prior surgeries), iron deficiency anemia with intestinal bleeding of unknown origin, malnutrition, edema and protein-losing enteropathy. Medical treatment with corticosteroids and immunosuppressants is seldom effective, and patients require nutrition support measures and, occasionally, surgery to control the symptoms of this disease.</p></span><span id="sec0060" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0080">Authorship</span><p id="par0130" class="elsevierStylePara elsevierViewall"><ul class="elsevierStyleList" id="lis0005"><li class="elsevierStyleListItem" id="lsti0005"><span class="elsevierStyleLabel">1.</span><p id="par0135" class="elsevierStylePara elsevierViewall">Concept and design of the manuscript: P. Guisado and G. Fraile Rodríguez.</p></li><li class="elsevierStyleListItem" id="lsti0010"><span class="elsevierStyleLabel">2.</span><p id="par0140" class="elsevierStylePara elsevierViewall">Data collection: P. Guisado and G. Fraile Rodríguez.</p></li><li class="elsevierStyleListItem" id="lsti0015"><span class="elsevierStyleLabel">3.</span><p id="par0145" class="elsevierStylePara elsevierViewall">Data analysis and interpretation: P. Guisado and G. Fraile Rodríguez.</p></li><li class="elsevierStyleListItem" id="lsti0020"><span class="elsevierStyleLabel">4.</span><p id="par0150" class="elsevierStylePara elsevierViewall">Drafting, review and approval of the submitted manuscript: P. Guisado and G. Fraile Rodríguez.</p></li></ul></p></span><span id="sec0065" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0085">Conflict of interest</span><p id="par0155" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:15 [ 0 => array:2 [ "identificador" => "xres307807" "titulo" => "Abstract" ] 1 => array:2 [ "identificador" => "xpalclavsec290887" "titulo" => "Keywords" ] 2 => array:2 [ "identificador" => "xres307808" "titulo" => "Resumen" ] 3 => array:2 [ "identificador" => "xpalclavsec290888" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Background" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "The clinical problem" ] 6 => array:3 [ "identificador" => "sec0015" "titulo" => "Diagnosis" "secciones" => array:3 [ 0 => array:2 [ "identificador" => "sec0020" "titulo" => "Signs and symptoms" ] 1 => array:2 [ "identificador" => "sec0025" "titulo" => "Endoscopy and imaging tests" ] 2 => array:2 [ "identificador" => "sec0030" "titulo" => "Histology" ] ] ] 7 => array:2 [ "identificador" => "sec0035" "titulo" => "Differential diagnosis" ] 8 => array:2 [ "identificador" => "sec0040" "titulo" => "Etiology" ] 9 => array:2 [ "identificador" => "sec0045" "titulo" => "Treatment and prognosis" ] 10 => array:2 [ "identificador" => "sec0050" "titulo" => "Areas of uncertainty" ] 11 => array:2 [ "identificador" => "sec0055" "titulo" => "Conclusions" ] 12 => array:2 [ "identificador" => "sec0060" "titulo" => "Authorship" ] 13 => array:2 [ "identificador" => "sec0065" "titulo" => "Conflict of interest" ] 14 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2013-04-24" "fechaAceptado" => "2013-07-26" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec290887" "palabras" => array:5 [ 0 => "Cryptogenic multifocal ulcerous stenosing enteritis" 1 => "Protein-losing enteropathy" 2 => "Abdominal pain" 3 => "Gastrointestinal bleeding of obscure origin" 4 => "Weight loss" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec290888" "palabras" => array:5 [ 0 => "Enteritis ulcerosa criptogénica estenosante y multifocal" 1 => "Enteropatía pierde proteínas" 2 => "Dolor abdominal" 3 => "Sangrado digestivo de causa oculta" 4 => "Pérdida de peso" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">We studied a patient with edema secondary to protein losing enteropathy, and recurrent bouts of bloating and abdominal pain secondary to intestinal subocclusion episodes. After the clinical study, the patient was diagnosed of cryptogenic multifocal ulcerous stenosing enteritis (CMUSE), that is a rare disease, probably caused by mutations in the gene PLA2G4A, and characterized by multiple short stenosis of the small bowel with superficial ulcers, which do not exceed the submucosa layer. Inflammatory bowel disease (Chron's disease), intestinal tuberculosis and intestinal ulcers secondary to non-steroidal anti-inflammatory drugs are the main differential diagnosis. To sum up, physicians should included CMUSE in the differential diagnosis of recurrent abdominal pain, iron deficiency anemia, occult intestinal bleeding, edema and protein losing enteropathy.</p>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A propósito del estudio de un paciente con anasarca, enteropatía pierde proteínas y dolor abdominal recurrente secundario a episodios de suboclusión intestinal, al que se le diagnostica de enteritis ulcerosa criptogénica, estenosante y multifocal (CMUSE), se revisa esta enfermedad rara y poco conocida, probablemente causada por mutaciones en el gen de PLA2G4A, que se caracteriza por múltiples estenosis cortas del intestino delgado con ulceraciones que no sobrepasan la submucosa. La enfermedad inflamatoria intestinal (enfermedad de Crohn), la tuberculosis intestinal y las ulceraciones intestinales asociadas a la toma de antiinflamatorios no esteroides son los principales diagnósticos diferenciales. En conclusión, CMUSE debería ser incluida en el diagnóstico diferencial del dolor abdominal recurrente, anemia ferropénica con sangrado intestinal oculto, edemas y enteropatía pierde proteínas.</p>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Guisado Vasco P, Fraile Rodríguez G. Úlceras estenosantes, múltiples, recurrentes y crónicas del intestino delgado: una entidad propia como causa de dolor abdominal, anemia ferropénica y enteropatía pierde proteínas. 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