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Una paciente de sexo femenino, adulta joven se internó con diagnóstico de hemorragia digestiva y descompensación hemodinámica. La colonoscopia mostró sangrado desde el ostium apendicular. Se realizó apendicectomía, con resolución de su cuadro de hemorragia digestiva y buena evolución posoperatoria. La anatomía patológica mostró angiodisplasia del apéndice cecal. Se realiza una revisión bibliográfica de esta rara patología y se hacen consideraciones sobre su frecuencia, forma de estudio y posibilidades terapéuticas. An adult, female patient was admitted with diagnosis of digestive bleeding and hemodynamic instability. Colonoscopy showed bleeding from the appendicular ostium. An appendectomy was performed, with resolution of the digestive hemorrhage and good postoperative outcome. Pathology showed angiodysplasia of the cecal appendix. A bibliographical revision is done with considerations on the frequency, diagnostic workup and alternative therapies.

This case study describes a 71-year-old man with signet-ring cell gastric adenocarcinoma and malignant sigmoidal polyp; and typical features of Saint’s triad and Heyde syndrome. He had digestive bleeding, two types of hernia, diverticulosis, arterial hypertension, malignant polyp, and antecedent of smoking, lung tuberculosis, and surgical correction of aortic valve stenosis. There is a hypothetical inverse relationship between herniosis and development of malignancy; however, the patient herein described presented gastric and sigmoidal cancers. Gastrointestinal malignancies are sometimes associated with paraneoplastic entities, isolated or manifested as syndromes, but neither Saint’s triad or Heyde syndrome have been included. This patient persisted clinically stable during the preoperative period, but suddenly died; Trousseau’s syndrome would be the most probable mechanism of sudden death in this setting. Case reports can stimulate further studies to get additional knowledge about unusual entities. Este estudio de caso describe un hombre de 71 años de edad, con adenocarcinoma gástrico con células en anillo de sello y un pólipo maligno sigmoideo; y características típicas de la tríada de Saint y del síndrome de Heyde. Tuvo una hemorragia digestiva, dos tipos de hernias, divertículos, hipertensión arterial, y pólipo maligno; con antecedente de tabaquismo, tuberculosis pulmonar, y corrección quirúrgica de estenosis de la válvula aórtica. Hay una hipotética relación inversa entre hernioses y el desarrollo de malignidades; sin embargo, el paciente que se describe en el presente documento presentó cánceres gástrico y sigmoideo. Neoplasias gastrointestinales se asocian a veces con entidades para neoplásicas aisladas o manifiestan síndromes, pero ni la tríada de Saint ni el síndrome de Heyde se ha incluido. Este paciente persistió clínicamente estable durante el período preoperatorio, pero de repente murió; síndrome de Trousseau sería el mecanismo más probable de muerte súbita en esta situación. Los informes de casos pueden estimular más estudios para obtener un conocimiento adicional sobre esas entidades inusuales.

El síndrome de Heyde es un diagnóstico poco usual, descrito por primera vez en 1958. Es la asociación de sangrado gastrointestinal por angiodisplasia y estenosis aórtica calcificada, siendo ambos procesos degenerativos en conjunto con un déficit adquirido tipo IIA del factor Von Willebrand (FvW). Se presenta el caso de un paciente con historia de disnea y dolor torácico asociado a sangrado gastrointestinal, con posterior diagnóstico de estenosis aórtica, con posterior reemplazo válvula aórtica transcatéter (TAVR).

Submitted by Paula Angeleli Bueno de Camargo null (paula.angeleli@gmail.com) on 2018-03-30T02:02:16Z No. of bitstreams: 1 Tese final.pdf: 2316860 bytes, checksum: 43999803a07375740929937ca3d56f8c (MD5) Approved for entry into archive by ROSANGELA APARECIDA LOBO null (rosangelalobo@btu.unesp.br) on 2018-04-02T19:22:46Z (GMT) No. of bitstreams: 1 camargo_pab_dr_bot_par.pdf: 1008950 bytes, checksum: b8be6c9e1fce22777fef1d5ff3802f31 (MD5) Made available in DSpace on 2018-04-02T19:22:46Z (GMT). No. of bitstreams: 1 camargo_pab_dr_bot_par.pdf: 1008950 bytes, checksum: b8be6c9e1fce22777fef1d5ff3802f31 (MD5) Previous issue date: 2018-03-01 Introdução: A Síndrome de Klippel Trenaunay (SKT) é uma anomalia vascular caracterizada por ser uma doença rara, com prevalência estimada em 1 caso para cada 100.000 pessoas, diagnosticada clinicamente pela presença de pelo menos dois dos seguintes achados: malformação capilar, malformações venosas e hipertrofia dos tecidos afetados. A literatura científica respalda o manejo dos sintomas e tratamento das veias varicosas em alguns casos selecionados. A melhor abordagem terapêutica é crucial para esses pacientes, tornando-se necessário o melhor conhecimento da história natural da doença, o que justifica o estudo do perfil genético desses pacientes, visto que alguns autores revelaram o papel crucial dos microRNAs na regulação da angiogênese, que parece ser o ponto-chave da SKT. Objetivos: Determinar o perfil global de expressão de miRNA’s em pacientes com SKT em comparação com amostras de sangue de pessoas saudáveis, sem a síndrome. Métodos: A amostra foi constituída por pacientes adultos (> 18 anos) com diagnóstico clínico de SKT, acompanhados no Ambulatório de Malformações Vasculares do Serviço de Cirurgia Vascular e Endovascular do Hospital das Clínicas de Botucatu da Faculdade de Medicina de Botucatu/ UNESP. Como referência, foram utilizadas amostras de sangue de adultos saudáveis (> 18anos), doadores de sangue do Hemocentro da Faculdade de Medicina de Botucatu/ UNESP Botucatu. Resultados: Entre os pacientes com SKT, foram encontrados 3 miRNA com expressão diminuída em comparação com os controles: hsa-miR-320a, hsa-miR-486-5p e hsa-miR-574-3p. Através de busca em bases de dados genéticas, observou-se que todos estes miRNA’s guardam relação com angiogênese e apenas dois guardam relação com AGGF1, elemento sabidamente envolvido na SKT. Através dessas mesmas buscas observou-se a menção a outros 5 genes como tendo relação com SKT, sendo o ING5 comum a todos os miRNA’s de interesse. Conclusão: Os pacientes com SKT apresentaram alterações em seu perfil global de expressão de miRNA, com 3 miRNA’s apresentando expressão diminuída, relacionados a angiogênese, AGGF1 e outros genes, em especial ING5. Introduction: Klippel Trenaunay Syndrome (KTS) is a vascular malformation characterized by being a rare disease, with a prevalence estimated in 1 case per 100,000 people, diagnosed clinically by the presence of at least two of the following findings: capillary malformation, venous malformations, and hypertrophy of affected tissues. The scientific literature supports the management of symptoms, treatment of varicose veins in selected cases and, rarely, amputations. The best therapeutic approach is crucial for these patients, making it necessary to have a better knowledge of the natural history of the disease, which justifies the study of the genetic profile of these patients, since some authors have revealed the crucial role of microRNAs in the regulation of angiogenesis, which seems to be the key point of KTS. Objectives: To determine the overall expression profile of miRNA’s in patients with KTS compared to blood samples from healthy people without the syndrome. Methods: The sample consisted of adult patients (> 18 years) with clinical diagnosis of KTS, followed at the Vascular Malformation Outpatient Service of the Vascular and Endovascular Surgery Service of Botucatu Medical School, Botucatu Medical School / UNESP. Blood samples from healthy adults (> 18 years old), blood donors from the Botucatu Medical School / UNESP Botucatu Blood Center were used as reference. Results: Among the patients with SKT, 3 miRNA’s with decreased expression were found compared to subjects without the disease: hsa-miR-320a, hsa-miR-486-5p and hsa-miR-574-3p. Through search in genetic databases, it was observed that all these miRNA are related to angiogenesis and only two closely relate to AGGF1, elements known to be involved in SKT. Through these same searches, mention was made of the other 5 genes as being related to KTS, with ING5 being common to all miRNA’s of interest. Conclusion: Patients with KTS presented alterations in their overall expression profile of miRNA, with 3 miRNA having decreased expression related to angiogenesis, AGGF1 and other genes, especially ING5.

