Did angiodysplasia associated with heyde's syndrome disappear spontaneously?: a case report.

BACKGROUND: Heyde's syndrome can be easily overlooked or misjudged in clinical practice because it shares common clinical manifestations with multiple diseases as well as limited accuracy of several corresponding examinations for diagnosing Heyde's triad. Moreover, aortic valve replacement is often delayed in these patients due to the contradiction between anticoagulation and hemostasis. Herein, we present a rare case of atypical Heyde's syndrome. The patient's severe intermittent gastrointestinal bleeding was not completely cured even through a local enterectomy. In the absence of direct evidence of acquired von Willebrand syndrome (AVWS) or angiodysplasia, her long-standing gastrointestinal bleeding was finally stopped after receiving transcatheter aortic valve implantation (TAVI). CASE PRESENTATION: A 64-year-old female suffered from refractory gastrointestinal bleeding and exertional dyspnoea. A local enterectomy was performed owing to persistent hemorrhage and repeated transfusions; subsequently, histological examination revealed angiodysplasia. Heyde's syndrome was not suspected until 3 years later, at which time the patient started bleeding again and was also found to have severe aortic valve stenosis upon echocardiography. TAVI was consequently performed when the patient was in a relatively stable condition even though the predisposition to bleed, but there was no evidence of angiodysplasia and AVWS during angiography at that time. The patient's above symptoms were significantly relieved after TAVI and followed up for 2 years without any significant ischemic or bleeding events. CONCLUSIONS: The visible characteristics of angiodysplasia or a shortage of HMWM-vWFs should not be indispensable for the clinical diagnosis of Heyde's syndrome. Enterectomy could be a bridging therapy for aortic valve replacement in patients with severe hemorrhage, and TAVI may be beneficial for moderate to high surgical-risk patients even if they have a potential risk of bleeding.

Tranexamic acid may be a useful pharmacotherapy for endoscopically resistant small bowel angiodysplasia.

Small bowel angiodysplasia (SBAD) is reported to account for nearly 50% of cases of small bowel bleeding. When SBAD occurs frequently, it is difficult to treat all the angiodysplasias endoscopically, and gastrointestinal bleeding often recurs. Hormone therapy, somatostatin analogs, thalidomide and vascular endothelial growth factor (VEGF)-neutralizing antibodies have been reported to reduce gastrointestinal angiodysplasia (GIAD) bleeding. However, there is no strong evidence to recommend them. Also, there are no guidelines for their use. Hereditary hemorrhagic telangiectasia (HHT) is a hereditary disease caused by abnormalities in VEGF, resulting in multiple GIADs. A treatment guideline has been created for GIAD in HHT, and the use of tranexamic acid, an antifibrinolytic agent, is the first recommendation pharmacotherapy for GIAD with gastrointestinal bleeding that is difficult to treat endoscopically. It has been reported that fibrinolysis is accelerated in GIAD patients who are not HHT, similar to HHT patients. The use of tranexamic acid for gastric antral vascular ectasia in GIAD has been reported to be useful. However, there are very few reports of its use for SBAD. There are concerns with tranexamic acid use regarding the development of thrombosis/embolism, but there are few reports of such side effects. Future clinical trials including tranexamic acid for SBAD are desired.

Gastrointestinal angiodysplasias diagnosed using video capsule endoscopy in 15 dogs.

BACKGROUND: Angiodysplasia (AGD) is rarely diagnosed in dogs with gastrointestinal bleeding (GIB) and is reported in case reports in dogs. OBJECTIVE: Describe signalment, clinical and diagnostic features of dogs with gastrointestinal (GI) AGD diagnosed by video capsule endoscopy (VCE). ANIMALS: Dogs with overt or suspected GIB which underwent VCE. METHODS: Dogs for which a VCE was submitted for overt or suspected GIB from 2016 to 2021 were selected retrospectively. Medical records and full-length VCE recordings where AGDs were initially detected, were reviewed by 2 trained internists. AGD was considered definitive if 2 readers detected it. Signalment, clinical signs, blood work, medications, concurrent diseases, findings of previous conventional endoscopy, and surgical exploration (if applicable) of dogs with AGD were recorded. RESULTS: Definitive AGD was diagnosed in 15 of 291 (5%) dogs (12 males, 3 females). Twelve (80%) had overt GIB, 11 (73%) had hematochezia, and 6 (40%) had microcytic and hypochromic anemia. AGD was missed by conventional endoscopy in 9/9 dogs and exploratory surgery in 3/3 dogs. Thirteen capsules were administered by mouth (1 incomplete study), and 2 via endoscopy directly into the duodenum. AGD was visualized in the stomach of 3 dogs, in the small intestine of 4, and in the colon of 13 dogs. CONCLUSION AND CLINICAL IMPORTANCE: Although rare, AGD should be considered in dogs with suspected GIB after a negative conventional endoscopy or surgical exporation. Video capsuel endoscopy appears to be a sensitive test to identify AGD within the GI tract.

