Abstract

Introduction: A Dieulafoy’s lesion, typically a large caliber submucosal artery that erodes gastrointestinal (GI) mucosa, is a less common cause of GI bleeding. While these vascular abnormalities may occur throughout the GI tract, appendiceal Dieulafoy’s lesions are exceedingly rare, with described cases often resulting in surgical intervention. We present a case of a Dieulafoy’s lesion in the appendiceal orifice leading to massive GI bleeding which was successfully diagnosed and treated endoscopically. Case Description/Methods: A 75-year-old female with end stage renal disease, hypertension, hyperlipidemia, and heart failure with preserved ejection fraction presented with abdominal pain and rectal bleeding for 2 weeks duration. At onset, she was admitted to an outside hospital where computed tomography (CT) angiography showed aortoiliac and mesenteric atherosclerosis without evidence of large vessel occlusion. Esophagogastroduodenoscopy did not reveal a bleeding source. She reported infraumbilical abdominal pain and continued rectal bleeding and was transferred to our medical center. Upon arrival, the patient was hemodynamically stable with active rectal bleeding on exam. Laboratory analysis revealed hemoglobin of 6.9 grams per deciliter, platelet count of 101 per milliliter, blood urea nitrogen of 6.6 milligrams/deciliter (mg/dl), and creatinine of 6 mg/dl. She was treated with 3 units of packed red blood cell and a proton pump inhibitor. Colonoscopy showed a Dieulafoy’s lesion at the appendiceal orifice. Hemostasis was achieved with placement of 2 hemoclips (Figure). She was discharged 6 days after colonoscopy without recurrence of bleeding. Discussion: Only 6 cases of appendiceal Dieulafoy’s lesions have been reported in the literature and all were treated with laparoscopic appendectomy (Table 1). To our knowledge, this is the first reported case of an appendiceal Dieulafoy’s lesion that was successfully treated with endoscopic placement of hemoclips. There is no data comparing the efficacy of endoscopic intervention versus laparoscopic appendectomy in treating appendiceal Dieulafoy’s lesions; however, this case highlights that therapeutic endoscopy may be both safe and effective. Further reports are needed to inform recognition and optimal approach to appendiceal Dieulafoy’s lesions. Furthermore, in cases where hemostasis is achieved endoscopically, longer term follow-up may inform if appendectomy can be safely avoided.Figure 1.: Successful hemostasis of a Dieulafoy’s lesion at the appendiceal orifice after placement of 2 hemoclips. Table 1. - Previously reported Appendiceal Dieulafoy’s Lesions% and Outcomes Case Report Patient Clinical presentation and course Endoscopic hemostasis attempted? Management? Xue et al, 2020 21 F Massive hematochezia with lower abdominal pain and LOC* N Laparoscopic appendectomy Choi et al, 2016 72 M Hematochezia with associated mild abdominal pain and bloating sensation, previous melena from duodenal ulcer N Laparoscopic appendectomy and cecum wedge resection Johnson et al, 2014 51 M RLQ# pain, Acute appendicitis with incidental finding of Dieulafoy’s lesion on mid-distal appendiceal wall N Laparoscopic appendectomy Reynolds et al, 2013 68 M Massive hematochezia with perfusion requirement N Laparoscopic appendectomy Lee et al, 2011 22 M Severe lower GI† bleed N Laparoscopic appendectomy So et al, 1995 42 M Melena and dizziness N Laparoscopic appendectomy %In all previously reported cases, a Dieulafoy’s lesion was diagnosed using colonoscopy to visualize blood emerging from the appendiceal orifice, then subsequent resection revealing an ulcerated appendiceal mucosal lesion and microscopy and histopathology demonstrating tortuous vasculature penetrating the circumferential and longitudinal muscular wall of the appendix. Abbreviations: *Loss of consciousness, #Right lower quadrant, †Gastrointestinal.

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