Background. Small bowel hemorrhages are rare and account for 2-10% of all gastrointestinal bleedings. In case that surgery is necessary, identification of the bleeding site is the most important problem. Case report. We presented here the case of a 65-year old man, admitted for urgent care of massive lower gastrointestinal bleeding. After reanimation and normalization of vital parameters, selective arteriography was done. A contrast extravasation site was identified at the level of jejunal branches of a. mesenterica superior and labeled by means of methylene blue application. Immediately after we performed conservative resection of the labeled jejunal loop in 10 cm length and terminoterminal anastomosis. The preparation was sent for histopathologic examination - small bowel angiodysplasia was identified. The patient was monitored in three month intervals in the next two years and new bleeding events were not observed. Conclusion. Bleeding caused by small bowel angiodysplasia is a significant diagnostic problem in cases in whom urgent surgery is required. Combined preoperative selective arteriography and methylene blue application make possible precise identification of the bleeding site as well as conservative small bowel surgery, avoiding thus the risk and danger of malabsorption syndrome.

A utilização da colonoscopia como método de diagnóstico e tratamento possibilitou avanços na abordagem da angiodisplasia de cólon, anomalia vascular responsável por um terço das hemorragias gastrointestinais baixas. Trata-se de paciente com 65 anos, insuficiência renal crônica dialítica e coronariopata grave, apresentando episódios de hematoquezia volumosa ao iniciar uso de agregantes plaquetários, necessitando múltiplas hemotransfusões. Colonoscopia diagnosticou lesões vasculares no ceco, onde existe grande risco de perfuração durante intervenções terapêuticas. Há descrições na literatura do uso de agentes hemostáticos tais como plasma de Argônio e vasoconstrictores, aplicados separadamente. Foi realizado tratamento endoscópico das angiodisplasias com aplicação de Argônio, após elevação das lesões com soro fisiológico, e pela primeira vez descrita no Brasil, acrescentado vasoconstrictor à solução.The utilization of colonoscopy as a diagnostic and treatment tool allowed for advances in the approach of colonic angiodysplasia. This entity is responsible for one third of the cases of lower gastrointestinal bleeding. We discuss a 65 year old patient who suffered from coronary heart disease and chronic renal insufficiency that required dialysis. This patient developed massive hematochezia after starting the use of platelet aggregation inhibitors and required multiple transfusions. The colonoscopy identified vascular lesions in the cecum, where exist a high risk of perforation during therapeutics interventions. There are descriptions in medical literature of the use of hemostatic agents such as argon plasma coagulation and vasoconstrictors used separately. The angiodysplasia was treated endoscopically by the application of argon plasma laser after elevating the lesions with saline and, described for the first time in Brazil, adding vasoconstrictor to the saline solution.