Colonoscopic and Clinical Features of Colonic Angiodysplasia: A Study in 54 Patients.

OBJECTIVES: Colonic angiodysplasia is a rare disease, it is nevertheless a common cause of lower gastrointestinal (GI) bleeding in older adults. The study summarized the colonoscopic and clinical features of colonic angiodysplasia to raise awareness among endoscopists regarding this disease. MATERIALS AND METHODS: We performed a retrospective study of enrolled patients diagnosed with colonic angiodysplasia between September 2013 and April 2022. Clinical and colonoscopic features of the patients with active bleeding were analyzed and compared with those of patients without bleeding. The comparisons were also conducted between the patients with active lower GI bleeding caused by colonic angiodysplasia and those by other diseases. RESULTS: In total, 54 eligible patients were included in this study; 55.55% of the participants were aged over 60 years. Ten patients (3 men and 7 women) with colonic angiodysplasia suffered from active lower GI bleeding, which was mainly located in the left and total colon. The patients with type 2 diabetes mellitus, radiotherapy history, antiplatelet drug use, and multiple lesions were more likely to endure lower GI bleeding. The duration between bleeding and admission was longer in the colonic angiodysplasia group than in the other diseases group ( P = 0.043). In the colonic angiodysplasia group, bleeding relapsed in 3 patients, and the recurrence rate was higher than in the other diseases group ( P < 0.001). CONCLUSION: Endoscopists should perform colonoscopy scrupulously and consider colonic angiodysplasia as a differential diagnosis in patients with lower GI bleeding, especially for older women and adults with chronic diseases, such as type 2 diabetes mellitus.

Special considerations in GI bleeding in VWD patients.

Gastrointestinal (GI) bleeding is an important cause of morbidity and mortality in von Willebrand disease (VWD). It has been noted that GI bleeding caused by angiodysplasia is overrepresented in VWD patients compared to other causes. The bleeding from angiodysplasia is notoriously difficult to treat; recurrences and rebleeds are common. A growing body of basic science evidence demonstrates that von Willebrand factor negatively regulates angiogenesis through multiple pathways. VWD is clinically highly associated with angiodysplasia. The predisposition to angiodysplasia likely accounts for many of the clinical difficulties related to managing GI bleeding in VWD patients. Diagnosis and treatment are challenging with the current tools available, and much further research is needed to further optimize care for these patients with regard to acute treatment, prophylaxis, and adjunctive therapies. In this review we provide an overview of the available literature on GI bleeding in VWD and explore the molecular underpinnings of angiodysplasia-related GI bleeding. Considerations for diagnostic effectiveness are discussed, as well as the natural history and recurrence of these lesions and which therapeutic options are available for acute and prophylactic management.

How to manage lower gastrointestinal bleeding in 2022?

Lower gastrointestinal bleeding (LGIB), originating mainly in the colon, rectum and anus, occurs most often in older patients (7th decade) with co-morbidity, half of whom have coagulation abnormalities due to anti-coagulant or anti-aggregant therapy. In three cases out of four, bleeding regresses spontaneously but can recur in up to one third of patients. The main causes are diverticular disease, vascular disorders (hemorrhoids, angiodysplasia) and colitis. Ten to 15% of patients present in hypovolemic shock. The main problem is to determine the precise location and etiology of bleeding. First-line steps include correction of hemodynamics, correction of coagulation disorders and transfusion, as necessary. Rectal digital examination allows differentiation between melena and hematochezia. In patients with severe LGIB, upper endoscopy can eliminate upper gastro-intestinal bleeding (UGIB). Computerized tomography (CT) angiography can pinpoint the source. If contrast material extravasates, the therapeutic strategy depends on the cause of bleeding and the general status of the patient: therapeutic colonoscopy, arterial embolization and/or surgery. In the absence of severity criteria (Oakland score≤10), ambulatory colonoscopy should be performed within 14 days. Discontinuation of anticoagulant and/or antiplatet therapy should be discussed case by case according to the original indications.

Heyde's syndrome: a systematic review of case reports.

