An enigmatic enteroscopic image in a patient with upper gastrointestinal bleeding
Article information
Quiz
She was recently discharged from the hospital after surgical resection of the main biliary tract with biliodigestive anastomosis on a Roux-en-Y loop for hilar cholangiocarcinoma Bismuth type II (pT2, G2, N1). The postoperative course was regular, and the patient was discharged 10 days after surgery. At presentation, the hemoglobin level had dropped to 8.3 g/dL, with an overall decrease of 3.0 g/dL after discharge. However, the hemodynamics were stable and abdominal examination was unremarkable.
Contrast-enhanced abdominal computed tomography (CT) performed on the same day revealed no active bleeding spots, surgical dehiscence, or fluid collection. In search of anastomotic bleeding, an anterograde double-balloon enteroscopy (a-DBE) was performed (Supplementary Video 1) within 24 hours. When moving up along the afferent loop, an increase in blood traces was seen. At the end of the mucosal lining, the expected hepaticojejunal anastomosis was covered by an enigmatic amorphous tissue of uncertain origin, with underlying oozing bleeding, floating back and forth in conjunction with endoscopic aspiration and insufflation maneuvers (Supplementary Video 1, Fig. 1). The lesion highlighted by the a-DBE (Supplementary Video 1, Fig. 1) was likely responsible for bleeding. What is it?
Two enteroscopic images of an enigmatic amorphous tissue of uncertain origin, with underlying oozing bleeding, at the site of the hepatico-jejunal anastomosis.
Answer
Based on the a-DBE findings, the patient was urgently referred to the interventional radiology room, where dehiscence of the biliodigestive anastomotic profile was highlighted. Pre-procedural transabdominal ultrasonography was performed first to quickly and non-invasively obtain a real-time assessment of the patient’s anatomy. Through this process, a 3-cm pseudoaneurysm of the hepatic artery propria was documented (Fig. 2A). The pseudoaneurysm was confirmed by arteriography (Fig. 2B) and corresponded in shape, size, and location to the structure visualized on the a-DBE. On the retrospectively reviewed contrast-enhanced CT images, a pseudoaneurysm was not evident. This indicates that at the time the CT scan was performed, the hepatic artery propria was actively bleeding (as suggested by the clinical presentation), and the coagulation process that subsequently allowed the formation of the pseudoaneurysm wall was incomplete.
(A) Pre-procedural ultrasonography documenting a voluminous pseudoaneurysm of the hepatic artery propria with the pathognomonic “Yin and Yang sign”, determined by the typical inflow and outflow of blood as a swirling motion in the sac through the neck. (B) Arteriography confirming the presence of a pseudoaneurysm of the hepatic artery, corresponding in shape, size, and location with the structure visualized at anterograde double-balloon enteroscopy. (C) Arteriography with embolization of the gastroduodenal artery. (D) Arteriography with stenting of the hepatic artery and subsequent exclusion of the pseudoaneurysm.
External percutaneous transhepatic biliary drainage was performed after dehiscence of the biliodigestive anastomosis was confirmed. The hepatic artery propria pseudoaneurysm was treated by the embolization of the gastroduodenal artery (Fig. 2C) and subsequent stenting of the hepatic artery, excluding the pseudoaneurysm (Fig. 2D). Six months after the event, the patient was in good general condition and received adjuvant chemotherapy with capecitabine.
Splanchnic pseudoaneurysms are rare causes of gastrointestinal bleeding. Their most frequent location is the splenic artery, followed by the hepatic artery (most frequently in the right branch), with hepatobiliary surgery being a possible cause.1,2 Pseudoaneurysms must be treated immediately due to the fact that their rate of rupture is much higher than that of true aneurysms.2 Treatment options include surgical, endovascular (embolization, stent placement) or percutaneous (thrombin injection) interventions.2 In high-expertise centers, endoscopic ultrasound-guided embolization can be attempted for refractory cases.3
In conclusion, endoscopic evidence of an amorphous, pulsating anatomic structure at break in continuity with the gastrointestinal wall should raise suspicions of a visceral artery pseudoaneurysm, especially in the presence of compatible previous surgery, procedure or medical illness, such as recent surgery or severe pancreatitis.
Supplementary Material
Supplementary Video 1.
Anterograde double-balloon enteroscopy revealed oozing bleeding at the end of the afferent limb, where amorphous tissue of unknown extraluminal origin was floating within the enteric lumen covering the anastomotic site.
Supplementary materials related to this article can be found online at https://doi.org/ce.2024.138.
Notes
Conflicts of Interest
There are no conflicts of interest to declare.
Funding
This study was partially funded by Italian Ministry of Health, Current research IRCCS.
Author Contributions
Conceptualization: GET, TP; Data curation: TP, BM; Supervision: AMI, GET; Writing–original draft: TP, GET; Writing–review & editing: all authors.