Desde 1960 houve grande avanço tecnológico na observação do aparelho digestivo com o advento dos endoscópios flexíveis, usados inicialmente para o trato digestivo superior e posteriormente também para o inferior, permitindo diagnósticos mais precisos e realização do tratamento em alguns casos. Mesmo em situações de urgência a colonoscopia pode ser utilizada como primeira escolha podendo ser diagnóstica e terapêutica. OBJETIVO: O presente estudo teve por objetivo avaliar quais foram os resultados principais das colonoscopias realizadas no Setor de Endoscopia Digestiva do Hospital Regional de Mato Grosso do Sul. MATERIAIS E MÉTODOS: Foram estudados 290 pacientes submetidos à colonoscopia no Serviço de Endoscopia Digestiva do Hospital Regional do Mato Grosso do Sul, no período de novembro de 2006 a agosto de 2007. Foram analisadas as seguintes variáveis: sexo, idade, caráter de indicação do exame (urgência e eletiva) e diagnóstico. RESULTADOS: A idade média foi de 52 anos, variando de 15 a 92 anos. A maioria dos pacientes era do sexo feminino (56,5%). Dos 290 exames realizados no período foram constatados 48,5% (n=141) de colonoscopias com diagnóstico normal. Dentre os anormais houve uma prevalência de doença diverticular dos colos (n=56), seguido de pólipos (n=37), doenças inflamatórias (n=31), neoplasias (n=18) e angiodisplasia (n=7). Os pólipos apresentaram uma maior concentração em colo sigmóide (n=18) seguido pelo colo descendente e reto. As doenças inflamatórias apresentaram como diagnóstico colonoscópio, colite actínica (n=11) seguida por retite actínica (n=7) e retite inespecífica (n=7). Houve um maior número de procedimentos oriundos do ambulatório (73%) em contraste com internados (20%) e provenientes do Pronto Socorro (7%). CONCLUSÃO: A doença diverticular e os pólipos foram os diagnósticos preponderantes, pois a maior indicação foi a hemorragia digestiva. Uma alta incidência de pólipos em sigmóide demonstrou a importância da retosigmóidoscopia, é um procedimento relativamente seguro e mais barato que a colonoscopia. A baixa incidência de angiodisplasia, resultado discordante da literatura pode ser explicada por uma idade média relativamente baixa, existe mais angiodisplasia no delgado. Since 1960 it there was a great technological breakthrough, including the observation of the digestive system with the advent of flexible endoscopy. Initially used to the upper digestive tract and subsequently to the colon, allowing more accurate diagnoses and, in some cases, the complete treatment. Even in urgency situations, colonoscopy can be used as first choice and work as a diagnostic tool or eve the therapeutic procedure. AIM: This study analyzed the results of colonoscopy held in the Digestive Endoscopy unit at the Hospital Regional Rosa Pedrossian in Mato Grosso do Sul. MATERIALS AND METHODS: It was performed a colonoscopy in 290 patients at the unit of Digestive Endoscopy in the Hospital Regional of Mato Grosso do Sul, in during november 2006 until august 2007. The results were analyzed and verified the following variables: sex, age, solicitation as elective or emergency and diagnosis. RESULTS: The age of the patients ranged from 15 to 92 years old and the mean age was 52 years. Most of the patients were female (56.5%). The colonoscopy tests showed that 48.5% (n=141) were normal. Among the abnormal there were a prevalence of colon diverticulum (n = 56), followed by polyps (n = 37), inflammatory diseases (n = 31), cancer (n = 18) and angiodisplasy (n = 7). Polyps most usual place were in colorectal sigmoid (n = 18) followed by colorectal downward and rectum. The inflammatory diseases were shown as actinic colitis (n = 11) followed by actinic proctitte (n = 7) and nonspecific proctite (n = 7). There was a higher prevalence of procedures from the ambulatory 73%, interns 20% and from the emergency room 7%. CONCLUSION: The predominant diagnosis was diverticulum disease, followed by polyps. Digestive hemorrhage was the main signal noticed by the doctors who asked for the exam. With a high incidence of polyps, and mostly in sigmoid, just evidenced the how important the retosigmoidoscopy is. It is a relatively safe and cheaper procedure in comparison to the colonoscopy and can be performed during the simple consultation. The low incidence of angiodisplasia could be explained by a relatively low average age, differing from literature.

As malformações vasculares representam uma das principais causa de hemorragia digestiva baixa em doentes idosos. O acometimento de indivíduos com menos de trinta anos é evento raramente descrito. OBJETIVO: O objetivo do presente relato é apresentar um caso de angiodisplasia, localizada na região cecal, em doente na terceira década de vida, diagnosticada por meio de arteriografia pré-operatória e confirmada pelo estudo anátomo-patológico. RELATO DO CASO: Homem, 29 anos, com queixa de anemia crônica de origem obscura há vários anos e refratária ao tratamento clínico e transfusional. Com o objetivo de investigar possível origem digestiva foi submetido à arteriografia seletiva abdominal que demonstrou a presença de malformação vascular, localizada na região cecal. Realizou-se colectomia direita onde foi possível confirmar a localização da enfermidade pela arteriografia pós-operatória do espécime extirpado e o diagnóstico definitivo de angiodisplasia pelo estudo anátomo-patológico. Após a intervenção cirúrgica apresentou evolução favorável, estando atualmente no 5º mês pós-operatório sem apresentar sinais de recidiva da anemia. CONCLUSÃO: Não obstante à raridade, a possibilidade de angiodisplasia deverá ser sempre considerada na investigação da anemia de causa obscura em doentes jovens.The vascular malformations represent one of the main causes of lower gastrointestinal bleeding in elderly patients. The incidence of the disease in patients with less than thirty years is a rare described event. The objective of the present report is to show a case of angiodysplasia, located in the cecum region, in-patient on his third decade of life, revealed by preoperative selective angiography and confirmed by histopathological study. Men, 29th years old, presented complaining a chronic anemia of obscure origin for several years and refractory to the clinical and transfusion therapy. With the objective to investigate a possible digestive origin he was submitted to the abdominal selective angiography that demonstrated the presence of vascular malformation located in the cecum. A right colectomy was done and then it was possible to confirm the localization of the disease for postoperative angiography of surgical specimen and the diagnosis of angiodysplasia for the anatomopathologic study. After surgical procedure presented favorable evolution, being currently in the 5th postoperative month without presenting signals of anemia. Despite of an uncommon possibility the presence of angiodysplasia has to be always considered in the clinical investigation of anemia of obscure cause in young patients.

As malformações vasculares representam uma das principais causa de hemorragia digestiva baixa em doentes idosos. O acometimento de indivíduos com menos de trinta anos é evento raramente descrito. OBJETIVO: O objetivo do presente relato é apresentar um caso de angiodisplasia, localizada na região cecal, em doente na terceira década de vida, diagnosticada por meio de arteriografia pré-operatória e confirmada pelo estudo anátomo-patológico. RELATO DO CASO: Homem, 29 anos, com queixa de anemia crônica de origem obscura há vários anos e refratária ao tratamento clínico e transfusional. Com o objetivo de investigar possível origem digestiva foi submetido à arteriografia seletiva abdominal que demonstrou a presença de malformação vascular, localizada na região cecal. Realizou-se colectomia direita onde foi possível confirmar a localização da enfermidade pela arteriografia pós-operatória do espécime extirpado e o diagnóstico definitivo de angiodisplasia pelo estudo anátomo-patológico. Após a intervenção cirúrgica apresentou evolução favorável, estando atualmente no 5º mês pós-operatório sem apresentar sinais de recidiva da anemia. CONCLUSÃO: Não obstante à raridade, a possibilidade de angiodisplasia deverá ser sempre considerada na investigação da anemia de causa obscura em doentes jovens. The vascular malformations represent one of the main causes of lower gastrointestinal bleeding in elderly patients. The incidence of the disease in patients with less than thirty years is a rare described event. The objective of the present report is to show a case of angiodysplasia, located in the cecum region, in-patient on his third decade of life, revealed by preoperative selective angiography and confirmed by histopathological study. Men, 29th years old, presented complaining a chronic anemia of obscure origin for several years and refractory to the clinical and transfusion therapy. With the objective to investigate a possible digestive origin he was submitted to the abdominal selective angiography that demonstrated the presence of vascular malformation located in the cecum. A right colectomy was done and then it was possible to confirm the localization of the disease for postoperative angiography of surgical specimen and the diagnosis of angiodysplasia for the anatomopathologic study. After surgical procedure presented favorable evolution, being currently in the 5th postoperative month without presenting signals of anemia. Despite of an uncommon possibility the presence of angiodysplasia has to be always considered in the clinical investigation of anemia of obscure cause in young patients.