OBJECTIVE: Heyde's syndrome (HS), a rare condition characterised by a unique relationship between severe aortic stenosis and angiodysplasia, is often diagnosed late increasing the risk for a prolonged hospital course and mortality in the elderly. The leading hypothesis explaining the aetiology of HS is acquired von Willebrand syndrome (AVWS) but not all studies support this claim. While individual cases of HS have been reported, here we present the first systematic review of case reports and focus on the prevalence of AVWS. DESIGN: A systematic search was conducted through PubMed/MEDLINE, CINAHL-EBSCO, Web of Science and Google Scholar since inception. The resulting articles were screened by two independent reviewers based on inclusion criteria that the article must be a case report/series or a letter to the editor in English describing HS in an adult patient. RESULTS: Seventy-four articles encompassing 77 cases met the inclusion criteria. The average age was 74.3±9.3 years old with a slight female predominance. The small intestine, especially the jejunum, was the most common location for bleeding origin. Capsule endoscopy and double balloon enteroscopy were superior at identifying bleeding sources than colonoscopy (p=0.0027 and p=0.0095, respectively) and oesophagogastroduodenoscopy (p=0.0006 and p=0.0036, respectively). The mean duration from symptom onset to diagnosis/treatment of HS was 23.8±39 months. Only 27/77 cases provided evidence for AVWS. Surgical and transcutaneous aortic valve replacement (AVR) were superior at preventing rebleeding than non-AVR modalities (p<0.0001). CONCLUSION: Further research is warranted for a stronger understanding and increased awareness of HS, which may hasten diagnosis and optimal management.

Endocuff-assisted push enteroscopy increases the detection of proximal small-bowel gastrointestinal angiodysplasias.

Gastrointestinal angiodysplasias (GIADs) are the most common causes for suspected small bowel bleeding. Fifty percent of GIADs do not need treatment due to bleeding cessation, while 45% have high re-bleeding rates, that significantly impact patient outcome and health resource utilization. We suspected that this high re-bleeding rate occurs because not all lesions are detected with present standard of care. This study evaluates whether device-assisted enteroscopy (DAE) utilizing the Endocuff (EC) device could improve GIAD detection. A retrospective chart review of a prospective data collection was performed from January 2006 to December 2018 at VA Loma Linda Healthcare System (VALLHCS) on both inpatients and outpatients referred for active and chronic suspected small bowel bleeding. The patients were initially monitored for bleeding lesions via video capsule endoscopy (VCE) after negative upper and lower endoscopy. GIADs observed between 0% to 40% small bowel transit time (SBTT) were referred for push enteroscopy (PE) with and without (±) the EC device. Twenty-five consecutive patients underwent PE ± EC. No patient had VCE done after PE ± EC. Using PE-EC, GIADs were detected in 9 of 25 (36%) of patients. Importantly, PE+EC detected GIADs in 23 of 25 (92%) patients. The sum of GIADs detected without EC was 26 ± 0.06 vs. 112 ± 0.2 using EC. The average detection rate for PE without EC was significantly lower (1.04 ± 0.06, mean ± SE) as compared to PE with EC (4.48 ± 0.23, mean ± SE, p<0.0005). Additionally, a positive correlation (r=0.51) between capsule enteroscopy (CE) location of GIADs and SBTT was found. The EC device increases the detection of GIADs in the proximal small bowel. We also reconfirm that the location of bleeding GIADs are within the reach of the push enteroscope (PE). Finally, PE + EC may also reduce GIAD miss rates, which may play a role in the reduction of re-bleeding episodes.

Colonic angiodysplasia: a culprit of obscure gastrointestinal bleeding in a patient with Heyde syndrome.

A 77-year-old woman presented with obscure gastrointestinal bleeding requiring multiple hospitalisations and blood transfusions. The patient underwent repeated investigations over four hospital admissions across a span of two months. These included upper and lower gastrointestinal endoscopy, video capsule endoscopy as well as CT enterography, without definitive localisation or treatment of the source of bleeding. Finally, a technetium-99m-labelled red blood cell scan demonstrated a 'blush' at the proximal transverse colon on delayed imaging. Targeted colonoscopic evaluation showed a subcentimetre angiodysplastic lesion in the corresponding spot at the proximal transverse colon with slow persistent oozing. Endoscopic clips were applied with successful haemostasis. The patient recovered well without further symptom recurrence 5 months postdischarge. We review the literature on colonic angiodysplasias and discuss the diagnostic challenges in obscure gastrointestinal bleeding.

Systemic bevacizumab to facilitate anticoagulation in antiphospholipid syndrome and bleeding gastrointestinal angiodysplasia.

