Evaluation of the Small Bowel with Video Capsule Endoscopy in Patients with Hereditary Hemorrhagic Telangiectasia: A Systematic Review and Meta-Analysis

Abstract

Introduction: Hereditary Hemorrhagic Telangiecstasia (HHT) also known as Osler-Weber-Rendu syndrome, an autosomal dominant condition, has been associated with mucocutaneous bleeding, pulmonary, cerebral and gastrointestinal arteriovenous malformations (AVMs). This disease has been classified into two groups: HHT1 and HHT2 based on different genetic mutations. Diagnosis of AVMs in the small bowel has involved double-balloon enteroscopy but the use of video capsule endoscopy as a safe and noninvasive procedure has been poorly understood in this population. We sought to evaluate the utility of capsule endoscopy in the diagnosis of AVMs in these patients in addition to characterizing relationships of small bowel AVMs and other clinical features in patients with HHT. Methods: A meta-analysis and systemic review of the literature was performed using MEDLINE, PubMED, and Cochrane databases to search for studies involving video capsule endoscopy in patients with HHT with dates ranging from database inception to June 2016. Our search revealed five studies that were suitable and included in our evaluation. Summary effects was estimated using a fixed effects model. Results: Five studies with a total of 129 patients were included in our meta-analysis. Video capsule endoscopy detected AVMs in approximately 68.9% of patients (p-value of 0.0005). Epistaxis and small bowel AVMs were found together in 74.4% of patients (p-value 0.006). Hepatic AVMs were associated with the presence of small bowel AVMs in 82.5% of patients (p-value 0.005). The occurance of Pulmonary AVMs and small bowel AVMs was not statistically significant (p-value 0.291). A separate meta-analysis was performed to identify defining characteristics in patients with HHT1 and HHT2 with EGD. Gastric AVMs were detected in 66% of HHT1 patients (p-value 0.196) and 43.4% of HHT2 patients (p-value 0.537). Conclusion: Video capsule endoscopy is a sufficient, non-invasive method in detecting small bowel AVMs in patients with Hereditary Hemorrhagic Telangiectasia. Epistaxis or hepatic AVMs were found to have an association with the presence of small bowel AVMs.