Capsule endoscopy is a diagnostic method of increasing use nowadays, with various applications such as, evaluation of obscure gastrointestinal bleeding, chronic diarrhea and anemia. The experience of 4 years is reported in a retrospective review of 109 medical records to determine the main indications and the most common diagnostic findings and the percentage of positive studies. The main indications were obscure bleeding in 65%, chronic anemia in 8%. Vascular lesions in 33%, being that, hematocistic spot was found in 53% of studies. The location of active bleeding was possible in five patients (4.5%). Capsule endoscopy shows a high diagnostic yield in obscure bleeding. A endoscopia por cápsula é um método cada vez mais empregado na prática clínica, com diversas aplicações tais como: investigação de sangramento gastrointestinal obscuro, diarreia e anemia. A experiência de 4 anos é relatada em revisão retrospectiva de 109 registros médicos objetivando determinar as indicações e achados mais comuns e o percentual de exames positivos. As principais indicações foram: sangramento gastrointestinal obscuro em 65%, anemia crônica em 8%, lesões vasculares em 33% e a mancha hematocística foi observada em 53% dos estudos. A localização do sangramento ativo foi possível em 4,5% dos casos. A endoscopia da cápsula demonstrou bom rendimento diagnóstico no sangramento obscuro.

Introducción: La cápsula colónica, PillCam© Colon fue utilizada por primera vez en el mundo en octubre de 2006 para la exploración no invasiva del colon. Estudios recientes han demostrado su buena rentabilidad diagnóstica, comparada con la colonoscopía convencional, en el estudio de la patología colónica. Objetivo: Determinar el rendimiento diagnóstico de la PillCam© Colon en la detección de lesiones colónicas, comparada con la colonoscopia convencional, en pacientes de la ciudad de Cartagena con sintomatología de los intestinos medio y bajo. Materiales y métodos: Estudio observacional, prospectivo de pruebas diagnósticas, en el cual se evaluó la validez (la sensibilidad, la especificidad y los valores predictivos) de la PillCam© Colon, comparada con la colonoscopia, en la detección de lesiones colónicas, en una cohorte de pacientes que consultaron a la clínica Universitaria San Juan de Dios por sintomatología de los intestinos medio y bajo en Cartagena, durante el periodo junio de 2011-enero de 2013. Los análisis se realizaron utilizando el software STATA 11.0. Resultados: Se estudió a 25 pacientes: 14 mujeres (56%) y 11 hombres (44%); todos ellos, con colonoscopia previa, y a quienes se les practicó una panendoscopia con la PillCam© Colon activada. Se logró la exploración completa del colon en el 76% de los casos (19 pacientes). El tiempo del tránsito colónico fue de 2,4 horas (DE±1,3 horas), y el del tránsito oroanal, de 6,2 horas (DE± 1,18). Los hallazgos de la PillCam© Colon y de la colonoscopia coincidieron en el 78,9% de los casos. La PillCam© Colon mostró lesiones no vistas en la colonoscopia en el 29,2% de los pacientes, con una sensibilidad para la detección de lesiones colónicas del 96% (IC 95% 54,1-100) y una especificidad del 46,2% (IC 95% 19,2-74,9), con un área bajo la curva ROC de 0,731 (IC 95% 0,59-0,87). Conclusión: La PillCam© Colon es una técnica no invasiva, altamente sensible y segura en la detección de lesiones colónicas, y que puede ser de utilidad en la práctica clínica en grupos de alto riesgo para la detección temprana del cáncer colorrectal y en los casos donde la colonoscopia sea incompleta o esté contraindicada Introduction: In October 2006 the PillCam© Colon colonic capsule was used for the first time ever for noninvasive colon. Recent studies have shown that is diagnostic performance is good in comparison with conventional colonoscopy for the study of colonic pathologies. Objective: The objective of this study was to compare the diagnostic yield of PillCam© Colon in detecting colonic lesions with the yield of conventional colonoscopy for patients with symptoms in the mid and lower intestinal regions in a hospital in Cartagena. Materials and methods: This was a prospective observational study of diagnostic tests which assessed the validity (sensitivity, specificity and predictive values) of the PillCam© Colon and compared them with colonoscopy for the detection of colonic lesions in a cohort of patients at the University Clínica Universitaria San Juan de Dios in Cartagena. Patients had symptoms in the mid and lower intestinal regions. Patients were examined from June 2011 to January 2013. Analyses were performed using STATA 11.0 software. Results: We studied 25 patients: 14 women (56 %) and 11 men (44 %). All had previously undergone colonoscopies. Panendoscopies were performed on these patients using an activated PillCam© Colon. We achieved complete colon examinations in 76% (19 patients). Colonic transit time was 2.4 hours (SD ± 1.3 hours) and oral-anal transit time was 6.2 hours (SD ± 1.18). 78.9% of the findings of the PillCam© Colon and colonoscopy agreed. The PillCam© Colon showed lesions not seen in colonoscopy in 29.2 % of patients. Its sensitivity for detecting colonic lesions was 96 % (95% CI 54.1-100) while its specificity was 46.2 % (95% CI 19.2 -74.9). The area under the ROC curve was 0.731 (95% CI .59 to .87). Conclusion: PillCam© Colon is a highly sensitive and reliable non-invasive method for detection of lesions in the colon. It may be useful in clinical practice for early detection of colorectal cancer in high risk groups and in cases that colonoscopy is incomplete or contraindicated