Bleeding gastrointestinal angiodysplasia may occur in patients with vasculitis and can be challenging to treat. We describe the novel use of bevacizumab therapy to treat bleeding gastrointestinal angiodysplasia and severe anemia in a patient with eosinophilic granulomatosis with angiitis complicated by antiphospholipid antibody syndrome requiring indefinite warfarin therapy. Studies confirmed multiple bleeding jejunal angiodysplasias unamenable to endoscopic intervention, and the patient required ongoing support with iron infusions and blood transfusions to maintain a minimally acceptable hemoglobin. Given the severe anemia, need for continued, indefinite antiplatelet and anticoagulation therapy, and failure of standard treatment approaches, the patient was initiated on systemic bevacizumab therapy, on the basis of prior documented success of bevacizumab to manage gastrointestinal telangiectasias in patients with hereditary hemorrhagic telangiectasia. Bevacizumab was highly effective, with rapid resolution of bleeding, normalization of hemoglobin, liberation from hematologic support and no adverse events, including no thromboembolic events. Vascular endothelial growth factor (VEGF-A) rose paradoxically after initiation of bevacizumab and normalized after its discontinuation. Given these findings, use of systemic bevacizumab to manage bleeding angiodysplasia in patients with acquired vascular disorders merits further study.

Rebleeding after hemoclip versus argon plasma coagulation for gastrointestinal angiodysplasias: a retrospective multicenter study.

BACKGROUND: Hemoclips are utilized for treating bleeding gastrointestinal angiodysplastic lesions (GIADs); however, the supporting evidence is limited. AIMS: Our aim is to evaluate the efficacy of hemoclips in preventing bleeding secondary to GIADs compared to argon plasma coagulation (APC). METHODS: This retrospective study included patients with bleeding gastric, small bowel or colonic GIADs that were endoscopically treated between January 2009 and November 2016. Patients that received hemoclips as monotherapy or in combination were compared to a randomly selected similar number of patients treated with APC. RESULTS: We included 157 patients that underwent APC and 141 who received hemoclips. During a median follow-up of 17 months, those with hemoclips had a 32.6% rebleeding vs. 46.5% in the APC group (P = 0.017). On multivariate regression analysis, use of hemoclips was not a significant predictor of rebleeding when compared to APC; hemoclips monotherapy (HR, 0.92; 95% CI, 0.54-1.59) and hemoclips combination (HR, 0.65; 95% CI, 0.41-1.01). When the multivariate analysis was restricted to subjects that resumed antithrombotics after endoscopy, rebleeding risk was lower when hemoclips were used in combination (HR, 0.46; 95% CI, 0.25-0.84) compared to APC. We noted a similar effect in the antithrombotic subgroup even after propensity score matching (HR, 0.51; 95% CI, 0.27-0.95). CONCLUSION: Treatment modality was not a significant predictor of rebleeding when studied for the entire population. However, the risk of rebleeding was lower with hemoclips combination therapy compared to APC in patients that resumed antithrombotic therapy, suggesting a potential role for a combined approach in this subgroup of patients.

Causes of gastrointestinal bleeding in oral anticoagulant users compared to non-users in a population-based study.

BACKGROUND/AIMS: Causes of gastrointestinal bleeding (GIB) in patients on oral anticoagulants (OACs) are not well established. The aims of the study were to compare the causes of GIB in patients on OACs and those not on OAC therapy. METHODS: A nationwide study of all GIB events in patients on OACs in Iceland from 2014-2019 was conducted. Bleeding events were obtained through ICD-10 codes and review of endoscopy databases, confirmed by review of medical records. For comparison, patients not on OACs from previous Icelandic population-based studies were used. RESULTS: Among 752 GIB events in 12,005 patients on OACs, 273 (1.9%) had verified upper and 391 (2.7%) had verified lower GIB. For lower GIB, multivariate analysis showed that OAC users were more likely to have colonic polyps (OR 6.6, 95% CI: 2.4 - 17.8, p < .001) or colorectal cancer (OR 3.7, 95% CI: 2.0 - 7.0, p < .001) but less likely to have ischemic colitis (OR 0.11, 95% CI: 0.04 - 0.26, p < .001). For upper GIB, bleeding from mucosal erosions (OR 4.0 95% CI: 2.5 - 7.9, p < .001) and angiodysplasia (OR 3.6, 95%CI: 1.5 - 8.6, p = .003) were more common in OAC users. CONCLUSIONS: A high proportion of GIB caused by colonic polyps and colorectal cancer among OAC patients indicates that OACs treatment may facilitate cancer diagnosis. The low proportion of ischemic colitis among those on OACs suggests that OACs provide a protective effect against ischemic colitis. OACs seem to increase the bleeding from angiodysplasia and mucosal erosive disease.