Three Lumens Full of Blood—An Unusual Case of Gastrointestinal Bleeding

Abstract

Question: A 58-year-old man was initially referred to our gastroenterology outpatient clinic for investigation of iron-deficiency anemia and positive fecal occult blood test (FOBT). His medical history was significant for simultaneous pancreas-kidney transplants 9 years earlier in the context of type 1 diabetes, for which he remained on triple immunosuppressive therapy with prednisolone, mycophenolate, and tacrolimus. His transplants were complicated by biopsy-proven chronic antibody-mediated rejection 4 years earlier, for which he received 6 months of intravenous immunoglobulin. There was no history of chronic liver disease or nonsteroidal medication use. Investigations demonstrated iron-deficiency anemia with a hemoglobin of 88 g/L, ferritin 14 μg/L, transferring saturation 5%, and a positive FOBT (1 of 3 samples). The patient underwent further evaluation with gastroscopy and colonoscopy, which were both unremarkable and did not identify a cause for iron-deficiency anemia. One month later, the patient re-presented to the emergency department with several days of melena and symptomatic anemia with a hemoglobin of 78 g/L. The patient underwent repeated gastroscopy, which did not reveal a cause for his symptoms. He received a blood transfusion and was referred for outpatient capsule endoscopy. Several weeks later, the patient represented with 3 days of melena and symptomatic anemia with a hemoglobin level of 45 g/L. The patient underwent an urgent push enteroscopy. What are the differential diagnoses of gastrointestinal bleeding in this patient? Look on page 335 for the answer and see the Gastroenterology website (www.gastrojournal.org) for more information on submitting to Gastro Curbside Consult. A pediatric colonoscope was inserted 60 cm beyond the pylorus. There was no evidence of esophageal varices, esophagitis, or gastric varices. There was no ulceration or stigmata of recent hemorrhage found in the stomach or duodenum. At 55 cm distal to the pylorus, the anastomosis of the pancreatic transplant was encountered. It demonstrated a side-to-side duodenojejunostomy with 2 short blind duodenal limbs and the native efferent jejunal limb (Figure A). At the anastomosis, multiple varices were noted, of which one vessel was actively bleeding (Figure B). Hemostasis was achieved with the injection of adrenaline (2 mL, 1:10,000) and 2 through-the-scope hemostatic clips (Figure C). The patient then underwent a computed tomographic scan with pancreas protocol to characterize the anatomy and vasculature at the duodenojejunal anastomosis. Multiple venous varices were identified that communicated with a distended transplant pancreatic vein (Figure D). The transplant pancreatic vein anastomoses with the proximal right common iliac vein, and therefore the varices were not due to portal venous hypertension. There was no anastomotic stenosis, no venous thrombosis, and no arteriovenous fistulae. The hemoglobin remained stable after endoscopy and the patient was discharged home 3 days later. At outpatient follow-up 2 months after discharge, there was no evidence of further gastrointestinal bleeding and the hemoglobin level was stable. The patient will continue to be closely observed in the outpatient setting, with a view to endovascular intervention if further gastrointestinal bleeding occurs. Ectopic variceal bleeding from anastomotic varices between the donor duodenum and recipient small bowel are rare, with only 3 cases reported in the literature.1Gopal J.P. Jackson J.E. Palmer A. Taube D. Rathnasamy Muthusamy A.S. Gastrointestinal bleeding in a pancreas transplant recipient: a case to remember.Am J Case Rep. 2020; 21e923197Crossref PubMed Scopus (3) Google Scholar, 2Rostambeigi N. Shrestha P. Dunn T.B. et al.Recurrent ectopic variceal bleed after pancreas transplantation with no portal hypertension: case report and outcomes of endovascular onyx embolization.Vasc Endovasc Surg. 2019; 53: 415-419Crossref PubMed Scopus (5) Google Scholar, 3Fontana I. Bertocchi M. Di Domenico S. et al.Percutaneous embolization of periduodenal varix due to portal hypertension in a patient with kidney-pancreas transplantation: a case report.Transplant Proc. 2010; 42: 2162-2163Crossref PubMed Scopus (5) Google Scholar The mechanism by which ectopic varices develop after pancreas transplantation are not clear. Potential explanations include portal hypertension secondary to cirrhosis, increased vascular resistance, rejection of pancreatic transplant, and chronic venous obstruction. In our case, the mechanism was not elucidated.