We report the case of a 71-year-old man with diagnosis of aortic valve stenosis for ten years, who came to hospital because of breathlessness during the previous two months and recent low intestinal hemorrhage. On admission, laboratory tests and upper gastrointestinal endoscopy and colonoscopy revealed anemia and bleeding cecal angiodysplasia. The echocardiography study showed a severe aortic stenosis. Classical Heyde syndrome is described as the association of aortic stenosis, bleeding gastrointestinal angiodysplasia and secondary anemia. The antecedent of mediastinal radiotherapy for treatment of Hodgkin´s disease during his youth, and eventual late cardiac adverse effects that may include aortic or mitral valve disturbances are highlighted. Electrocoagulation with argonium was performed on the sites of active bleeding during the colonoscopy. In sequence, surgical replacement by bioprothesis was done on the aortic valve. The patient remains asymptomatic, under long-term outpatient surveillance, with normal control evaluations. The aim of this case study is to emphasize difficulties related to diagnosis, and to highlight the role of endoscopy and imaging studies to confirm a hypothesis of this underestimated condition. Se presenta el caso de un varón de 71 años con estenosis aórtica diagnosticada diez años antes, que acude al hospital por disnea durante dos meses y reciente hemorragia digestiva baja. La analítica y la endoscopia oral y colonoscopia realizadas en el momento de la admisión revelaron anemia y angiodisplasia cecal con sangrado activo. El ecocardiograma mostró estenosis aórtica severa. El síndrome de Heyde clásico está descrito como asociación entre estenosis aórtica y sangrado por angiodisplasia gastrointestinal, con anemia secundaria. Se destaca el antecedente de radioterapia mediastinal para tratamiento de enfermedad de Hodgkin durante su juventud, y eventuales efectos adversos tardíos que pueden incluir trastornos de las válvulas aórtica y mitral. Durante la colonoscopia se realizó la electrocoagulación con argón del punto de sangrado activo. Posteriormente se realizó cirugía de sustitución valvular aórtica utilizando una bioprótesis. El paciente permanece asintomático, realizando un seguimiento ambulatorio a largo plazo, y con exámenes de control normales. El objetivo de este estudio es enfatizar las dificultades relacionadas al diagnóstico, y resaltar el papel de los estudios endoscópicos y de imágenes para confirmar una hipótesis de esta entidad subestimada.

Existe uma associação frequente entre estenose valvar aórtica e sangramento gastrintestinal, também conhecida como síndrome de Heyde. A base fisiopatológica dessa síndrome parece ser uma deficiência adquirida do fator de von Willebrand, que leva ao sangramento de malformações arteriovenosas angiodisplásicas. As alternativas de tratamento incluem a localização de pontos de sangramento e a cauterização, mas tal opção terapêutica está associada a alta recorrência. A substituição da válvula parece oferecer a melhor esperança de resolução a longo prazo do sangramento e deve ser considerada na maioria das vezes. Relatamos aqui o caso de uma paciente de 79 anos com estenose aórtica e sangramento gastrintestinal devido à angiodisplasia duodenal, tratada por implante transcateter de válvula aórtica. There is a frequent association between aortic valve stenosis and gastrointestinal bleeding, also known as Heyde's syndrome. The pathophysiologic basis for this syndrome seems to be an acquired von Willebrand factor deficiency, leading to bleeding of angiodysplastic arteriovenous malformations. Treatment options include the location and cauterization of bleeding points, which is associated to high recurrence rates. Valve replacement appears to offer the best hope of long-term resolution of bleeding, and should be considered in most cases. We report a 79 year-old patient with aortic stenosis and gastrointestinal bleeding due to duodenal angiodysplasia treated by transcatheter aortic valve implantation.

Objectives: to assess the efficacy of thalidomide in the treatment of relapsed or refractory bleeding secondary to gastrointestinal angiodysplasia. Material and methods: we carried out a prospective study of 12 patients with bleeding due to gastrointestinal angiodysplasia refractory to conventional therapy who were treated with thalidomide. For each patient, we considered: age, sex, underlying disease, previous therapies, dose and duration of thalidomide treatment, evolution of haemoglobin levels and adverse effects of treatment. The data obtained were analysed using descriptive statistics with SPSS v. 16. Results: seven men and 5 women with a mean age of 77 years were included in the present study. Five had some underlying pathology and all of them had received prior endoscopic/octreotide treatment. The dose of thalidomide administered was 200 mg/24 h and the duration of the treatment four months, with the exception of two patients in whom treatment was discontinued because of adverse side effects. Mean haemoglobin concentration before onset of treatment was 6.5 g/dL, at two months it was 11.3 g/dL and at the end of treatment 12.1 g/dL. Conclusions: thalidomide is an effective treatment in gastrointestinal bleeding due to angiodysplasia, but it was withdrawn due to side effects in 16% of the patients included in our study. Objetivos: valorar la eficacia de talidomida en la hemorragia recidivante y refractaria por angiodisplasias gastrointestinales. Material y métodos: se realizó un estudio prospectivo de 12 pacientes con hemorragia por angiodisplasias refractarios a la terapia convencional, que recibieron tratamiento con talidomida. Se determinó en cada caso: edad, sexo, patología de base, tratamientos previos, dosis y duración del tratamiento con talidomida, evolución de los valores de hemoglobina y efectos secundarios del tratamiento. Los datos fueron procesados mediante estadística descriptiva con SPSS versión 16. Resultados: se incluyeron 7 hombres y 5 mujeres con una edad media de 77 años; 5/12 presentaban patología de base y todos habían recibido tratamiento previo endoscópico/octreótide. La dosis de talidomida fue de 200 mg/24 horas y la duración del tratamiento cuatro meses, excepto en dos casos que se suspendió precozmente por efectos secundarios. La concentración de hemoglobina media antes de iniciar el tratamiento fue 6,5 g/dl, a los dos meses era 11,3 g/dl y al finalizar fue 12,1 g/dl. Conclusiones: talidomida es una alternativa eficaz en el tratamiento de la hemorragia digestiva por angiodisplasias, aunque se tuvo que retirar por efectos secundarios en un 16% de los pacientes.

A utilização da colonoscopia como método de diagnóstico e tratamento possibilitou avanços na abordagem da angiodisplasia de cólon, anomalia vascular responsável por um terço das hemorragias gastrointestinais baixas. Trata-se de paciente com 65 anos, insuficiência renal crônica dialítica e coronariopata grave, apresentando episódios de hematoquezia volumosa ao iniciar uso de agregantes plaquetários, necessitando múltiplas hemotransfusões. Colonoscopia diagnosticou lesões vasculares no ceco, onde existe grande risco de perfuração durante intervenções terapêuticas. Há descrições na literatura do uso de agentes hemostáticos tais como plasma de Argônio e vasoconstrictores, aplicados separadamente. Foi realizado tratamento endoscópico das angiodisplasias com aplicação de Argônio, após elevação das lesões com soro fisiológico, e pela primeira vez descrita no Brasil, acrescentado vasoconstrictor à solução. The utilization of colonoscopy as a diagnostic and treatment tool allowed for advances in the approach of colonic angiodysplasia. This entity is responsible for one third of the cases of lower gastrointestinal bleeding. We discuss a 65 year old patient who suffered from coronary heart disease and chronic renal insufficiency that required dialysis. This patient developed massive hematochezia after starting the use of platelet aggregation inhibitors and required multiple transfusions. The colonoscopy identified vascular lesions in the cecum, where exist a high risk of perforation during therapeutics interventions. There are descriptions in medical literature of the use of hemostatic agents such as argon plasma coagulation and vasoconstrictors used separately. The angiodysplasia was treated endoscopically by the application of argon plasma laser after elevating the lesions with saline and, described for the first time in Brazil, adding vasoconstrictor to the saline solution.

INTRODUÇÃO: As angiodisplasias são causas raras de hemorragia digestiva aguda. A natureza intermitente da hemorragia e a extensão de tubo digestivo envolvido colocam desafios particulares. OBJECTIVOS: Caracterizar as hemorragias digestivas agudas severas por angiodisplasia. DOENTES E MÉTODOS: Análise retrospectiva das hemorragias digestivas agudas por angiodisplasia admitidas na Unidade de Cuidados Intensivos de Gastrenterologia (UCIGE) dos Hospitais da Universidade de Coimbra, entre 1992 e 2008. RESULTADOS: Incluídos 21 doentes, representando 0,54% das admissões no período considerado. Registou-se predomínio do sexo masculino e idade média de 74 anos (42-92). Todos os doentes apresentavam co-morbilidades e 40% tomavam antiagregantes plaquetares ou anticoagulantes. A hemorragia foi proximal à ampola de Vater em 62% dos casos. Efectuou-se hemostase endoscópica em 18 doentes, que foi eficaz em todos eles. Outras terapêuticas menos utilizadas incluíram a embolização e enterectomia. Não ocorreu recidiva hemorrágica durante os internamentos. A mortalidade cifrou-se em 4,7%, correspondendo a um doente sujeito a laparotomia, falecido no pós-operatório imediato. CONCLUSÕES: A hemorragia digestiva aguda severa por angiodisplasia foi um evento raro na UCIGE. A endoscopia teve um papel fundamental no diagnóstico e na terapêutica. Os resultados corroboram a pertinência da admissão destes casos numa UCIGE, tendo em conta, nomeadamente, a importante comorbilidade que os caracteriza.INTRODUCTION: Angiodysplasias are rare causes of acute gastrointestinal bleeding. Brisk bleeding and extension of the bowel involved are associated with specific challenges. AIM: Descrption of the main features of gastrointestinal bleeding caused by angiodysplasia. PATIENTS AND METHODS: Retrospective analysis of gastrointestinal bleeding episodes caused by angiodysplasia in patients admitted to the Gastroenterology Intensive Care Unit of Hospitais da Universidade de Coimbra, between 1992 and 2008. RESULTS: Twenty-one patients were included, representing 0,54% of all admissions in the unit during the mentioned period. Most patients were men with an average age of 74 years (42-92). All patients had comorbidities; 40% were taking antiplatelet or anticoagulant drugs. Bleeding was proximal to the ampulla of Vater in 62% of the patients. Endoscopic hemostasis was performed and was successful in 18 patients. Other therapies rarely used were angiographic embolization and segmental enterectomy. There was no recurrent bleeding during hospital stay. CONCLUSIONS: Severe gastrointestinal bleeding caused by angiodysplasia was a rare event in the unit. Endoscopy had a major role both in diagnosis and therapy. The results underline the importance of criterious admission in the Gastroenterology Intensive Care Unit, attending to the comorbidities that characterize these patients.

Background: the clinical impact of small-bowel angiodysplasia has not been defined. We present a prospective study to determine the features of individuals with a higher risk of rebleeding or a worse clinical outcome. Patients and methods: thirty patients with angiodysplasia found on CE were included and followed for 12 months. Angiodysplasia were classified by their size as small (≤ 10 mm) or large (> 10 mm). We also studied angiodysplasia lesion numbers in each patient. Rebleeding was defined as a hemoglobin drop of more than 2 g/dl in the absence of melena or hematochezia in the case of occult GI bleeding, or with any or both manifestations. Results: a therapeutic procedure was carried out in 13 patients (43.4%). Individuals with large angiodysplasia had higher transfusion requirements, a higher proportion of therapeutic procedure performed after CE, lower hemoglobin concentration, and a lower rebleeding rate. Patients with ten or more angiodysplasia lesions had also higher transfusion requirements and lower hemoglobin levels, but we found no differences in the number of therapeutic procedures or rebleeding rate between both groups. On follow up rebleeding was detected in 5 patients (16.7%), all of them with small angiodysplasias. Rebleeding was more frequent in patients who did not receive further interventions (23.53 vs. 7.69%; p = 0.037). Conclusions: angiodysplasia size ≥ 10 mm determines a worse clinical impact and more possibilities of receiving a therapeutic procedure. Our findings support that patients with large lesions would benefit from therapeutic interventions with a reduction in rebleeding rate. Introducción: no se ha definido con exactitud el impacto clínico de las angiodisplasias del intestino delgado. Presentamos un estudio prospectivo para determinar las características de los individuos con mayor riesgo de recidiva hemorrágica o peor evolución clínica. Pacientes y métodos: en este estudio se incluyeron treinta pacientes con angiodisplasias, halladas con la cápsula endoscópica, que fueron seguidos durante 12 meses. Las lesiones se clasificaron por su tamaño en pequeñas (≤ 10 mm) o grandes (> 10 mm). Estudiamos también el número de angiodisplasias en cada paciente. La recidiva hemorrágica se definió como una caída en las cifras de hemoglobina de 2 g/dl, en ausencia de melenas o hematoquecia para la hemorragia de origen oscuro o en presencia de cualquiera de estas manifestaciones. Resultados: se realizaron procedimientos terapéuticos en 13 pacientes (43,4%). Los pacientes con angiodisplasias grandes tuvieron mayores requerimientos transfusionales, un mayor número de procedimientos diagnósticos realizados tras la cápsula endoscópica, cifras inferiores de hemoglobina y menor tasa de recidiva hemorrágica. Los pacientes con diez o más angiodisplasias recibieron también más transfusiones y presentaron cifras inferiores de hemoglobina, pero no hubo diferencias en los procedimientos terapéuticos o recidiva hemorrágica entre ambos grupos. En el seguimiento, la recidiva hemorrágica se detectó en 5 pacientes (16,7%), todos con angiodisplasias pequeñas. Esta fue más frecuente en pacientes que no recibieron tratamiento (23,53 vs. 7,69%; p = 0,037). Conclusiones: el tamaño ≥ 10 mm de las angiodisplasias determina un mayor impacto clínico y más posibilidades de recibir tratamiento. Nuestros hallazgos indican que pacientes con lesiones de mayor tamaño se beneficiarían de procedimientos terapéuticos con una reducción de la tasa de recidiva hemorrágica.

El desarrollo de la cápsula endoscópica y la enteroscopia de doble balón ha aumentado las tasas de diagnóstico y tratamiento en la hemorragia digestiva de origen oscuro, en la que las angiodisplasias del intestino delgado son la causa más frecuente. Sin embargo, el 25-40% de los pacientes, no subsidiarios o no respondedores a la terapéutica endoscópica, angiográfica o quirúrgica, pueden tener alto riesgo de resangrado y carecen de tratamiento médico claramente eficaz. La utilidad de la terapia hormonal está cuestionada actualmente, sin olvidar los frecuentes y limitantes efectos secundarios. El octreótido suele ser eficaz en el control de la hemorragia pero no parece la terapia de mantenimiento más óptima. Los beta-bloqueantes no selectivos, de igual manera que en la profilaxis de la hemorragia por hipertensión portal, pueden ser útiles en combinación con otros fármacos. Recientemente, el octreotido LAR, una formulación de liberación retardada que se administra una vez al mes por vía intramuscular, y la talidomida oral, un potente inhibidor de la angiogénesis, han demostrado su eficacia y seguridad a largo plazo en casos graves aislados.

El desarrollo de la cápsula endoscópica y la enteroscopia de doble balón ha aumentado las tasas de diagnóstico y tratamiento en la hemorragia digestiva de origen oscuro, en la que las angiodisplasias del intestino delgado son la causa más frecuente. Sin embargo, el 25-40% de los pacientes, no subsidiarios o no respondedores a la terapéutica endoscópica, angiográfica o quirúrgica, pueden tener alto riesgo de resangrado y carecen de tratamiento médico claramente eficaz. La utilidad de la terapia hormonal está cuestionada actualmente, sin olvidar los frecuentes y limitantes efectos secundarios. El octreótido suele ser eficaz en el control de la hemorragia pero no parece la terapia de mantenimiento más óptima. Los beta-bloqueantes no selectivos, de igual manera que en la profilaxis de la hemorragia por hipertensión portal, pueden ser útiles en combinación con otros fármacos. Recientemente, el octreotido LAR, una formulación de liberación retardada que se administra una vez al mes por vía intramuscular, y la talidomida oral, un potente inhibidor de la angiogénesis, han demostrado su eficacia y seguridad a largo plazo en casos graves aislados. The development of capsule endoscopy and double-balloon enteroscopy has increased diagnostic and therapeutic rates in obscure gastrointestinal hemorrhage, where angiodysplasia of the small bowel is the most frequent cause. Nevertheless, almost 25-40% of patients who are not candidates or do not respond to endoscopic, angiographic, or surgical management may be at high risk of rebleeding, and therefore lack a clearly effective medical therapy. The utility of hormonal therapy remains unclear and is burdened by adverse effects. Subcutaneous octreotide usually controls bleeding but does not seem adequate for mainteinance therapy. Non-selective beta-blockers alone or in combination with other treatments, as in the prophylaxis of portal hypertension variceal bleeding, may be helpful. Recently, octreotide LAR, a depot formulation administered once a month intramuscularly, and oral thalidomide, a powerful inhibitor of angiogenesis, have demonstrated their effectiveness and safety for long-term therapy in anecdotal case reports and deserve further investigation.

BACKGROUND: Angiodysplasia is a distinct mucosal vascular lesion associated with acute or chronic gastrointestinal bleeding. It occurs most frequently in the right colon and is extremely rare. Its etiology is unknown, but theories of its pathogenesis have evolved from its similarity to colonic angiodysplasia and the lesion appears to be associated with renal insufficiency. CASE REPORT: Sixty-five-year-old woman with repeated melena and severe anemia due to angiodysplasia in the first portion of the duodenum. The diagnosis was done by upper endoscopy. As the patient presented repeated gastrointestinal bleeding with hemodynamic instability and recurrent anemia, surgery was indicated. At laparotomy the lesion was identified and resected with an Y-en-Roux reconstruction. The patient went on well at the immediate and late (four years) postoperative periods with no more recurrent gastrointestinal bleeding. CONCLUSION: Although most of the patients suffering from gastrointestinal angiodysplasia goes on well with conservative management, there is a small portion of them that will need a more aggressive approach, as in this case. RACIONAL: Angiodisplasia duodenal é uma lesão vascular distinta da mucosa intestinal associada com sangramento agudo ou crônico. Embora seja extremamente rara no duodeno, ocorre com maior freqüência no cólon direito. Tem etiologia desconhecida, mas sua patogênese parece semelhante com a do cólon. Em alguns casos está associada à insuficiência renal. RELATO DO CASO: Paciente de 65 anos de idade com episódios repetidos de melena e anemia severa devido à presença de lesão angiodisplásica na primeira porção do duodeno, cujo diagnóstico fôra estabelecido por endoscopia digestiva alta. Como ela apresentara-se com episódios repetidos de hemorragia digestiva alta acompanhados de instabilidade hemodinâmica, foi-lhe indicada cirurgia. À laparotomia, identificou-se a lesão, que foi ressecada, sendo realizada reconstrução em Y-de-Roux. Evoluiu bem no período pós-operatório imediato e encontra-se sem novos episódios de sangramento 4 anos após a cirurgia. CONCLUSÃO: Embora a maioria dos pacientes com angiodisplasia gastrintestinal evolua satisfatoriamente com manejo conservador, existe pequena fração que necessitará de abordagem mais agressiva. Neste caso, a paciente apresentou claro benefício da conduta cirúrgica proposta, visto que não apresentou mais episódios de hemorragia digestiva alta.

Aim: to evaluate the utility of double-balloon enteroscopy for small-bowel disease. Design: a prospective study of 50 consecutive enteroscopies performed from December 2004 to July 2005 to analyze diagnoses and treatments. Patients: 44 patients (33 had undergone a previous capsule endoscopy) with indications for obscure digestive hemorrhage, angiodysplasia, Peutz-Jeghers syndrome, ulcer, suspected Crohn's disease, tumors, and refractory celiac disease. Results: we carried out enteroscopy studies in 44 patients by the oral route and, in 6 additional patients, by both the oral and anal routes. We reached the ileon with the oral route in all cases but one (jejunal stenosis), and in 4 cases out of 7 with the anal route, with an average duration of 73 minutes. We found angiodysplasia in 19 cases, as well as NSAID-related enteropathy, Crohn's disease, diverticulosis, and Waldenström's disease. We performed biopsies in 31% of cases with diagnoses of adenocarcinoma, lymphangiectasia secondary to tumor in celiac disease, and Whipple's disease. We treated 19 patients with angiodysplasia (1 to 20 synchronous lesions) with argon, and 4 patients with polyps using polipectomy (sporadic polyps or Peutz-Jeghers syndrome). A retained capsule in one patient with stenosis was removed. Conclusions: double-balloon enteroscopy is a useful and effective technique in the diagnosis and treatment of small intestine diseases, thus complementing capsule endoscopy. More studies are needed to analyze its impact on the management of this condition. Objetivo: valorar la utilidad de la enteroscopia de doble balón en enfermedades de intestino delgado. Diseño: estudio prospectivo descriptivo de 50 enteroscopias consecutivas (diciembre 2004 a julio 2005), analizando diagnóstico y terapéutica. Pacientes: cuarenta y cuatro pacientes (33 con cápsula endoscópica previa) con hemorragia digestiva oculta o lesiones detectadas mediante cápsula (angiodisplasias, pólipos, úlceras, enfermedad de Crohn, celiaca refractaria y tumores). Resultados: se realizó enteroscopia vía oral a 44 pacientes y vía oral más anal a 6. Por vía oral se progresó hasta íleon en todos los casos excepto uno (estenosis yeyunal), por vía anal en 4 de 7 (3 casos con dificultad de paso por Bahuin). La duración media fue 73 minutos. Se detectaron angiodisplasias (19 casos), úlceras en enteropatía por AINE, enfermedad de Crohn, enfermedad de Waldenström, y divertículos. Se realizaron biopsias en yeyuno-íleon en un tercio de los casos (adenocarcinoma, linfangiectasias secundarias a tumor sobre celíaca, enfermedad de Whipple). Se realizó terapéutica con argón a 19 pacientes con angiodisplasias (1 a 20 lesiones), polipectomía a 4 pacientes con pólipos esporádicos o Peutz Jeghers (1 a 15 pólipos) y se extrajo una cápsula endoscópica retenida en una estenosis. Conclusiones: la enteroscopia de doble balón es una técnica útil y eficaz en el diagnóstico y tratamiento de las lesiones del intestino delgado, complementando a la cápsula endoscópica. Hacen falta más estudios para analizar el impacto esperado en el cambio del manejo de estos pacientes.

Journal Article; Review; Obscure gastrointestinal bleeding is still a clinical challenge for gastroenterologists. The recent development of novel technologies for the diagnosis and treatment of different bleeding causes has allowed a better management of patients, but it also determines the need of a deeper comprehension of pathophysiology and the analysis of local expertise in order to develop a rational management algorithm. Obscure gastrointestinal bleeding can be divided in occult, when a positive occult blood fecal test is the main manifestation, and overt, when external sings of bleeding are visible. In this paper we are going to focus on overt gastrointestinal bleeding, describing the physiopathology of the most usual causes, analyzing the diagnostic procedures available, from the most classical to the novel ones, and establishing a standard algorithm which can be adapted depending on the local expertise or availability. Finally, we will review the main therapeutic options for this complex and not so uncommon clinical problem. Yes

A propósito de um caso clínico, os autores fazem uma revisão da literatura sobre angiodisplasia gastrointestinal como causa comum de hemorragia oculta no idoso, sua associação com estenose aórtica e a eficácia da terapêutica estroprogestativa na sua prevenção e tratamento.

There is a frequent association between aortic valve stenosis and gastrointestinal bleeding, also known as Heyde’s syndrome. The pathophysiologic basis for this syndrome seems to be an acquired von Willebrand factor deficiency, leading to bleeding of angiodysplastic arteriovenous malformations. Treatment options include the location and cauterization of bleeding points, which is associated to high recurrence rates. Valve replacement appears to offer the best hope of long-term resolution of bleeding, and should be considered in most cases. We report a 79 year-old patient with aortic stenosis and gastrointestinal bleeding due to duodenal angiodysplasia treated by transcatheter aortic valve implantation.

Introducao A Estenose aortica (EAo) e a patologia valvular adquirida mais frequente em paises desenvolvidos, presente em 4% das pessoas com mais de 85 anos. Pacientes com EAo grave podem ter comorbidades, apresentando maior risco para eventos tromboticos e de sangramento. A Sindrome de Heyde foi descrita em 1958 por E.C. Heyde observando a relacao entre a estenose da valva aortica e sangramento gastrointestinal. Em 1992, Warkentin et al. observaram a perda do multimero de mais alto peso molecular entre [...]